Volume 14, No. 2, October 2010 Pan Arab Journal of Official Journal of the Pan Arab Neurosurgical Society

ALGIERS

VIIIth PAN ARAB CONGRESS OF NEUROSURGICAL SOCIETY November 26-29, 2010

Indexed by EMBASE, Excerpta Medicus & WHO Eastern Mediterranean Index Medicus

Official Journal of Pan Arab Neurosurgical Society Volume 14, No. 2 ISSN 1319-6995 October 2010

Patron: HRH Prince Khalid Bin Sultan Bin Abdulaziz

EDITOR-IN-CHIEF ASSOCIATE EDITORS Khalaf Al Moutaery Abdeslam El Khamlichi, Morocco Antoine Nachanakian, Lebanon MANAGING EDITOR

Valerie McCue EDITORIAL BOARD Benaissa Abdennebi, Algeria DESKTOP PUBLISHING Saleem Abdulrauf, Saudi Arabia (USA) Vittoriana Crisera Ahmed Ammar, Egypt Fawaz Assaad, Syria ARABIC ABSTRACTS Yousef Al-Awadi, Kuwait Translation Department Saleh Baeesa, Saudi Arabia Riyadh Military Hospital El Fatih Bashir, Sudan Mohammed Boucetta, Morocco Fady Charbel, Lebanon (USA) Please address all correspondence to: Mohamed Lotfy Ibrahim, Egypt Bermans Iskandar, Lebanon Prof. Khalaf Al Moutaery Mahmoud Z Karmi, Jordan Editor-in-Chief Mohamed Hassan Katramiz, Syria c/o Riyadh Military Hospital Moncef Khaldi, Tunisia Pan Arab Journal of Neurosurgery Abdulkarim Msaddi, UAE P O Box 7897, Riyadh 11159 Waleed Murshid, Saudi Arabia Kingdom of Saudi Arabia Ibrahim Sbeih, Jordan Fax: (966 1) 476 8273 Bassem Sheikh, Saudi Arabia Email: [email protected] Ghassan S Skaf, Lebanon www.panarabneurosurgery.org.sa Mondher Yedeas, Tunisia

ADVISORY BOARD For advertising enquiries, please contact: Issam A Awad, USA Peter Mclaren Black, USA Ms. Valerie McCue Rudolf Fahlbusch, Germany Tel: (966 1) 477 7714/ 479 1000, ext. 25443 Richard G Fessler, USA Fax: (966 1) 476 8273 Michael Gaab, Germany Email: [email protected] Jens Haase, Denmark Patrick W Hitchon, Lebanon (USA) Tetsuo Kanno, Japan Yoko Kato, Japan Douglas Kondziolka, USA Donlin M Long, USA Ashok K Mahapatra, India Aim & Scope of the Journal: The Pan Arab Journal of Neuro- Ossama Al-Mefty, USA surgery aims to stimulate scientific research and associated Wai Sang Poon, Hong Kong communication among hospitals and universities throughout the Albert Rhoton, USA Arab world by publishing peer-reviewed, original articles highlighting Concezio Di Rocco, Italy current advances throughout the region in Neurosurgery and Marc Sindou, France related sciences. Manuscripts from throughout the world are also Robert F Spetzler, USA published, as well as summaries of any MD thesis successfully com- Dietmar Stolke, Germany pleted in any Arab University. In addition, an Academic Calendar is Alexander R Vaccaro, USA included in order to inform subscribers of any upcoming related sym- Andreas Weidner, Germany posia both throughout the region and abroad. M Gazi Yasargil, USA ii PAN ARAB JOURNAL OF NEUROSURGERY VOLUME 14, NO. 2, OCTOBER 2010 iii iv PAN ARAB JOURNAL OF NEUROSURGERY Editorial

Message from PANS President Elect & 8th PANS Congress President

Dear Colleagues,

In November of this year, Algiers will have the privilege to be the host city for the 8th Pan Arab Neurosurgical Congress. I seize this opportunity to express my sincere gratitude to the members of the Executive Committee of our society who put their trust in us.

It is in the nature of things that topics chosen for this meeting should be in harmony with the state of the art in neurosurgery but also taking into account our daily preoccupations. Surgical techniques such as surgery in awake patients, cortical mapping in surgery of eloquent areas, epilepsy, Parkinson’s disease, and endoscopy are more and more practised and will be covered in this meeting. Speakers will also share their knowledge on spinal, paediatric and vascular surgeries. Obviously, the Scientific Programme will include a session of “free papers“. In connection with these sessions, teachers will focus on “How I do it?” their experience in surgical approaches whereas others will tell us about “What is new?” in neurosurgery. Finally, our female colleagues will hold a round table conference to discuss their own problems.

A meeting of the Executive Committee will be held where we will debate a pre established agenda.

On the other hand, I would like to highlight the prominent part of the first pillar of our society, the Pan Arab Journal of Neurosurgery. Its regular issue and high scientific level of articles, not only from the Arab countries but also from all over the world, give evidence to the importance and the current place of the Arab neurosurgical society.

I am sure that with the enthusiasm of all the attendees, the choice of the topics covered, the beauty of Algiers, ”the white city”, will all contribute to attendees experiencing an interesting and memorable congress. With the efforts of everybody, I wish the best for our society. As the proverb says “Where there is a will, there is a way”.

Prof. Benaissa Abdennebi PANS President Elect 8th PANS Congress President Chief, Department of Neurosurgery Salim Zemirli Hospital El Harach Algiers Algeria

VOLUME 14, NO. 2, OCTOBER 2010 ix Case Review

Intracranial hypotension

Makarand Kulkarni1, Sanjay Mongia2, K Ravishankar3, Vinay Chauhan3, Manoj Deshmukh1

Abstract: Intracranial hypotension is a clinical condition which mostly presents with orthostatic headache. It is caused by CSF leak through dura. There is varied clinical presentation and aetiology of the condition. Magnetic resonance imaging is one of the most important tools in the diagnosis of this condition. We report 5 cases of intracranial hypotension with different aetiology and clinical presentation. (p104-107)

Key words: Intracranial hypotension and orthostatic headache.

Introduction Intracranial hypotension (ICH) is characterised by cerebro- more so in sitting position. The initial magnetic resonance spinal fluid (CSF) leak leading to reduced CSF pressure imaging (MRI) was normal but was repeated after 15 days, within the cranium. It has a diverse aetiology varying from as the headache worsened. There was bilateral, thin subdural spontaneous(9), post (13), post spinal surgery collection with diffuse pachymeningeal enhancement (Figs. (7), post traumatic CSF leak, thoracomeningeal fistula, after 1 and 2). VP shunt and leakage of the CSF through spinal meningeal cyst.1,9 There is a case report of ICH due to transdural thoracic disc herniation.14 There can be medical causes of this condition which include dehydration, diabetic coma and uraemia.

Clinically it is characterised by orthostatic headache. Apart from headache, patient may present with nausea, vomiting, anorexia and neck pain. There may be diplopia, tinnitus, vertigo and galactorrhea.8

We report 5 cases of different aetiology and clinical presen- Figure 1 ↑ T2W image showing thin subdural collection over bilateral cerebral surface. Figure 2 → T1W post contrast im- tation. Four of our patients presented with postural headache age showing diffuse pachymeningeal enhancement. while one presented with only diplopia. Two patients had spontaneous onset, one patient was post traumatic, one post Magnetic resonance imaging of spine was normal. The spinal surgery and one had rupture of perineural cyst in the patient was treated conservatively with bed rest, hydration pleural space. and oral theophylline, with regression of clinical symptoms. Follow-up MRI after 5 months showed completely resolved Case Report subdural collection. This was a case of spontaneous ICH Case 1: Thirty-seven-year-old male presented with headache, and illustrates delay in the appearance of MR features.

Case 2: Sixty-five-year-old female presented with sudden

1 onset of severe headache. Initial clinical picture was Consultant Radiologist 2Consultant Neurosurgeon suggestive of subarachnoid haemorrhage and hence 3Consultant Neurologist computerised tomography (CT) and MRI were done which Lilavati Hospital and Research Centre Mumbai were negative. Lumbar puncture was done with opening India CSF pressure with 11 cm of water. There were no cells in

Correspondence: the CSF. With these negative findings she was diagnosed as Dr. Sanjay Mongia migraine. Her clinical symptoms worsened and the Consultant Neurosurgeon headache now related more with sitting position. Magnetic Lilavati Hospital and Research Centre Mumbai resonance imaging was repeated with screening of entire India spine showing thin bilateral subdural collection (Fig. 3). Email: [email protected] The post contrast MRI showed diffuse pachymeningeal

104 PAN ARAB JOURNAL OF NEUROSURGERY INTRACRANIAL HYPOTENSION • Mongia, et al

relieved only in head low position. Magnetic resonance imaging showed thin subdural collection over bilateral cerebellar surface with diffuse dural enhancement on post contrast study (Figs. 10 and 11). The patient responded to conservative therapy with bed rest in head low position and oral theophylline. Physicians should be aware of this complication after spinal surgery.7

Figure 3 ↑ T2W image showing thin bilateral subdural collection. Figure 4 → Post contrast T1W image showing diffuse pachy- meningeal enhancement.

Figure 7 ↑ Constructive interference in steady-state sequence showing CSF leak from the floor of anterior cranial fossa. Figure 8 → T2W coronal images at the level of pituitary. There is reduced distance between optic chiasm and pituitary.

Figure 5 ↑ Sagittal T1W image showing prominent pituitary with reduced distance between optic chiasm and pituitary. Figure 6 → T2 coronal image of dorsal spine showing a right-sided peri- neural cyst at D10-11 level with a large right-sided pleural effusion. enhancement (Fig. 4). The was mildly prominent with reduced distance between optic chiasm and Figure 9 ↑ T2W sagittal images showing descent of iter (a line pituitary gland (Fig. 5). The spine screening showed a large is drawn from the tuberculum sella to the beginning of straight perineural cyst in the right-sided neural foramen between sinus. The aqueduct is below this line). Figure 10 → T2W D10 and D11 level with a large right-sided pleural effusion images showing thin subdural collection over the cerebellar surface. (Fig. 6).

In this patient, the mechanism of ICH was spontaneous rupture of perineural cyst in the pleural space resulting in hypo- tension. The patient after being treated conservatively with bed rest, hydration and oral theophylline had regression of clinical symptoms.

Case 3: Forty-one-year-old male presented with post head injury CSF leak and severe postural headache. Magnetic resonance imaging showing site of CSF leak (Fig. 7). There Figure 11 - Post contrast T1W images showing enhancing dura over the cerebellum. was descent of brain with inferior displacement of optic chiasm (Fig. 8) and descent of iter (Fig. 9). Surgical repair of the site of CSF leak was done by transsphenoidal route Case 5: Sixty-five-year-old female presented with spon- with complete recovery of the clinical symptoms of ICH. taneous onset of diplopia. The symptoms were more in sitting position however, there was no headache. The MRI Case 4: Sixty-four-year-old female underwent lumbar showed diffuse pachymeningeal enhancement on post con- decompression surgery. On fourth postoperative day she trast study (Fig. 12). The patient recovered with bed rest and developed severe postural headache which could be hydration.

VOLUME 14, NO. 2, OCTOBER 2010 105 INTRACRANIAL HYPOTENSION • Mongia, et al

Figure 12 - Post contrast T1W images showing diffuse dural enhancement.

Table 1 - Summary of cases. Case No. Age Sex Clinical Presentation Aetiology MRI findings Treatment 1 37 M Headache in sitting position Spontaneous Thin bilateral subdural collection with Conservative with oral pachymeningeal enhancement theophylline 2 65 F Headache, in the beginning Spontaneous Thin bilateral subdural collection with Conservative with oral not postural but subsequently pachymeningeal enhancement, theophylline postural reduced suprasellar cistern and perineural cyst in dorsal spine with right pleural effusion 3 47 M Postural headache Traumatic Cerebrospinal fluid leak through anterior Cerebrospinal fluid leak cranial fossa, effacement of suprasellar repaired by transsphenoidal cistern and descent of iter route 4 64 F Severe postural headache Post spinal surgery Thin subdural collection over cerebellar Conservative with oral surface with dural enhancement theophylline 5 65 F Diplopia Spontaneous Diffuse pachymenigeal enhancement Conservative with hydration

Discussion tion may be needed.2 The CSF supports the brain in the cranial cavity. When CSF leak occurs there is decreased pressure within the cranial Imaging in ICH cavity leading to descent of the brain in the upright position, Magnetic resonance imaging is now considered as one of causing stretching effect over the pain sensitive the most useful modalities in the diagnosis of ICH. The and thus causing severe headache. The stretching of the cranial MRI features include thin subdural collection over cranial may also lead to dysfunction and symp- bilateral cerebral surface, diffuse pachymeningeal enhance- toms like diplopia, vertigo, tinnitus and facial numbness.8 ment on post contrast study, descent of the brain, abnormal enhancement and enlargement of the pituitary gland and The diagnosis of ICH is confirmed by demonstrating venous engorgement.9 decreased opening of CSF pressure below 60 mm of water (normal 65 - 195 mm of H2O). Occasionally there may be a The subdural haematoma is usually thin and bilateral with dry tap in the beginning and in some cases a gentle aspira- no significant mass effect. The possible pathophysiology of

106 PAN ARAB JOURNAL OF NEUROSURGERY INTRACRANIAL HYPOTENSION • Mongia, et al subdural haematoma/collection is stretching and rupture of Conclusion the bridging veins of the subdural space due to descent of Headache is a non-specific symptom but orthostatic brain. headache and associated cranial nerve dysfunction strongly suggest possibility of ICH. Magnetic resonance imaging There is diffuse pachymeningeal (dural) enhancement as with contrast is the most important diagnostic imaging against the leptomeningeal enhancement of meningitis. It modality. There may be delay in the classical MR appear- can be secondary to venous engorgement. ance as is seen in 2 of our cases. Usually the condition resolves with conservative management such as oral caffeine The brain descent can be seen on MRI as effacement of and theophylline but refractive cases may require epidural prepontine cistern, inferior displacement of optic chiasm, blood patch and surgical management of CSF leak. descent of iter (opening of aqueduct) below the incisural line (connecting anterior tuberculum sellae to the point of In our series of 5 cases 4 patients presented with headache confluence of straight sinus with inferior sagittal sinus and and one with diplopia. All the symptoms were postural. the great cerebral vein) and descent of the cerebellar 8 Two patients were spontaneous with no cause, one with tonsils. spontaneous rupture of perineural cyst in pleural space, one was due to post traumatic CSF leak and one after spinal The pituitary gland enlargement and enhancement can surgery. Three patients responded to conservative treatment occur due to venous engorgement. This may lead to hormo- and one needed surgical repair of site of CSF leak and one nal disturbances. patient was lost to follow-up.

Although the MRI features are frequent findings these findings are not the absolute rule. There may be delay in the References 1. Ali SA, Cesani F, Zuckermann JA, et al: Spinal-cerebrospinal onset of classical MR features. fluid leak demonstrated by radiopharmaceutical cisterno- graphy. Clin Nucl Med 1998, 23(3): 152-155 The spinal MRI findings in ICH have been described as 2. Bell WE, Joynt RJ, Sahs AL: Low spinal fluid pressure extraarachnoid fluid collection, spinal meningeal enhance- syndromes. Neurol 1960, 10: 512-521 6 3. Benamor M, Tainturier C, Graveleau P, Pierot L: Radionuclide ment and dilated epidural veins. In addition to diagnosis of cisternography in spontaneous intracranial hypotension. Clin ICH spinal MRI may in some cases detect the site of CSF Nucl Med 1998, 23(3): 150-151 leak. Computerised tomography may also be 4. Berroir S, Loisel B, Ducros A, et al: Early epidural blood patch used in detecting the CSF leak. in spontaneous intracranial hypotension. Neurol 2004, 63: 1950-1951 5. Chung SJ, Kim JS, Lee MC: Syndrome of cerebral spinal fluid The radioisotope cisternography hypovolemia: clinical and imaging features and outcomes. The radioisotope cisternography can help in identifying the Neurol 2000, 55(9): 1321-1327 site of leak by providing indirect evidence to suggest 6. Clarot F, Callonnec F, Douvrin F, et al: Giant cervical epidural ICH.3,5 veins after lumbar puncture in a case of intracranial hypo- tension. Am J Neuroradiol 2000, 21(4): 787-789

7. Karremann M, Sojka S, Huehn R, Preiss U, Schmidt F: Intra- Treatment methods cranial hypotension syndrome following surgery of the spine: A Most of the patients with spontaneous ICH respond to rare cause of postural headache in adolescence. J Ped Neurol conservative therapy which include bed rest and oral 2007, 5: 323-325 hydration. The supine position helps in healing the dural 8. Paldino M, Mongilner AY, Tenner MS: Intracranial hypotension syndrome: a comprehensive review. Neurosurg Focus 2003, rent. Administration of steroids, oral and intravenous 15(6): ECP2 caffeine and theophylline (400 mg/day) have been pro- 9. Schievink WI: Spontaneous spinal cerebrospinal fluid leaks posed.9 Caffeine given orally or intravenously is reported to and intracranial hypotension. JAMA 2006, 295(19): 2286-2296 be effective in 75 - 85% of patients with post-lumbar 10. Schievink WI, Morreale VM, Atkinson JL, et al: Surgical treatment of spontaneous spinal cerebrospinal fluid leaks. J puncture headache. The mechanism, by which caffeine Neurosurg 1988, 88(2): 243-246 increases CSF production, is unclear. Caffeine is an 11. Schievink WI, Reimer R, Folger WN: Surgical treatment of spon- adenosine receptor antagonist that decreases cerebral blood taneous intracranial hypotension associated with a spinal arach- flow and secondarily increases CSF production. noid diverticulum. Case report. J Neurosurg 1994, 80(4): 736-739 12. Sencakova D, Mokri B, McClelland RL: The efficacy of epidural

blood patch in spontaneous CSF leaks. Neurol 2001, 57(10): Autologus blood patch in the spinal epidural space is 1921-1923 supposed to be the most effective treatment. If unsuccessful 13. Turnbull DK, Shepherd DB: Post-dural puncture headache: it can be repeated.4,9,12 The epidural blood probably forms a pathogenesis, prevention and treatment. Br J Anaesth 2003, 91(5): 718-29 dural tamponade and seals the leak. Surgical treatment of 14. Winter SC, Maartens NF, Anslow P, Teddy PJ: Spontaneous CSF leak is needed in refractory cases and in those patients intracranial hypotension due to thoracic disc herniation. Case where the site of leak is identified.10,11 report. J Neurosurg 2002, 96(3 Suppl): 343-345

VOLUME 14, NO. 2, OCTOBER 2010 107 Case Review

Extradural with intradural-extramedullary and intramedullary tuberculoma of the spine without bony involvement

Nigel Peter Symss, Goutham Cugati, Anil Pande, Ravi Ramamurthi, MC Vasudevan

Abstract: Extradural with intradural-extramedullary and intramedullary spinal tuberculomas causing compression is an uncommon entity. We report a case of a 20-year-old female patient who was undergoing treatment for intracranial tuberculous infection with triple chemotherapy antituberculous regimen. One month later she presented to us with progressive paraparesis and MRI scan showed a dorsal intradural-extramedullary lesion. She underwent laminectomy and excision of the lesion. She was readmitted 2 weeks later with flaccid paraplegia, and MRI scan showed an intramedullary lesion extending from D3 - D7 level. This case is unusual in regard to the concurrent extradural with intradural-extramedullary and intramedullary with intracranial tuberculomas. Also interesting is the absence of bony involvement. (p108-111)

Key words: Extradural, intradural-extramedullary, intramedullary, intracranial and tuberculomas.

Introduction Involvement of the neuraxis by tuberculosis has always knowledge, this is the first such case to be reported in the been uncommon relative to its involvement of other systems. literature where the spinal tuberculomas are located extra- The potential for excellent recovery following adequate dural, intradural-extramedullary and intramedullary. treatment, compared with the increased morbidity following a missed diagnosis, is a stimulus for continued awareness of Case Report the condition and must be considered in the differential A 20-year-old non-immunocompromised female patient diagnosis of patients presenting with spinal cord compres- was admitted to another hospital with complaints of fever, sion. The disease is only rarely seen in developed countries; headache, vomiting, neck pain and stiffness of 3 weeks however it still remains endemic in developing countries duration. She was evaluated and based on CT scan which such as Asia and Africa. Also the incidence of spinal showed a solitary non enhancing hypodense lesion in the tuberculomas is likely to increase with the rise in the pons and MRI scan (Fig. 1) of the brain which showed incidence of HIV infections. multiple ring enhancing lesions in the right cerebellum, right posterior cingulate gyrus and hypothalamus was This case is being reported because it is unusual to have diagnosed to have intracranial tuberculous infection. This extradural, intradural-extramedullary with intramedullary was supported by CSF analysis which was suggestive of tuberculomas without bony involvement, along with con- tubercular infection: appearance-turbid, sugar-52 mg/dl, current intracranial tuberculous infection. To the best of our protein-198 mg/dl, cells 206 per cu mm, 90% lymphocytes. Ziehl Neelsen stain was negative for acid fast bacilli. She was started on three drugs antituberculous therapy (rifam- picin, isoniazide, pyrazinamide). She presented to us one Department of Neurosurgery month later with decreased sensations in the lower limbs, Post Graduate Institute of Neurological Surgery progressive difficulty in walking due to weakness of the Dr A. Lakshmipathi Neurosurgical Centre VHS Hospital lower limbs, low back pain with a tight band like sensation Chennai around the umbilicus, for the last 4 days. She had no India bladder or bowel involvement. On examination her higher Correspondence: intellectual functions, fundus, cranial nerves and upper Dr. Nigel Peter Symss limbs were normal. She had paraparesis, proximal weak- Post Graduate Institute of Neurological Surgery Dr. A Lakshmipathi Neurosurgical Centre ness was 3/5 and distal weakness 4/5, tone and bulk was VHS Hospital, IT Corridor normal, with brisk reflexes and extensor plantar response. She Taramani Main Road Chennai 600 113 had impaired crude and fine touch, pain and temperature, India and position and vibration sensations below L4 - L5 Email: [email protected] dermatome. Her systemic examination was normal.

108 PAN ARAB JOURNAL OF NEUROSURGERY TUBERCULOMA OF THE SPINE WITHOUT BONY INVOLVEMENT • Symss, et al

X-rays of the dorsal and lumbosacral spine (Fig. 2) were normal. Magnetic resonance imaging scan of the dorso- lumbar spine (Fig. 3) showed a posteriorly placed hetero- genous intradural-extramedullary intensely enhancing lesion at D11 and D12 level. The lesion was causing pressure on the distal thoracic cord with hyperintense signal changes seen in the conus, suggestive of oedema. There was also an intra- medullary hyperintense signal seen at D5 level.

The patient underwent a D10 - D12 laminectomy. There Figure 1 - MRI scan of the brain sagittal showing the lesions. was an extradural component of the lesion which could be excised easily as it was well encapsulated, with a plane of cleavage with the dura, except in one area where it was adherent and appeared to have an intradural extension. The dura was opened and was found thickened with arachnoid adhesions. The lesion was extending intradurally and was not adherent to the cord and could be separated and totally excised leaving the cord intact. The dura was closed after repair.

In the postoperative period she had transient worsening of her lower limbs power which gradually improved. She had urinary retention after catheter removal and was sent home with an indwelling Foleys catheter. The histopathological examination revealed a granulomatous lesion comprising Langhans giant cells, inflammatory cells with caseating necrosis suggestive of caseating tuberculoma. She was con- Figure 2 - X-rays dorsal spine showing no involvement of the bone. tinued on antituberculous treatment. She was readmitted 2 weeks later with complaints of rapid worsening of weakness of the lower limbs, absent sensations below the xiphisternum and fecal incontinence for the last 5 days. On examination, she had flaccid paraplegia, with grossly impaired sensa- tions below D4, and absent sensations below D7 dermatome. Anal tone was lax, with no sphincter contractions. Magnetic resonance imaging scan of the dorsal spine showed an intramedullary ring enhancing lesion extending from D3 - D7 level with diffuse oedema (Fig. 4). She underwent a D3 - D6 laminectomy and myelotomy. A thick grayish capsule was encountered and on aspi- ration there was thick yellow pus. The capsule was excised totally. Postopera- tively she had good improve- ment in her lower limb power and sensations but continued to have bladder and bowel dis- turbances. On 5 years follow- up she has near normal power in the lower limbs, with normal bowel function.

Figure 3 - MRI of the dorsal spine, plain and contrast showing the lesion.

VOLUME 14, NO. 2, OCTOBER 2010 109 TUBERCULOMA OF THE SPINE WITHOUT BONY INVOLVEMENT • Symss, et al

Figure 4 - MRI plain and contrast showing intramedullary lesion.

Discussion Mycobacterium tuberculosis can involve the neural and The majority of extradural tuberculous granulomas are sec- perineural tissues directly.3 Our patient presented with ondary to an osseous lesion.24 However, there are reports of neurological deficits without radiological evidence of bony spinal extradural tuberculous lesions without evidence of involvement. She initially presented with a slowly pro- any bony involvement.8,16,21 Tandon et al, are of the opinion gressive paraparesis with sensory disturbances and on that most of these lesions are secondary to undetected bony investigation and surgery was found to have an extradural lesions and that many of these patients, if followed-up, with intradural-extramedullary tuberculoma. Two weeks would reveal a tuberculous lesion of the spine at the following her discharge after surgery she returned with corresponding level.24 flaccid paraplegia, absent sensations in the lower limbs with bladder and bowel involvement. In general, patients with Intradural-extramedullary tuberculoma of the spinal cord is intradural-extramedullary tuberculomas have presented an uncommon entity and the least common cause of spinal with a gradual onset over weeks to months with progressive compression in tuberculosis.10 The lesions are so uncom- weakness of the legs, occasionally associated with sphincter mon that they appear in the literature only as isolated case or sensory disturbances. A history of exposure to or reports.2,5,6,14 The intramedullary location of tuberculomas concurrent tuberculosis is often obtainable, and the patients are also a rare cause of spinal cord compression. Their have been treated for tuberculosis meningitis in the past.1,19 occurrence is much less frequent than intracranial tuber- Occasionally symptoms may occur acutely following trauma.3 culomas. Dastur found only 6 spinal tuberculomas as Intramedullary spinal tuberculomas commonly present as against 260 intracranial tuberculomas.4 Mathai and Chandy subacute spinal cord compression, with progressive lower found 2 against 143.13 It is extremely rare to have spinal limb weakness, paraesthesia and bladder bowel dysfunc- tuberculomas concomitant with intracerebral disseminated tion. The paraplegia may either be spastic (61%) or flaccid tuberculomas (Table 1).7,9,15,22,23,25,26 (33%).12

The neurological manifestations of spinal tuberculosis are Magnetic resonance imaging plays an important role in the most often secondary to involvement of the bone but detection and diagnosis of spinal tuberculomas. Hypo- or

Table 1 - List of reported cases of concurrent spinal and intracranial tuberculomas. Author Year Age/Gender Spinal location Level Intracranial location Shen WC 1993 30/Male Intramedullary Cervical Brainstem, cerebellum, cerebral hemisphere Kim MS 2000 49/Female Intradural-extramedullary Dorsal Disseminated Yen HL 2003 67/Male Intramedullary Dorsolumbar Brainstem, cerebellum, cerebral hemisphere Thacker MM 2004 6/Female Intramedullary Dorsal Frontal hemisphere, cerebellum Shenoy SN 2004 38/Male Intramedullary Cervical Cerebral hemispheres Muthukumar 2007 21/Male Intradural-extramedullary Dorsal Disseminated George P 2007 28/Male Intramedullary Dorsolumbar Temporal lobe Symss NP 2009 20/Female Extradural, intradural- Dorsal Hypothalamus, posterior cingulated gyrus, extramedullary, intramedullary cerebellum

110 PAN ARAB JOURNAL OF NEUROSURGERY TUBERCULOMA OF THE SPINE WITHOUT BONY INVOLVEMENT • Symss, et al isointensity on T2-WI within the spinal cord with surround- experiences. Part II: Spinal cord and its coverings. Neurol ing hyperintense oedema is suggestive of intramedullary India 1972, 20(3): 127-131 6. Garcia Allut A, Gelabert González M, Bollar Zabala A, Martinez tuberculomas. Central hyperintensities are also detected at Rumbo R, Reyes Santias R: Intradural extramedullar tuber- times due to a variable amount of caseous necrosis with culoma of spinal cord. Case report. J Neurosurg Sci 1985, 29 liquefaction. On T1-WI, fusiform swelling of the cord is (2): 113-115 seen along with iso- to hyperintense foci, surrounded by 7. George P, Agrawal A, Kumar S, Shetty JP, Shetty RK: Tuber- 18 cular meningitis with concurrent intracranial and intra-spinal hypointense oedema of the cord. Such findings should tuberculomas. Euro J Gen Med 2007, 4(2): 91-94 prompt a contrast enhanced study, which may show a single 8. Johnston JDH, Ashbell TS, Rosomoff HL: Isolated intraspinal or conglomerate disc or ring enhancing lesions.18 Since extradural tuberculosis. N Eng J Med 1962, 266: 703-5 intraspinal tuberculomas produce a mass effect that can 9. Kim MS, Kim KJ, Chung CK, Kim HJ: Intradural extra- medullary tuberculoma of the spinal cord: a case report. J jeopardize spinal cord function, the modern management of Korean Med Sci 2000, 15(3): 368-70 spinal tuberculomas is generally accepted as surgical 10. Kumar S, Puri V, Pal DS: Intradural extramedullary tuber- excision of the lesion. Surgery also confirms the diagnosis culoma. Neurol India 1991, 39: 213 and should be followed by a full course of chemotherapy 11. Lin SK, Wu T, Wai YY: Intramedullary spinal tuberculomas 17 during treatment of tuberculous meningitis. Clin Neurol with three antituberculous drugs. This has brought about Neurosurg 1994, 96(1): 71-8 a dramatic decrease in the incidence of postoperative 12. MacDonnell AH, Baird RW, Bronze MS: Intramedullary tuberculous meningitis. Parsons and Pallis have treated a tuberculomas of the spinal cord: case report and review. Rev case successfully with antituberculous therapy and steroids Infect Dis 1990, 12(3): 432-9 19 13. Mathai KV, Chandy J: Tuberculous infections of the nervous alone. Rao has reported the successful medical manage- system. In: Ojemann RG, Shillito J (eds), Clinical Neurosurgery. 20 ment of 4 patients of intramedullary tuberculomas. The Baltimore, Williams & Wilkins Co 1967, pp 145-77 appearance of intramedullary spinal tuberculomas devel- 14. Mathuriya SN, Khosla VK, Banerjee AK: Intradural extra- oping paradoxically during effective treatment of tuber- medullary tuberculous spinal granulomas. Clin Neurol 11,17 Neurosurg 1988, 90(2): 155-8 culous infections have been described in the literature. 15. Muthukumar N, Sureshkumar V, Ramesh VG: En plaque Muthukumar et al, have also reported an extensive en- intradural extramedullary spinal tuberculoma and concurrent plaque intradural-extramedullary tuberculoma in conjunc- intracranial tuberculomas: paradoxical response to antituber- tion with asymptomatic multiple intracranial tuberculomas culous therapy. Case report. J Neurosurg Spine 2007, 6(2): 15 169-73 as a paradoxical response. Surgical decompression is 16. Natarajan M: Intraspinal granulomas. Neurol India 1974, 22 necessary to treat the expanding spinal tuberculoma with (3): 163-8 the continuation of antituberculous therapy and steroids.17 17. Nomura S, Akimura T, Kitahara T, Nogami K, Suzuki M: Surgery for expansion of spinal tuberculoma during antituberculous chemotherapy: a case report. Pediatr Neurosurg 2001, 35(3): Conclusion 153-7 It is unusual to have extradural, intradural-extramedullary 18. Parmar H, Shah J, Patkar D, Varma R: Intramedullary tuber- with intramedullary tuberculomas without bony involve- culomas. MR findings in seven patients. Acta Radiol 2000, 41 (6): 572-7 ment along with concurrent intracranial tuberculous infection. 19. Parsons M, Pallis CA: Intradural spinal tuberculomas. Neurol Patients with intradural-extramedullary lesions usually present 1965, 15(11): 1018-1022 with gradual onset, progressive paraparesis, whereas intra- 20. Rao GP: Spinal intramedullary tuberculous lesion: medical medullary lesions present as subacute spinal cord compres- management. Report of four cases. J Neurosurg 2000, 93(1 Suppl): 137-41 sion and should be considered in the differential diagnosis 21. Rao SB, Dinakar I, Rao KS: Extraosseous extradural tuber- of spinal cord compression. Surgical excision of the lesions culous granuloma simulating a herniated lumbar disc. Case followed by an 18 month course of antituberculous chemo- report. J Neurosurg 1971, 35(4): 488-90 therapy with three drugs is the treatment of choice. 22. Shen WC, Cheng TY, Lee SK, Ho YJ, Lee KR: Disseminated tuberculomas in spinal cord and brain demonstrated by MRI with gadolinium-DTPA. Neuroradiol 1993, 35(3): 213-5 References 23. Shenoy SN, Raja A: Concurrent intramedullary and intra- 1. Arseni C, Samitca DC: Intraspinal tuberculous granuloma. cerebral tuberculomas. Neurol India 2004, 52(4): 514-516 Brain 1960, 83: 285-292 24. Tandon PN, Pathak SN: Tuberculosis of the central nervous 2. Bucy PC, Oberhill HR: Intradural spinal granulomas. J system. In: Spillane JD (ed), Tropical Neurology. London, Neurosurg 1950, 7(1): 1-12 Oxford University Press 1973, pp 37-62 3. Compton JS, Dorsch NWC: Intradural extramedullary tuber- 25. Thacker MM, Puri AI: Concurrent intra-medullary and intra- culoma of the cervical spine. J Neurosurg 1984, 60(1): 200- cranial tuberculomas. J Postgrad Med 2004, 50(2): 107-109 203 26. Yen HL, Lee RJ, Lin JW, Chen HJ: Multiple tuberculomas in 4. Dastur HM: A tuberculoma review with some personal the brain and spinal cord: a case report. Spine 2003, 28(23): experiences. Part I: Brain. Neurol India 1972, 20(3): 111-126 E499-502 5. Dastur HM: A tuberculoma review with some personal

VOLUME 14, NO. 2, OCTOBER 2010 111 Review Article

Minimally invasive neurosurgery

Allen L Ho1, Peter M Black2

Introduction The aim of minimally invasive neurosurgery is to achieve functional loss. Recently established and emerging intraop- resolution of clinical issues with fewer disturbances of normal erative imaging techniques have substantially increased the structures, and more favourable postoperative outcomes modern neurosurgeon’s ability to meet this goal.33,150 including quicker recovery and fewer complications. In a sense, minimally invasive surgery is the ultimate goal of all Preoperatively, functional magnetic resonance imaging neurosurgical procedures - to achieve the desired results (fMRI) is a versatile tool for functional assessment of the 2,75,106,108,176 without interfering with the normal work of the brain. This regions of tumour invasion. Functional magnetic paper will discuss three developments in minimally invasive resonance imaging assesses increased neural activity by neurosurgery: for traditional cortical surgery, brain mapping monitoring the accompanying increase in blood flow. and imaging; for base surgery, neuroendoscopy; and Specifically, it targets deoxyhaemoglobin as an endogenous for vascular neurosurgery, endovascular techniques. (p1-11) contrast enhancing agent which produces the fMRI signal. Functional magnetic resonance imaging has also been A. Neuronavigation and functional imaging applied preoperatively in conjunction with electrocortical Neuronavigation and functional imaging are revolutionizing stimulation (ECS) to more accurately map functional 107,145 preoperative and intraoperative decision making in many regions of the brain prior to tumour resection. Further- neurosurgical disease processes. As new technology and more, magnetic resonance spectroscopy (MRS) can be emerging imaging modalities lead to higher resolution combined with anatomical imaging to contribute metabolic imaging of not just anatomic structures, but the real-time information to surgical planning. Physiological MRS biology and physiology of the brain, their incorporation into imaging signal is observed beyond the T2-hyperintense the modern neurosurgical operating room will lead to more region in anatomical imaging in 60 and 65% of patients 123 accurate clinical assessment and better outcomes. Below, with and without contrast enhancement, respectively. we review the most current protocols and novel imaging Intraoperatively, MRS has been developed and incorporated techniques on the horizons deployed to address brain into the neuronavigational operating room to help delineate tumours and epilepsy. the surrounding disease of the lesion, in addition to its 53,134 anatomical boundaries. Magnetic resonance spectro- Neuronavigation and imaging plays an especially important scopy can also be utilized for more efficient stereotactic 35,37,76 role in neurosurgical treatment of tumours because of the biopsies and radiosurgical planning. competing priorities of attaining the greatest amount of tumour resection for better survival but simultaneously Diffusion tensor imaging (DTI), flouro deoxyglucose positron preserving as much normal tissue as possible to minimize emission tomographic imaging (FDG-PET), and SPECT can also aid neurosurgeons discriminate between compro- mised and normal tissue. Diffusion tensor imaging is based on fractional anisotropy that allows for identification of 4 1Harvard Medical School subcortical white matter tracts. Utilized preoperatively, it Boston, MA allows surgeons to anticipate the effect of lesions on normal USA fibre tracts, as well as gauge the effectiveness of therapy in 78 2Department of Neurosurgery cellularly dense tumours. Diffusion tensor imaging-based Brigham and Women’s Hospital fibre tract maps have been validated using subcortical Boston, MA 14 USA stimulation mapping. Thus, DTI can provide a safety margin around motor tract for use in surgical planning and Correspondence: 113 Prof. Peter M Black can help preserve tracts during surgery. Flouro deoxy- Department of Neurosurgery glucose positron emission tomographic imaging relies on Brigham and Women’s Hospital 75 Francis Street cerebral metabolism, determined by FDG uptake, compared Boston, MA 02115 to baseline to generate an image mapping function in the USA 163 Tel: (1 617) 525 7796 brain. Both FDG-PET and SPECT can be used to Fax: (1 617) 734 8342 discriminate between radiation necrosis and tumour recur- Email: [email protected] rence in the presence of ambiguous MRI data.20

VOLUME 14, NO. 2, OCTOBER 2010 1 MINIMALLY INVASIVE NEUROSURGERY • Ho & Black

Magnetoencephalography (MEG) is similar to EEG except for memory lateralization in individual patients.52,70,135 The that it measures magnetic field changes that accompany only drawback to fMRI is its limitations in terms of neuronal activity rather than voltage changes to assess brain geometric distortion and tendency to lose signal in the activity. A group has recently used preoperative MEG to anterior MTL.135 target the central sulcus in patients with AVMs near the motor cortex. Locating the central sulcus in relation to the B. Endoscopic skull base surgery lesion is important in deciding between surgical or radio- Traditional skull base neurosurgery provides decent expo- surgical treatment options.154 Functional magnetic resonance sure and, over time, neurosurgeons refined open operative imaging and Wada testing have also proven to be important techniques to achieve generally positive results. However, preoperative modalities for evaluation of patients with this comes at the expense of disturbing normal brain AVMs. Wada testing is a series of behavioural tests applied structure through the need for brain retraction and manipu- after one side of the brain is anaesthetized via ICA injection lation of neurovasculature. Open skull base techniques also of sodium amytal. Its initial function was for language are more limited in their visualization of sellar, suprasellar dominance lateralization(171) but the technique has since and retrochismal regions. The advent of neuroendoscopy been modified to encompass memory lateralization(124) and has revived a less invasive endonasal route that is supplant- enhanced to be more highly localized with selective catheri- ing more invasive approaches in skull based neurosurgical zation.18 Functional magnetic resonance imaging and Wada practice.72,140,180 The endoscopic endonasal approach repre- testing have been used with dominant hemisphere pre- sents a new paradigm in neurosurgery since it provides Sylvian AVMs to evaluate suitability for intraoperative ECS wider access to the skull base with increased visualization and optical imaging.21 and often leads to reduced postoperative discomfort and complications.50,65,67,87 Functioning imaging utilized to address epilepsy is focused on localizing areas of epileptic activity, in addition to The indications for the transsphenoidal endoscopic tech- lateralization and localization of surrounding structures. nique are virtually the same as conventional microscopic Subdural grids and strips have been used traditionally in pituitary surgery. The technique is especially advantageous tandem with depth electrodes to identify seizure foci via for use with children, elderly, and/or compromised patients ictal and interictal recordings.85 There are many new because of its minimal invasiveness, quick recovery time intraoperative optical imaging modalities that can be and elimination of postoperative breathing difficulties employed to localize seizure foci.74 Flouro deoxyglucose usually caused by nasal packing.48,49 The pure endoscopic (1,55,56,167) positron emission tomographic imaging , H215O endonasal approach is performed with a rigid endoscope PET(164), and spike triggered fMRI(51,79,148,149) have been used inserted into one nostril endonasally. The endoscope offers to interictally localize seizure foci in the neocortex, as well visualization without a working channel. The sphenoid as within the medial temporal lobe. Medial temporal lobe sinus is accessed by extending an irrigating shaft through an (MTL) MEG(104) and ictal SPECT(169) may also be utilized enlargement of the natural ostium or nasal septum elevation to identify epileptogenic foci. Indeed, MEG has been from the sphenoid keel. This surgical canal allows for a confirmed by subdural grid electrode recordings to be a wider closer view of targets in the skull base.28,89,114 reliable identifier of seizure foci in neocortical epilepsy.125 In another comparison study of MEG accuracy in identi- One of the strengths of the endoscopic endonasal approach fying seizure focal versus MRI, EEG and ictal and interictal is its versatility. Though the technique proceeds through a subdural grid electrodes, MEG was second only to ictal smaller opening than most skull based approaches, the intracranial recordings in predicting a positive surgical out- endoscope circumvents this challenge and actually allows come.175 Finally, MEG was also able to accurately localize for increased visualization and access to the entire midline seizure foci to the correct lobe, and could thus be used for skull base from the cribiform plate to the anterior foramen guidance in the placement of subdural electrodes.118 magnum, making the technique applicable for a large variety of intrasellar and extrasellar lesions.46,47,95-97,99,109 When dealing with MTL epilepsy, the most important Anatomical studies of this approach have allowed for consideration is loss of memory. Preoperative functional enhancement of many procedures.5,6,31,46,68 The standard imaging modalities are especially helpful in this regard.135 transsphenoidal approach has also been recently modified to The current gold standard for assessing risk of postoperative allow for additional neuroendoscopic access, many times in amnesia following anterior temporal lobectomy is Wada tandem with microscopy, to parasellar areas from the clivus testing.165 Functional magnetic resonance imaging is also to the cavernous sinus and above all of the planum now being applied to lateralize memory function for the sphenoidale via anterior, anterolateral, and posterolateral same procedure. In comparison studies between Wada routes.41,42,46,54,57,62-64,94,102,105,116,119,174 The suprasellar region testing and fMRI, both seem to be equally useful modalities of the brain is now also being similarly accessed to address

2 PAN ARAB JOURNAL OF NEUROSURGERY MINIMALLY INVASIVE NEUROSURGERY • Ho & Black suprasellar tumours(25,41,54,58,81,82,94,96,98,100,102,103,105), and to directions, and reducing nasal pain and facial swelling expose the suprasellar optic pathways and other neuro- caused by spreading of the speculum to extend the surgical vasculature.26 Finally, the current margins of the expanded field. It also may negate the need for postnasal packing, endoscopic endonasal approach for the skull base are the improving postoperative breathing and decreasing headache frontal sinus and cribiform plate anteriorly, the medial and dry oral mucosa associated with postnasal packing. orbital walls laterally and anteriorly, the cavernous sinus Complications of sublabial or anterior rhinoseptal incisions and carotid arteries within the sellar region, and the dorsum necessary for standard transsphenoidal microsurgery are sella and posterior clinoids posteriorly.30,158 also avoided.27 Reduced postoperative discomfort has made this the preferred methodology of choice and has Some limitations to the endoscopic endonasal mostly involve greatly improved patient compliance.27,180 the novelty of the technique. The unfamiliarity of the anatomical terrain of endonasal structures has led to longer The efficacy of the endoscopic endonasal approach has initial operative times.45 Furthermore, there is a lack of been demonstrated in a wide variety of pathologies.47,65,111 instrumentation designed specifically for this approach(24,38), Looking at just tumours of the skull base, where total which makes operating within such a small margin tumour resection is ideal, the approach has had measurable unwieldy, leading to complications including profuse haem- success when compared to traditional transcranial and orrhage, venous bleeding of the nasal mucosa, and finally, transsphenoidal microsurgery approaches. The midline rupture of the intracavernous internal carotid artery (ICA). approach from below averts the need for brain retraction or Secondly, though the field of visualization is generally neurovascular manipulation and allows for early devas- increased with endoscopy, endoscopes only provide two- cularization of lesions, in addition to the increased access dimensional flat images in contrast to the three-dimensional and visualization afforded by the endoscope.47,65,111 Micro- stereoscopic images available via microscope.45 Develop- scope based removal of suprasellar lesions using an expanded ment of the bimanual binostril technique has addressed approach has previously reported resection rates of 22 to some of these issues by allowing increased instrument 46%.54,117 A transsphenoidal surgery series reported by manipulation and microsurgical dissection in the same Couldwell et al, had a gross rate of total resection of cranial manner as regular microsurgery.29 base lesions of 66%.42 More selective microscopic approach series for craniopharyngiomas had a rate of gross Perhaps the most problematic complication is incomplete total removal of between 12.5 - 89%.34,54,112,117 However, closure of the cranial base which leads to CSF fistulae.34,47,111 Frank et al, reported a rate of 70% utilizing the expanded Use of a pedicled nasoseptal flap to reconstruct the cranial endoscopic approach.65 Furthermore, other recent endoscopic base(73,133) separating the subarachnoid space from the series of suprasellar lesions report similar comparable or sinonasal tract can greatly reduce the occurrence of CSF improved results when contrasted with microscopic fistulae.101 Other common neurosurgical complications like approaches. A most recent series by Dehdashi et al, on meningitis(42,54,65), arterial vessel injury(47,65,111), and cranial resection of anterior base lesions had a gross total resection nerve injury have been rare.42,54 This approach obviates rate of 73%. Preoperative visual deficits were improved in brain retraction, minimizes optic apparatus manipulation, all but one patient in expanded endoscopic approach series and allows early identification of the pituitary gland and (47,50,111), versus 85% improvement in visual fields reported infundibulum.58 by expanded microscopic approach.54

By now, thousands of endoscopic operations have been Overall, the efficacy of the endoscopic endonasal approach performed by pituitary surgeons worldwide with high has been exhaustively demonstrated due to its overall effec- success rates proven by postoperative imaging and patient tiveness, lower complication rate(43), and improved patient follow-up.26,49,63,92 In fact, the use of endoscopy matches, compliance.73 There is a general consensus developing and in many cases, exceeds results of standard trans- among leaders in the technique that the endoscopic sphenoidal microsurgery.27 A combination of factors related endonasal approach has the potential to reduce morbidity to the approach have lent themselves to better outcomes and hospital stays, and improve the quality of life in patients overall as defined by rates of positive clinical outcomes, when compared with traditional transcranial and even increased patient safety and convenience. endonasal microsurgical approaches.23,65,66,87,88,96 For these reasons, it is considered an important minimally invasive The most important operative advantage of the endoscopic surgical technique that is increasingly becoming a standard endonasal approach is that it averts the need for brain retraction of care in skull base neurosurgery.27 or neurovascular manipulation, and allows for early devas- cularization of lesions. The procedures also avoids the use C. Endovascular neurosurgery of a speculum, allowing for wider movements in all Endovascular neurosurgery is a field where newly developed

VOLUME 14, NO. 2, OCTOBER 2010 3 MINIMALLY INVASIVE NEUROSURGERY • Ho & Black technology is rapidly being tested and assimilated in a drops in morbidity and mortality rates. However, this clinical setting to achieve less invasive interventions and strategy does have its limitations: stents designed for the favourable outcomes for diseases with historically signifi- heart could not be advanced beyond the carotid siphon in cant morbidity and mortality. We will review the current many patients, and even with slow inflations of the balloon endovascular therapies and future directions for endo- and undersizing of the stent, vessel rupture and dissections vascular treatment of cerebral ischemia and structural still occurred.115 The Gateway balloon-Wingspan stenting neurovascular defects. system was developed to address these issues (Boston Scientific/Target, Natick, MA). An intracranial athero- Every year more than 700,000 individuals experience new stenotic lesion is expanded to 80% of the vessel diameter or recurrent strokes, and this number will increase to more and a self-expanding nitinol stent is then laid over with 3 than 1,000,000 by the year 2020.143 Intracranial arthero- mm of overlap on either side. Food and Drug Administra- sclerotic disease (ICAD) represents an 8 - 10% subset of tion approval was granted with a study that showed a ischemic strokes(147,177), and by 2020 will have an overall decrease in angiographic stenosis from 75 - 32%, and an burden greater than that of subarachnoid haemorrhage and ipsilateral stroke rate of 7%.16 Recent results from a arteriovenous malformation (AVM) combined.19 In the multicentre study have reported a 6.1% major complication same way, ICA stenosis and vertebral artery (VA) origin rate and 5% death rate associated with the procedure.60 stenosis are both significant causes of stroke morbidity and mortality, and can also be managed via endovascular There are several distal protection filters and stents neurosurgical techniques with the right indications.22,90,122,132 approved by the FDA for use in high-risk symptomatic Because of sheer volume of patients, acute ischemic stroke patients with ICA stenosis. While stents will still prove holds the most potential for expansion of endovascular useful in select patients, filters will likely capture the neurosurgery in the next few decades, due in large part to majority of the future market shares because of their ease of the under utilization of current endovascular techniques.3,139 use, safety and maintenance of continual blood flow.83 Restenosis of the ICA can then be treated with cutting While one of the most significant advances made in the balloon angioplasty that has produced clinically efficient medical treatment of stroke was tissue-type plasminogen results with low morbidity and can be repeated with activator (t-PA), which was shown to make a significant recurrence.11,12 Current endovascular treatment for VA difference in outcomes if used in patients within 3 hours of origin stenosis is similar to carotid artery stenting but stroke, the Merci retriever (Concentric Medical, Mountain restenosis rates exceed 40% because of smaller vessel View, CA) was the first approved endovascular mechanical diameter and tortuosity.159 Drug-eluting stents have been device approved for clot removal.166 Used in conjunction utilized in VA origin stenosis but long-term efficacy of these with t-PA, the device extends the time window to 8 hours devices remains to be realized.138 and achieves significant recanalization results.69 Recent trials have reported ICA recanalization rates of 53 and 63% Endovascular treatment of cerebral ischemia is a modality with the device alone, and with the addition of adjunctive with enormous growth potential because of a large and ever endovascular treatment, respectively.61 A microcatheter- expanding patient population, and under-utilization of avail- based device called the Penumbra (Penumbra, Inc., San able techniques. Tissue-plasminogen activator and the Leandro, CA) was also recently developed and approved by Merci retriever are used in less than 5% of clinically eligible the Food and Drugs Administration (FDA) to macerate and patients.3 Improved real-timing imaging will also aid in the aspirate soft clot, and then remove fibrous thrombus in evaluation and treatment of stroke. Transcranial colour- fibrous occlusions. Complete restoration of flow and 45% coded duplex ultrasonography has already proved more good outcomes at 30 days were achieved with the useful than CT scanning in differentiating between Penumbra in a Phase 1 trial of patients not eligible for t-PA intracerebral haemorrhage from stroke and identifying treatment.17 For similarly difficult patients that fail to stroke complications.15,120 Similarly, intravascular ultra- respond to t-PA and intra-arterial thrombolysis, balloon- sound is a useful modality to evaluate the effectiveness of mounted and self-expanding stents have been placed to carotid artery stenting procedures.13,160-162 recanalize vessels. Though the long-term effects of intra- cranial stents have yet to be determined, recanalization rates Device development and biologics also represents a fertile in these patients are higher than any other published tech- area of growth for endovascular neurosurgery. New con- nique. Furthermore, these are patients who would have cepts for stent design have emerged for the treatment of otherwise suffered from large vessel strokes.152 ICAD. Magnetic stents to capture circulating endothelial cells, stents coated with endothelial progenitor cell anti- The advent of staged angioplasty and stenting strategy in bodies to attract circulating endothelial cells and initiate the treatment of symptomatic ICAD has lead to substantial early endothelialization, and biodegradable, reabsorbable

4 PAN ARAB JOURNAL OF NEUROSURGERY MINIMALLY INVASIVE NEUROSURGERY • Ho & Black poly L,D-lactic acid, and even more rapidly reabsorbable endovascularly. Vein of Galen malformations and carotid- magnesium stents are all in the development and evaluation cavernous fistulae (CCF) are two structural abnormalities pipeline.80,153,157,172 New drug eluting stents have also been that can be handled exclusively by neuroendovascular proposed to help mitigate restenosis but concerns about late therapy. In a recent study, nearly 75% of patients managed thrombosis and a lack of long-term evaluation have held up with vein of Galen malformations treated endovascularly wide acceptance of these devices.138 New devices for distal were normal at follow-up. Endovascular embolization, protection of the VA could provide the same staging surgery, and stereotactic radiosurgery are all combined to foundation and embolic protection as are already currently treat complex intracranial and spinal dura-based AVMs and available for carotid circulation.137 Finally, animal models AVF s. 83,110 of experimentally induced stroke have shown that human umbilical blood cells can permeate through the blood brain The main challenges of treating giant aneurysms are barrier, migrate towards areas of ischemic disruption and recanalization rates and elimination of vascular masses via differentiate along both neural and astrocytic lineages platinum coiling. Stent-assisted coiling is the most widely resulting in recovery from stroke. One can envision the used modality but promising new techniques are currently delivery of human stem cells by endovascular surgeons into being developed and explored. The development of vascular distributions that have been compromised during flexible, resheathable and coated stents could cure giant the early stroke period.36 aneurysms in areas of low perforator densities. The recently approved enterprise stent (Cordis Endovascular, The use of coils to address neurovascular aneurysms has Miami Lakes, FL) is 80% resheathable into the delivery blossomed in the past decade. With over 70 types of microcatheter. Asymmetric stent with a portion of low- Guglielmi detachable coils alone, more than 40% of the porosity stainless steel can be valuable for regions of high aneurysms in the US were being coiled, and more than perforator densities(39,126,127,146,178,181), and stents with unique 300,000 patients treated worldwide by the year 2005.179 strut designs or telescoping capabilities may allow for flow This increase in use has been accompanied by a decrease in diversion to obviate the need for coiling.91 New morbidity and mortality. Cerebral aneurysm treatment via innovations in coil design are focused on embedding coils coiling lead to a 25% reduction in the risk of death or with materials that can be better integrated within structures dependency associated with treatment.128 Detachment and actually promote a healing response. Platinum coils mechanisms for the coil are the main differentiating factor infused with polyglycolic acid/lactide to this end have between the many coiling devices now available. These achieved ambiguous results but studies have identified include electrolytic (current mediated detachment), high subpopulations of responders and nonresponders to this fluid pressure (syringe mediated detachment), and technology.49,131 Collagen-based coils have been proposed biologically active coils that function to increase volume to allow for continuous placement of intra-aneurysmal and/or density of coil packing, and induce an immuno- embolic material until complete obliteration is achieved. logical response that will encourage reconstruction of the This expansile collagen (hydrogel) adds volume to the coil parent vessel. Diagnosis and treatment of cerebral vasos- and the most recent trial suggests that occlusion rates may pasm has also been extended beyond the usual nimodipine be increased when 75% of the coil length or last coil is pharmacological treatments. If utilized within the critical 2 hydrogel coated.40,92 Stents can also be used to treat AVFs hour therapeutic time window(144), compliant and non- of the transverse and sigmoid sinuses, recovering blood compliant balloons for angioplasty have improved blood flow in an occluded sinus, and involuting the fistula. The flow, decreased mismatch, and lead to better patient use of platinum coils and hydrocoils along with various outcomes with vasospasms.7 However, there still exists types of stents (covered and porous) and liquid embolic many hurdles in the field. Coiling of large and giant agents has been explored.71,84,168 aneurysms still have a 50% recanalization rate and there still exists significant room for development of endovas- Finally, the use of liquid embolic agents also hold promise cular treatment of bifurcation and fusiform lesions.83 with regard to aneurysms and other malformations when used in conjunction with endovascular Endovascular treatment of vascular malformations of the devices. Onyx (ev3 Neurovascular, Irvine, CA) is a non- central nervous system has not enjoyed the same boom over adhesive ethylene vinyl alcohol copolymer that have the past decade as coiling for aneurysms but remains a field demonstrated a recanalization rate of 4% with stents in poised to benefit significantly from advances in endovas- treatment of giant aneurysms, compared to 36% recanali- cular modalities. Currently AVM and arteriovenous fistulae zation rate without stents.32,129 The ability of Onyx to be (AVF) of the brain and spine lie within the domain of the injected multiple times for prolonged periods of time has endovascular neurosurgeon, though not exclusively. Only been particularly useful for the treatment of AVMs and 15% or fewer of these malformations can be treated AVFs. Onyx treatment has produced more than a 60%

VOLUME 14, NO. 2, OCTOBER 2010 5 MINIMALLY INVASIVE NEUROSURGERY • Ho & Black

86 reduction in AVM volume. Recent studies have also 3): S53-55 shown obliteration rates between 16 and 20% per- 4. Alexander AL, Lee JE, Lazar M, Field AS: Diffusion tensor cent.156,170,173 imaging of the brain. Neurotherapeutics 2007, 4(3): 316-329 5. Alfieri A, Jho HD, Schettino R, Tschabitscher M: Endoscopic endonasal approach to the pterygopalatine fossa: anatomic Middle cerebral artery (MCA) aneurysms are the main type study. Neurosurg 2003, 52: 374-378; Discussion 378-380 of bifurcation aneurysm being targeted for new 6. Alfieri A, Jho HD: Endoscopic endonasal cavernous sinus endovascular stent development. The Neuroform Y-stent is surgery: an anatomic study. Neurosurg 2001, 48: 827-836; a technically challenging but potential useful device for Discussion 836-827 (151) 7. Beck J, Raabe A, Lanfermann H, Berkefeld J, De Rochemont MCA lesions , and aggressive balloon techniques in the Rdu M, Zanella F, Seifert V, Weidauer S: Effects of balloon MCA have also been reported as effective with bifurcation angioplasty on perfusion- and diffusion-weighted magnetic aneurysms.130 Fusiform aneurysms need to be isolated and resonance imaging results and outcome in patients with 44 cerebral vasospasm. J Neurosurg 2006, 105: 220-227 circumvented. Approaches through standard femoral 8. Becker TA, Kipke DR, Preul MC, Bichard WD, McDougall CG: artery access or direct access in the neck for stent recon- In vivo assessment of calcium alginate gel for endovascular struction may be helpful.77 Additionally, use of Onyx or embolization of a cerebral arteriovenous malformation model other liquid embolic agents in combination with balloons or using the Swine rete mirabile. Neurosurg 2002, 51: 453-458; Discussion 458-459 stents has shown limited success in recent studies and may 9. Becker TA, Preul MC, Bichard WD, Kipke DR, McDougall CG: represent a new strategy for addressing difficult le- Calcium alginate gel as a biocompatible material for endo- sions.32,121,129 vascular arteriovenous malformation embolization: six-month results in an animal model. Neurosurg 2005, 56: 793-801; Discussion 793-801 Again, endovascular surgeons are looking towards bio- 10. Becker TA, Preul MC, Bichard WD, Kipke DR, McDougall CG: logics to provide the next stage of device development for Preliminary investigation of calcium alginate gel as a bio- aneurysm treatment. Biodegradable coils embedded with compatible material for endovascular aneurysm embolization growth factors, antibody-tethered viruses, or radiation- in vivo. Neurosurg 2007, 60: 1119-1127; Discussion 1127-1118 inducible promoters to heal aneurysms necks would be the 11. Bendok BR, Hopkins LN: Cutting balloon angioplasty to treat 142 carotid in-stent restenosis. J Invasive Cardiol 2004, 16: A16; holy-grail of aneurysm treatment. Preliminary work Discussion A16 indicates that this future may not be far off. Sponges 12. Bendok BR, Roubin GS, Katzen BT, Boulos AS, Levy EI, soaked with vascular smooth muscle cells infected with Limpijankit T, Qureshi AI, Guterman LR, Hopkins LN: Cutting balloon to treat carotid in-stent stenosis: Technical note. J adenovirus encoding recombinant transforming growth Invasive Cardiol 2003, 15: 227-232 factor-β were placed in canine aneurysms resulted in 13. Benedek I, Hintea T: Current developments in interventional increased neointimal thickness and successful gene trans- treatment of total terminal aortic occlusions-laser, stenting and fection.141 New embolic agents are also being designed to balloon angioplasty: experience of cardiology clinic of Targu- Mures. Rom J Intern Med 2005, 43: 223-232 address vascular malformations. A polymer formed from 14. Berman JI, Berger MS, Chung SW, Nagarajan SS, Henry RG: seaweed called calcium alginate is in development because Accuracy of diffusion tensor magnetic resonance imaging of the ability of the by-products of calcium and alginate to tractography assessed using intraoperative subcortical stimu- flow freely in the bloodstream without causing thom- lation mapping and magnetic source imaging. J Neurosurg 8-10,155 2007, 107: 488-494 boembolitic complications. Agents that are free of 15. Bertram M, Khoja W, Ringleb P, Schwab S: Transcranial thomboembolic risk, dissolvable if misplaced, and have colour-coded sonography for the bedside evaluation of mass neuroprotective properties for use in tandem with effect after stroke. Eur J Neurol 2000, 7: 639-646 radiosurgery (for higher grade lesions) would be powerful 16. 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Sani S, Lopes DK: Treatment of a middle cerebral artery Thompson PJ, Duncan JS: The application of functional MRI of bifurcation aneurysm using a double neuroform stent "Y" memory in temporal lobe epilepsy: a clinical review. Epilepsia configuration and coil embolization: technical case report. 2004, 45: 855-863 Neurosurg 2005, 57: E209; Discussion E209 136. Prevedello DM, Thomas A, Gardner P, Snyderman CH, Carrau 152. Sauvageau E, Samuelson RM, Levy EI, Jeziorski AM, Mehta RL, Kassam AB: Endoscopic endonasal resection of a RA, Hopkins LN: Middle cerebral artery stenting for acute synchronous pituitary adenoma and a tuberculum sellae ischemic stroke after unsuccessful Merci retrieval. Neurosurg meningioma: technical case report. Neurosurg 2007, 60: 2007, 60: 701-706; Discussion 706 E401; Discussion E401 153. Schatz RA: Interventional cardiology: evolution and future. Am 137. Qureshi AI, Kirmani JF, Harris-Lane P, Divani AA, Ahmed S, Heart Hosp J 2006, 4: 202-204 Ebrihimi A, Al Kawi A, Janjua N: Vertebral artery origin stent 154. Shimamura N, Ohkuma H, Ogane K, Manabe H, Yagihashi A, placement with distal protection: technical and clinical results. Kikkawa T, Suzuki S: Displacement of central sulcus in Am J Neuroradiol 2006, 27: 1140-1145 cerebral arteriovenous malformation situated in the peri-motor 138. Qureshi AI, Kirmani JF, Hussein HM, Harris-Lane P, Divani AA, cortex as assessed by magnetoencephalographic study. Acta Suri MF, Janjua N, Alkawi A: Early and intermediate-term Neurochir (Wien) 2004, 146: 363-368; Discussion 368 outcomes with drug-eluting stents in high-risk patients with 155. Soga Y, Preul MC, Furuse M, Becker T, McDougall CG: symptomatic intracranial stenosis. Neurosurg 2006, 59: 1044- Calcium alginate provides a high degree of embolization in 1051; Discussion 1051 aneurysm models: a specific comparison to coil packing. 139. Qureshi AI, Suri MF, Nasar A, He W, Kirmani JF, Divani AA, Neurosurg 2004, 55: 1401-1409; Discussion 1409 Prestigiacomo CJ, Low RB: Thrombolysis for ischemic stroke 156. Song DL, Leng B, Xu B, Wang QH, Chen XC, Zhou LF: in the United States: data from National Hospital Discharge Clinical experience of 70 cases of cerebral arteriovenous Survey 1999-2001. Neurosurg 2005, 57: 647-654; Discussion malformations embolization with Onyx, a novel liquid embolic 647-654 agent. Zhonghua Wai Ke Za Zhi 2007, 45: 223-225 140. Rhoton AL Jr: Operative techniques and instrumentation for 157. Sousa JE, Costa MA, Tuzcu EM, Yadav JS, Ellis S: New neurosurgery. Neurosurg 2003, 53: 907-934; Discussion 934 frontiers in interventional cardiology. Circulation 2005, 111: 141. Ribourtout E, Desfaits AC, Salazkin I, Raymond J: Ex vivo 671-681 gene therapy with adenovirus-mediated transforming growth 158. Spencer WR, Das K, Nwagu C, Wenk E, Schaefer SD, factor beta1 expression for endovascular treatment of Moscatello A, Couldwell WT: Approaches to the sellar and aneurysm: results in a canine bilateral aneurysm model. J parasellar region: anatomic comparison of the microscope Vasc Surg 2003, 38: 576-583 versus endoscope. Laryngoscope 1999, 109: 791-794 142. Ribourtout E, Raymond J: Gene therapy and endovascular 159. SSYLVIA Study Investigators: Stenting of Symptomatic

10 PAN ARAB JOURNAL OF NEUROSURGERY MINIMALLY INVASIVE NEUROSURGERY • Ho & Black

Atherosclerotic Lesions in the Vertebral or Intracranial Arteries 170. van Rooij WJ, Sluzewski M, Beute GN: Brain AVM (SSYLVIA): study results. Stroke 2004, 35(6): 1388-1392 embolization with Onyx. Am J Neuroradiol 2007, 28: 172-177; 160. Stone GW, Colombo A, Teirstein PS, Moses JW, Leon MB, Discussion 178 Reifart NJ, Mintz GS, Hoye A, Cox DA, Baim DS, Strauss BH, 171. Wada J, Rasmussen T: Intracarotid injection of sodium amytal Selmon M, Moussa I, Suzuki T, Tamai H, Katoh O, Mitsudo K, for the lateralization of cerebral speech dominance: Grube E, Cannon LA, Kandzari DE, Reisman M, Schwartz Experimental and clinical observations. J Neurosurg 1960, 17 RS, Bailey S, Dangas G, Mehran R, Abizaid A, Serruys PW: (2): 226-282 Percutaneous recanalization of chronically occluded coronary 172. Waksman R: Biodegradable stents: they do their job and arteries: procedural techniques, devices, and results. Catheter disappear. J Invasive Cardiol 2006, 18: 70-74 Cardiovasc Interv 2005, 66: 217-236 173. Weber W, Kis B, Siekmann R, Kuehne D: Endovascular 161. Stone GW, Kandzari DE, Mehran R, Colombo A, Schwartz treatment of intracranial arteriovenous malformations with RS, Bailey S, Moussa I, Teirstein PS, Dangas G, Baim DS, onyx: technical aspects. Am J Neuroradiol 2007, 28: 371-377 Selmon M, Strauss BH, Tamai H, Suzuki T, Mitsudo K, Katoh 174. Weiss MH: The transnasal transsphenoidal approach. In: O, Cox DA, Hoye A, Mintz GS, Grube E, Cannon LA, Reifart Apuzzo MLJ (ed), Surgery of the Third Ventricle. Baltimore, NJ, Reisman M, Abizaid A, Moses JW, Leon MB, Serruys PW: Williams & Wilkins 1987, pp 476-494 Percutaneous recanalization of chronically occluded coronary 175. Wheless JW, Willmore LJ, Breier JI, Kataki M, Smith JR, King arteries: a consensus document: part I. Circulation 2005, 112: DW, Meador KJ, Park YD, Loring DW, Clifton GL, 2364-2372 Baumgartner J, Thomas AB, Constantinou JE, Papanicolaou 162. Stone GW, Reifart NJ, Moussa I, Hoye A, Cox DA, Colombo A, AC: A comparison of magnetoencephalography, MRI, and V- Baim DS, Teirstein PS, Strauss BH, Selmon M, Mintz GS, EEG in patients evaluated for epilepsy surgery. Epilepsia Katoh O, Mitsudo K, Suzuki T, Tamai H, Grube E, Cannon LA, 1999, 40: 931-941 Kandzari DE, Reisman M, Schwartz RS, Bailey S, Dangas G, 176. Wilkinson ID, Romanowski CA, Jellinek DA, Morris J, Griffiths Mehran R, Abizaid A, Moses JW, Leon MB, Serruys PW: PD: Motor functional MRI for pre-operative and intraoperative Percutaneous recanalization of chronically occluded coronary neurosurgical guidance. Br J Radiol 2003, 76: 98-103 arteries: a consensus document: part II. Circulation 2005, 112: 177. Wityk RJ, Lehman D, Klag M, Coresh J, Ahn H, Litt B: Race 2530-2537 and sex differences in the distribution of cerebral athero- 163. Tai YF, Piccini P: Applications of positron emission tomography sclerosis. Stroke 1996, 27: 1974-1980 (PET) in neurology. J Neurol Neurosurg Psychiatry 2004, 75: 178. Yiemeng H, Ciprian NI, Rekha VT, Kenneth RH, Scott HW, 669-676 Dale BT, Hui M, Stephen R: Flow modification in canine 164. Tatlidil R, Xiong J, Luther S: Presurgical lateralization of seizure intracranial aneurysm model by an asymmetric stent: studies focus and language dominant hemisphere with O-15 water using digital subtraction angiography (DSA) and image-based PET imaging. Acta Neurol Scand 2000, 102: 73-80 computational fluid dynamics (CFD) analyses. Presented at 165. Tharin SMDPD, Golby AMD: Functional brain mapping and its the Medical Imaging 2006: Physiology, Function, and Structure applications to neurosurgery. Neurosurg 2007, 60(4): ONS185- from Medical Images, San Diego, Ca, February 12th, 2006 202 179. Yuki I, Murayama Y, Vinuela F: Development of medical 166. The National Institute of Neurological Disorders and Stroke rt- devices for neuro-interventional procedures: special focus on PA Stroke Study Group: Tissue plasminogen activator for acute aneurysm treatment. Expert Rev Med Devices 2005, 2: 539- ischemic stroke. N Engl J Med 1995, 333(4): 1581-1587 546 167. Theodore WH, Sato S, Kufta C, Balish MB, Bromfield EB, 180. Zada G, Kelly DF, Cohan P, Wang C, Swerdloff R: Endonasal Leiderman DB: Temporal lobectomy for uncontrolled seizures: transsphenoidal approach for pituitary adenomas and other the role of positron emission tomography. Ann Neurol 1992, sellar lesions: an assessment of efficacy, safety, and patient 32: 789-794 impressions. J Neurosurg 2003, 98: 350-358 168. Troffkin NA, Given CA 2nd: Combined transarterial N-butyl 181. Zhou W, Ciprian NI, Stephen R, Kenneth RH, Adam BP, Daniel cyanoacrylate and coil embolization of direct carotid-cavernous RB: Angiographic analysis of blood flow modification in fistulas. Report of two cases. J Neurosurg 2007, 106: 903-906 cerebral aneurysm models with a new asymmetric stent. Proc 169. Van Paesschen W: Ictal SPECT. Epilepsia 2004, 45(Suppl 4): SPIE 2004, 5369: 307-318 35-40

VOLUME 14, NO. 2, OCTOBER 2010 11 Case Report

Post-traumatic dorsal spinal extradural haematoma without osseous lesion

Lamia Bencherif1, Mohammed Benzagmout2, Zidane Ihabe1, Leila Mahfouf1, Abdennebi Benaissa1

Abstract: We report a case of traumatic spinal extradural haematoma without associated osseous/disc lesion, and without any complication of haemostasis in a young patient who was a victim of a sporting accident. After the trauma, the patient had presented with simple dorsal rachialgia without neurological signs. Dorsal spinal plain x-rays were normal. Soon after, he returned to his sessions of body-building, however, 20 days later, he presented with muscular weakness and progressive paraparesia evolving rapidly to paraplegia.

The patient was hospitalized in neurosurgical emergency. Spinal MRI revealed dorsal extradural haematoma extending from D2 to D4. Dorsal laminectomy was performed in emergency allowing the complete evacuation of the haematoma. No macroscopic abnormality was noted at surgery. However, the patient did not show any sign of motor recovery and the patient was reoperated on one day later because of persistent haematoma on CT scan with control. The motor recovery was dramatic after the second surgery; then, the patient was refereed to rehabilitation therapy.

Key words: Spinal extradural haematoma, MRI, paraplegia, surgery and rehabilitation therapy. (p112-114)

Introduction Upon review of the literature, only a few publications re- inflammatory drugs for 5 days with a good improvement of garding traumatic spinal extradural haematoma have been rachialgia. Then, the patient was instructed in muscular reported compared to spontaneous extradural haematoma. exercises as the pain was thought to be of muscular origin. Also, this type of traumatic extradural haematoma is classi- cally associated with osseous fracture and/or the presence of Three weeks later, the patient started to complain of pro- haemostasis abnormalities. However, in our case the patient gressive paraparesis which rapidly worsened to paraplegia presented with traumatic dorsal extradural haematoma without within 48 hours. At this stage, the patient was referred to any associated disc or osseous lesion, and no crasis prob- our emergency department where initial neurological ex- lems; the clinical symptoms begun 20 days after the trauma. amination revealed complete paraplegia (grade 0/5) with vigorous deep tendon reflexes, hypotonia, positive Babinski Case Report sign, but with no sensory deficit or genito-urinary problems. We report the case of a young athletic male, who experi- enced direct spinal trauma during judo training. At once, the The diagnosis of spinal cord compression was considered patient experienced acute dorsal pain. Spinal plain x-rays and magnetic resonance imaging (MRI) of the dorsal spine were normal and the patient received analgesics and anti- was done. It revealed a bi-convex collection with T1- and T2-weighted hypersignal intensity extending from D2 to D4 without any disc or osseous lesion (Fig. 1). 1 Department of Neurosurgery Hôpital Salim Zemirli El Herrech Algiers Diagnosis of extradural haematoma was made according to Algeria the imaging features and recent history of trauma. Selective spinal cord angiography was negative. Operative decision 2Department of Neurosurgery University Hospital Hassan II was taken and the patient underwent dorsal laminectomy to Fez release the spinal cord and evacuate the haematoma. Morocco

Correspondence: Results Dr. Lamia Bencherif Department of Neurosurgery At surgery, the meninges were macroscopically normal; a Hôpital Salim Zemirli El Harrech dural tear was found with no osseous fracture or ligamentous Algiers Algeria tear. In the immediate postoperative period, the patient did Fax: (213 2) 150 6562 not show any sign of motor recovery. Computerised tomo- Email: [email protected] graphy scan of control was performed and demonstrated

112 PAN ARAB JOURNAL OF NEUROSURGERY DORSAL SPINAL EXTRADURAL HAEMATOMA WITHOUT OSSEOUS LESION • Bencherif, et al

Discussion According to Bruyn and Bosna, only 10% of extradural spinal haematomas are posttraumatic with different local- isations and associated osseous lesions.2 In the Foo and Rossier series which included 5 personal cases with a literature review of 38 cases of spinal extradural haematomas reported since 1927; these cases were divided into two groups: a group of spinal extradural haematomas associated with osseous fractures which represented 16 cases from the literature in addition to their own 5 cases; the other group consisted of spinal extradural haematomas without osseous fractures and included 22 cases from the literature.5 In this latter group, the aetiology of bleeding was related to various aetiologies: arteriovenous malformations, coagulopathy, hypertension, and treatment with anticoagulants. However, in 4 cases the aetiology remained unknown.

Similarly, Lesoin et al, reported a series of 8 cases of post- traumatic spinal extradural haematomas in which 4 cases were described without osseous fractures.7 In this group, the haematoma resulted from anticoagulant treatment in 2 cases and was related to arterial bleeding from ruptured inter- spinous ligaments in the other 2 cases. Figure 1 - MRI in sagittal and axial views showing the extra- dural haematoma (arrow). In our case, there were no osseous lesions or associated disc bulge, as well as no factors favouring bleeding such as persistent haematoma at the level of the laminectomy. As a hypertension, arteriovenous malformations, anticoagulant consequence, the patient was reoperated on 24 hours after treatment or coagulopathy. No interspinous ligament or the first intervention. Surgical exploration revealed bleeding spinal muscle rupture was noted peroperatively. from the bone edges of the laminectomy which was controlled with bone wax; there was also muscular bleeding due to The delay in the clinical expression could be explained by unsuccessful haemostasis. Then, neurological examination the venous nature of the bleeding which is generally minimal on the third postoperative day showed dramatic motor and had a progressive and delayed formation. Indeed, the recovery. The full details of this examination are shown in bleeding was the consequence of the break of the venous Table 1. Afterwards, the patient was discharged to reha- extradural plexuses secondary to the strength of cutting at bilitation by the end of the second week. the time of the accident. This bleeding had probably been exacerbated by physical exercise which had increased the Table 1 – Results of neurological examination of lower limbs venous pressure in the extradural space and led to the following second surgery. accumulation of blood in the extradural space causing Joint Movement Right Left spinal cord compression. For that reason, we strongly advise Hip Flexion 3- 3- bed rest in cases of serious spinal trauma. Even in cases Extension 3- 3+ with normal radiological imaging and irrelevant clinical Abduction 3+ 3+ Adduction 3+ 3+ presentation. Internal rotation 3+ 3+ External rotation 3+ 3+ The delay of the clinical expression could be explained by Knee Flexion 3- 3+ the venous nature of the bleeding but also by separating the Extension 3- 3+ meningeal layers. This last hypothesis explains the spon- Ankle Plantar flexion 3+ 3+ taneous regression of some non-traumatic spinal extradural Dorsiflexion 3- 3+ haematomas.4 Invertion 3+ 3+ Evertion 3- 3+ Feet Flexor hallucis 3 3+ The absence of osseous fracture is related to the type of Flexor digitorum 3 3+ trauma (minimal or violent) and the degree of ligamentous Extensor hallucis 3 3+ elasticity which explains the rarity of osseous fractures in Flexor digitorum 3 3+ infants.

VOLUME 14, NO. 2, OCTOBER 2010 113 DORSAL SPINAL EXTRADURAL HAEMATOMA WITHOUT OSSEOUS LESION • Bencherif, et al

In addition, the dorsal region manifests clinically less the trauma. Thus, we believe that the prognosis is often rapidly compared to the cervical region where the canal is good when the duration between initial presentation, in the more stenotic. In the series of Foo and Rossier, 43% of form of spinal pain and signs of medullary compression are haematomas were located at the cervical spine and 18% in protracted, which suggests that spinal cord compression is the dorsal spine.6 Whatever location, spinal haematomas not so significant. have a tendency of being posterolateral where there is more space. In our case, the extradural haematoma was also According to the literature, in a study of 20 cases of located posteriorly significantly compressing the cord. traumatic spinal extradural haematoma, the postoperative motor recovery was 95.3% for a partial motor and sensory The clinical differential diagnoses include spinal cord con- deficit, 87% for patients with complete motor and partial tusion or ischaemia but generally clinical symptoms occur sensory deficit, and 45.3% with complete motor and sensory immediately after the trauma in these conditions. deficit.8 According to Foo and Rossier, patients operated between 12 and 36 hours following presentation show Magnetic resonance imaging remains the method of choice better recovery than those operated 36 hours after.7 in diagnosing this type of haematoma, and showing the degree of extension and compression of the cord. Comput- Conclusion erised tomography scan is useful in demonstrating osseous Bed rest following spinal trauma appears to be mandatory lesions such as sagittal fractures which could be missed in even without any associated lesions in order to reduce any simple radiographs. The MRI aspect depends on the age of possible bleeding. In the event of any neurological signs, the haematoma, as in our case the hypersignal intensity in MRI is indicated as it remains the examination of choice T1- and T2-weighted images of the haematoma was related showing the degree of cord compression. The main prog- to the stage of methaemoglobin with the typical bi-convex nostic factors are the duration between the start of spinal layer, and a known history of trauma confirming the diag- cord compression and the trauma, the degree of medullary nosis of extradural haematoma.3 compression and the clinical stage at which operative procedure is carried out. Radiological differential diagnosis includes subdural haem- atoma and the axial sequences assisted in confirming such. References Subdural haematoma shows a concavity towards the cord 1. Bouderka MA, Bouaggad A, Aitbenali S, Barrou H, Abassi O: and this may be the basis for performing a myelography, Hématome extra-dural cervical révélateur d’une hémophilie. which is helpful in locating the lesion either extradural or Neurochir 1999, 45(3): 247-9 2. Bruyn GW, Bosma NJ: Spinal extradural hematoma. In: subdural but not the actual nature of the lesion. Vinken PJ, Bruyn GW (eds), Handbook of Clinical Neurology. Amsterdam, North-Holland Publishing 1976, Vol. 26, pp 1-30 Occasionally, trauma leads to vascular malformation such 3. Chen HH, Hung CC: Spontaneous spinal epidural hematoma: as arteriovenous fistulas which may be undersized and not Report of seven cases. J Formos Med Assoc 1992, 91(2): 214-8 visible macroscopically in some cases. In our case, angio- 4. Clark DB, Bertand G, Tampieri D: Spontaneous spinal epidural graphy was normal and the meninges were normal at hematoma causing paraplegia: Resolution and recovery surgery. Haemostasis complications were also considered without surgical decompression. Neurosurg 1992, 30(1): 108-11 but complete analysis was negative. According to the 5. Foo D, Rossier A: Post-traumatic spinal epidural hematoma. J Neurosurg 1982, 11: 25-32 literature, a case of minor trauma has led to extended 6. Foo D, Rossier A: Preoperative neurological status in predicting cervical haematoma. In that case, the disparity about the surgical outcome of spinal epidural hematomas. Surg Neurol cause of haematoma had extended the exploration which 1981, 15(5): 389-401 revealed a factor VIII rate of 45% and the haematoma was 7. Lesoin F, Rousseaux M, Viaud C, et al: Hématomes épiduraux 1 rachidiens post-traumatiques: Huit observations. Annales de related to minimal type A haemophilia. Chirurgie 1985, 39(4): 251-5 8. Nuti C, Fotso MJ, Duthel R, Hatem O, Dumas B, Brunon J: Our patient showed initially partial motor recovery 3/5 Hématomes épiduraux non traumatiques du rachis: despite delayed evacuation of the haematoma three days présentation de 20 cas: Revue de la littérature et étude des aspects évolutifs. Neurochir 2003, 49(6): 562-570 after the occurrence of clinical symptoms and 23 days after

114 PAN ARAB JOURNAL OF NEUROSURGERY Case Report

Lumbar radicular pain caused by epidural varices

Majid Reza Farrokhi, Hadi Niknam

Abstract: Low back pain and sciatica are mainly caused by herniated lumbar disc and spondylotic lumbar canal stenosis. However, some rare entities such as lumbar epidural venous anomalies can result in acute low back pain and sciatica, among which epidural varices are less encountered. It can be not only idiopathic but also secondary to some vascular malformations. In this study, we report two cases of lumbar epidural varices presenting with low back pain and acute severe sciatica. (p115-116)

Key words: Low back pain, sciatica, spinal epidural venous anomalies andepidural varices.

Introduction Epidural venous plexus enlargement, presenting with low S1 root. An adequate specimen was sent to a pathologist back pain and radiculopathy, is an uncommon cause of nerve and the varix was coagulated by bipolar diathermy. Then roots impingement. It is estimated at 4.5% of operations for we divided and removed it. Excellent postoperative recov- lumbar disc herniation.2 Possible underlying aetiologies that ery of radicular pain was achieved. A pathologist reported a may result in symptomatic epidural varices include vascular tissue with endothelial membrane consistent with a vascular anomalies(5), iliac, superior or inferior vena cava thrombosis lesion (Fig. 2). (2,10), Budd-Chiari syndrome(3), intracranial hypotension(1), pregnancy(7,8) and portal hypertension.9 Nevertheless, some cases are idiopathic and have no underlying aetiology. We report two cases of such rare entities. Figure 1 - MRI with con- trast at L5 - S1 space Case 1: A 24-year-old male sailor was admitted with low showed an epidural varix back pain since few weeks prior to the admission with acute in a 24-year-old man onset of severe right sciatica upon lifting heavy items.4 He with acute and severe had no history of any underlying disease and also did not right sciatica. have any varicose veins in the lower extremities. There was no neurological deficit. He had positive right straight leg rising (SLR) at 30° and positive left crossed straight leg rising (CSLR) at 45°. X-ray was normal. Magnetic reso- nance imaging (MRI) of lumbosacral spine was performed and a right paracentral small mass in L5 - S1 space causing an impingement on right S1 root was noted, but to some extent its appearance was different to protruded disc. In lumbosacral MRI with contrast, mild enhancement was seen (Fig. 1). The patient's surgical treatment was per- formed by right L5 - S1 laminatomy. There were enlarged epidural veins in L5 - S1 space, causing pressure on the right

Figure 2 - Haematoxylin and eosin staining shows fragments of degenerated fibrocartilage and vascular wall with haemor- Shiraz Neurosciences Research Center Shiraz University of Medical Sciences rhagic areas consistent with vascular malformation (H&E × 200). Shiraz Iran Case 2: A 50-year-old male carpenter was admitted with Correspondence: Dr. Majid Reza Farrokhi low back pain and acute onset of severe right sciatica upon Shiraz Neurosciences Research Center lifting heavy objects.4 He had no history of any underlying Chamran Hospital, Chamran Boulevard Shiraz disease and also did not have any varicose veins in the Iran lower extremities. The neurological test was normal. He Email: [email protected] / [email protected] had positive right SLR at 40° and positive left CSLR at 45°.

VOLUME 14, NO. 2, OCTOBER 2010 115 LUMBAR RADICULAR PAIN CAUSED BY EPIDURAL VARICES • Farrokhi & Niknam

X-ray was normal too. In lumbosacral MRI, there was a appearance of mass was different to the protruded disc or right paracentral small mass in L4 - L5 space, causing an free fragment and moderate enhancement was seen after the impingement on right L5 root (Fig. 3). In lumbosacral MRI, injection. According to anatomical classifications, both by contrast, mild enhancement was seen. The patient's varices corresponded to type 2. surgical treatment was performed through right L4 - L5 laminatomy. We saw enlarged epidural veins in L4 - L5 Conclusion space, causing pressure on right L5 root. Specimen was sent We should consider varicose vein as a possible diagnosis for to pathology. We coagulated and removed the epidural any patient who would be referred with severe acute onset varix. Again, excellent postoperative pain relief was radicular pain without any evidence of disc herniation in achieved. MRI (sagittal plane), but with small, enhancing or non- enhancing mass in axial plane of MRI. Abdominal sonography is recommended to rule out any vascular malformation or venous thrombosis. Figure 3 - MRI at L4 - L5 space showed an In secondary types, treatment is directed toward elimination epidural varix in a 50- of underlying cause, but in idiopathic ones, surgery is the year-old man presented treatment of choice with good result. with acute and severe sciatica. Acknowledgements: We would like to thank Ms. Hosseini and Ms. Gholami for their kind assistance in Shiraz Neurosciences Research Center.

Discussion References Neural compression syndromes comprise various aetiologies, 1. Albayram S, Wasserman BA, Yousem DM, Wityk R: Intra- causing sciatica, among which herniated lumbar disc and cranial hypotension as a cause of radiculopathy from cervical epidural venous engorgement: case report. Am J Neuroradiol lumbar spondylotic spinal canal stenosis are more frequent. 2002, 23: 618-21 2. Blattler W, Krayenbuhl C: Spinal canal stenosis syndrome by An acute lumbar disc herniation with nerve root compres- venous collateralization of an inferior cava thrombosis. sion is usually a reasonably straightforward clinical entity Phlebologie 1993, 46(3): 411-4; Discussion 415 and its appropriate diagnosis and treatment is not difficult. 3. Bozkurt G, Cil B, Akbay A, et al: Intractable radicular and low back pain secondary inferior vena cava stenosis associated Nevertheless, there are some rare entities which mimic with Budd-Chiari syndrome: Endovascular treatment with cava acute lumbar disc herniation, such as vertebral haemangioma stenting: case report and review of the literature. Spine 2006, with epidural extension, epidural cavernous haemangioma, 31(12): 383-6 epidural arteriovenous malformation, paravertebral arterio- 4. Demaerel P, Petré C, Wilms G, Plets C: Sciatica caused by a dilated epidural vein: MR findings. Eur Radiol 1999, 9(1): 113-4 venous fistula with epidural venous drainage and epidural 5. Demeulenaere A, Spell L, Lafitte F: Vertebro-epidural lumbo- 5 varix. These vascular anomalies can be ruled out by spinal sacral vascular malformations. An unusual cause of lumbo- MRI, MRA and angiography. Lumbar radicular pain caused sciatic pain. J Neuroradiol 1999, 26(4): 225-35 by epidural varices is uncommon. It is estimated at 4.5% of 6. Genevay S, Palazzo E, Huten D, Fossati P, Meyer O: Lumbo- 2 radiculopathy due to epidural varices: two cases report and a operations for lumbar disc herniation. Three anatomical review of the literature. Joint Bone Spine 2002, 69(2): 214-217 types, thrombosed varix (type 1), non-thrombosed varix (type 7. Gormus N, Ustun ME, Paksoy Y, et al: Acute thrombosis of 2) and localized haematoma (type 3) are described.6 The inferior vena cava in a pregnant women presenting with mechanism of radicular pain is not fully understood. It may sciatica: a case report. Ann Vasc Surg 2005, 19: 120-2 8. Hirabayashi Y, Shimizu R, Fukuda H, et al: Effects of the preg- be secondary to compression effect of varix but it may also nant uterus on the extradural venous plexus in the supine and be secondary to difficulties in venous return. Both of the lateral positions, as determined by magnetic resonance imaging. patients in this study had sudden onset and severe radicular Br J Anaesth 1997, 78: 317-9 pain upon attempting to lift heavy objects that might cause 9. LaBan MM, Wang AM, Shetty A, et al: Varicosities of the para- vertebral plexus of veins associated with nocturnal spinal pain an increase in the epidural venous pressure and result in as imaged by magnetic resonance venography: a brief report. varix formation. Any acute increase in epidural venous Am J Phys Med Rehabil 1999, 78: 72-6 pressure can cause acute enlargement of these varices and 10. Paksoy Y, Gormus N: Epidural venous plexus enlargements sudden acute radicular pain.11 Diagnosis can be established presenting with radiculopathy and back pain in patients with inferior vena cava obstruction or occlusion. Spine 2004, 29: by MRI at preoperational stages. The images vary accord- 2419-24 ing to the anatomical types and the age of the lesion. In the 11. Slin'ko EI, Al-Qashqish II: Surgical treatment of lumbar epidural mentioned cases, abnormal disc bulging was not seen at L5 varices. J Neurosurg Spine 2006, 5(5): 414-23 - S1 and L4 - L5 spaces, respectively. To some extent the

116 PAN ARAB JOURNAL OF NEUROSURGERY Case Report

Idiopathic lumbar epidural lipomatosis: A rare cause of lumbar canal stenosis

Govindan Thiagarajan, Sivashanmugam Dhandapani

Abstract: Idiopathic lumbar epidural lipomatosis is relatively rare and is one of the causes of lumbar canal stenosis. Most reports found in the literature have an association, either with chronic steroid use or with co-existent obesity. This article describes one such case with co-existing obesity. The available literature is reviewed. (p117-118)

Key words: Lumbar canal stenosis, intermittent neurogenic claudication, spinal epidural lipomatosis, idiopathic and obesity.

Introduction Epidural lipomatosis is a rare disorder, mostly reported in T2- (Fig. 2) and fat suppression (Fig. 3) magnetic resonance those with either chronic exogenous steroid administration imaging sequences of the lumbar spine showed extensive or increase in endogenous steroids due to endocrino- fat deposition in the epidural space, all around the dural sac, pathies.2,6,9 Most cases have associated co-existent obesity. constricting the dural sac from L3 to S1 level. The inter- Some are truly idiopathic.1,7 This article describes one such vertebral discs and the vertebral bodies were normal, as case and reviews the literature. were the dimensions of the bony canal.

Case Report Mr. J, a 56-year-old Tanzanian national, developed intermit- tent neurogenic claudication, about 2 years prior to reporting at our institution. By the time he presented to us his walking distance had drastically reduced. He had no history to suggest any previous trauma, chronic drug use or bladder/ bowel dysfunction. Examination revealed a very obese individual, with no neurological signs at rest. On being made to walk, which he could barely do for 20 feet, he could not walk further due to the pain in the gluteal and calf Figure 1 - MRI T1-weighted image - Axial. muscles, with decreased sensation in both legs and feet. No vascular deficit was evident pre or post exercise. The spinal movements were within normal limits. He weighed 110 kgs and his height was 170 cms. The body mass index (BMI) being 38.1 kg/sq.m.

Plain x-rays of the lumbar spine were normal. T1- (Fig. 1),

Department of Neurosurgery Pondicherry Institute of Medical Sciences Kalapet, Pondicherry - 605014 India

Correspondence: Dr. Govindan Thiagarajan Figure 2 - MRI T2-weighted image - Sagittal. Figure 3 - MRI Department of Neurosurgery fat suppression image - Sagittal. Pondicherry Institute of Medical Sciences Kalapet, Pondicherry - 605014 India He underwent a decompressive laminectomy from L3 - L5. Email: [email protected] / [email protected] The usual difficulties encountered in patients with bony

VOLUME 14, NO. 2, OCTOBER 2010 117 IDIOPATHIC LUMBAR EPIDURAL LIPOMATOSIS • Thiagarajan & Dhandapani canal stenosis and ligamental hypertrophy was notably preponderance(9), with an isolated report in a female, with absent. Bony and ligamental decompression revealed exten- no obesity.8 Though both the dorsal and lumbar locations sive fat deposition all around the dural sac (Fig. 4), from L3 are equally affected, the dorsal locations present at a much to the upper part of S1. The fat was removed piecemeal, earlier age than the lumbar region. Magnetic resonance decompressing the dural sac satisfactorily. The postopera- imaging is the investigation of choice.4,9,10 tive period was uneventful. At discharge, he was free of the original symptoms and could walk without any pain or The compression may be from the extra dural fat itself or decreased sensation around the entire hospital complex. He due to the engorged epidural veins, secondary to compres- was advised graded increase in physical activity and weight sion by the fat.5 While surgical decompression has been the reduction. preferred mode of therapy, either decompressive lamin- ectomy, with excision of the fat or endoscopic removal of fat, few cases have been treated by reduction in weight; reduced fat in diet, and reduction/discontinuance of steroid intake.1,2,6,7,9

References 1. Fan CY, Wang ST, Liu CL, Chang MC, Chen TH: Idiopathic spinal epidural lipomatosis. J Chinese Med Assoc 2004, 67(5): 258-261 2. Fassett DR, Schmidt MH: Spinal epidural lipomatosis: a review of its causes and recommendations for treatment. Neurosurg Focus 2004, 16(4): E11 3. Frank E: Endoscopic suction decompression of idiopathic epidural lipomatosis. Surg Neurol 1998, 50(4): 333-335 4. Ishikawa Y, Shimada Y, Miyakoshi N, Suzuki T, Hongo M, Kasukawa Y, Okada K, Itoi E: Decompression of idiopathic Figure 4 - Peroperative view. lumbar epidural lipomatosis: diagnostic magnetic resonance imaging evaluation and review of the literature. J Neurosurg Spine 2006, 4(1): 24-30 Discussion 5. Kawai M, Udaka F, Nishioka K, Houshimaru M, Koyama T, Kameyama M: A case of idiopathic spinal epidural lipomatosis Epidural lipomatosis is one of the rare causes of neural presented with radicular pain caused by compression with compression in the dorsal and lumbar regions, presenting enlarged veins surrounding nerve roots. Acta Neurol Scand either as progressive myelopathy or radiculopathy. 2002, 105(4): 322-325 6. Lee RKT, Chau LF, Yu KS, Lai CW: Idiopathic spinal epidural lipomatosis. J HK Coll Radiol 2002, 5: 105-108 This condition is often associated with either chronic 7. Lisai P, Doria C, Crissantu L, Meloni GB, Conti M, Achene A: exogenous steroid administration or increase in endogenous Cauda equina syndrome secondary to idiopathic spinal steroids, secondary to endocrinopathies. Many have obesity as epidural lipomatosis. Spine 2001, 26(3): 307-309 an associated finding.6,9,10 Body mass index was found to 8. Ohta Y, Hayashi T, Sasaki C, Shiote M, Manabe Y, Shoji M, 9 Abe K: Cauda equina syndrome caused by idiopathic sacral be high in most cases (27.5 kg/sq.m). While there was epidural lipomatosis. Intern Med 2002, 41(7): 593-594 strong correlation to the BMI and the extent of the level of 9. Robertson SC, Traynelis VC, Follett KA, Menezes AH: Idio- epidural lipomatosis, there was no relation to the BMI and pathic spinal epidural lipomatosis. Neurosurg 1997, 41(1): 68- the thickness of the lipomatosis at any given level.4 74; Discussion 74-75 10. Sato M, Yamashita K, Aoki Y, Hiroshima K: Idiopathic spinal epidural lipomatosis. Case report and review of the literature. Some cases are truly idiopathic. There is a definite male Clin Orthop Relat Res 1995, 320: 129-134

118 PAN ARAB JOURNAL OF NEUROSURGERY Case Report

Pituitary apoplexy following open cholecystectomy

Pralaya K Nayak1, K Mohini Rao2

Abstract: Pituitary apoplexy is an uncommon but well-described clinical syndrome resulting from haemorrhage into a pituitary adenoma. A 35-year-old lady presented to us with pituitary apoplexy following open cholecystectomy. Computed tomography scan of brain showed a giant pituitary macro adenoma with intratumoural haemorrhage. High degree of suspicion in the diagnosis of pituitary apoplexy is required in a patient who deteriorates in consciousness or symptoms of headache and visual disturbances developing following any kind of surgery.

Key words: Pituitary apoplexy, pituitary adenoma, complication and cholecystectomy. (p119-121)

Case Report A 35-year-old lady underwent open cholecystectomy for cle were noted. Retro sellar extension with compressive gall stone disease whose consciousness deteriorated in the effect on midbrain was seen. postoperative period. She had transient hypotension follow- ing surgery. These symptoms developed 5 days prior to admission to our institute. She had no specific previous medical history related to pituitary tumour. Initial neuro- logical evaluation revealed of E2, V2 and M3. There was sluggish pupillary light reflex and downward conjugate deviation of both eyes.

Computed tomography scan of brain showed a giant pitui- tary macro adenoma with intratumoral haemorrhage producing mass effect over hypothalamic area (Fig. 1). Magnetic resonance imaging was performed for surgical planning with FSE, T1, T2, flair, diffusion and GRE sequences along with contrast enhancement. Magnetic resonance imaging revealed T1W isointense lesion with intralesional hyperintense component having both intra and supra sellar extension. T2- weighted sequence showed heterogeneous hyperintensity and evidence of blood fluid level with decomposed blood product, TIC showed mild heterogeneous enhancement. Para sellar extension with partial encasement of the carotid arteries was noted. Superior extension with complete efface- Figure 1 - Scan showing pituitary macro adenoma with intratu- ment of the supra sellar cistern, compressive displacement moural haemorrhage. of optic chiasma, tract and the anterior recess of third ventri- Patient was operated by transsphenoidal approach under operating microscope. Near total tumor decompression was achieved and patient was kept on steroid and oral thyroid 1Department of Neurosurgery hormone supplement. 2Department of Pathology Neelachal Hospital Her hormone profile was T3-69 (58 - 159) ng/dl, T4-5.47 India (4.9 - 11.7) µg/dl, TSH-5.7 (0.35 - 4.94) µIU/ml, GH-26.2 Correspondence: (0 - 5) ng/ml, PRL-17.3 (1.2 - 29.93) ng/ml and cortisol- Dr. Pralaya Kishore Nayak Neelachal Hospital 6.69 (5 - 25) mcg/dl. Postoperative values were T3-31 (58 - Kharvel Nagar, Bhubaneswar-751001 159) ng/dl, T4-6.4 (4.9 - 11.7) µg/dl, TSH-0.56 (0.35 - 4.94) India Tel: (91 986) 125 1219 µIU/ml, GH-13 (0 - 5) ng/ml and the rest were within Fax: (91 674) 253 6593 normal limits. Test for insulin-like growth factor-1 (IGF-1) Email: [email protected] was not done.

VOLUME 14, NO. 2, OCTOBER 2010 119 PITUITARY APOPLEXY • Nayak & Rao

Histopathology study revealed sinusoidal diffuse pattern of however, diabetes insipidus is exceptional. In less acute monomorphic population of cells. Cells showed oncocytic forms, the sudden nature of the headache and ophthalmol- change, ill-defined outline with prominent vascularity. Dual ogy signs can suggest diagnosis. population of acidophilic and basophilic cells was seen. Fibrous stromal tissues with few normal looking chromo- In pituitary apoplexy, hypertension may be an important phobe acinar clusters were seen. Large thin walled congested, predisposing factor.8 Patients who present with unusual dilated, thrombosed blood vessels and haemorrhage was neurological symptoms after general anaesthesia should un- present. Immuno stains showed GH immunoreactive score dergo neurological and radiological investigations.10 4 which suggested a growth hormone pituitary macro adenoma. Standard x-ray reveals destruction of the sella turcica. Computed tomography shows either a haematoma or a Patient did not improve neurologically and succumbed to cystic cavity in the pituitary gland which must be perfectly the disease on 7th postoperative day. described together with the integrity of the bone structures. Magnetic resonance imaging is an essential tool, which can Discussion be used to ascertain the volume and suprasellar extension of Pituitary apoplexy is a rare complication of pituitary tumours.9 the tumour, its texture, possible compression of adjacent Since first introduced by Bailey in 1898, the incidence of structures and determine the age of the haemorrhage. This pituitary apoplexy varies from 0.6 - 22.8% and it is 2 - 10% imaging technique can also isolate rare optochiasmatic of operated adenomas.5,1 Pituitary apoplexy results from apoplexy requiring intracranial evacuation.1 Circulating necrosis or haemorrhage of a pituitary adenoma. insulin-like growth factor-1 (IGF-1) is increasingly being used as a screening test and in ongoing monitoring of Pituitary apoplexy has been reported to occur spontane- treated acromegaly.3 The reduction of the tumour mass as ously in the majority of cases or in association with various well as postoperative decrease in growth hormone and IGF- inducing factors.6 The various predisposing factors of pituitary I blood concentrations is the result of surgical removal of apoplexy are pituitary irradiation, alternation of intracranial the tumour.4 pressure gradients, minor head trauma, hormone therapy, pregnancy, diabetes mellitus, diabetic ketoacidosis, cerebral Emergency surgery is mandatory and recommended by angiography, anticoagulants medication, dynamic study of most authors.5 Transsphenoidal surgery is safe and effective. pituitary gland, haemodialysis and surgeries including cardiac Outcome depends on the time lapse to decompression.1 It is surgery, lumbar laminectomy, thyroidectomy and appen- indicated if there are associated abnormalities of visual dectomy.5,6 However, one isolated case report, following acuity or visual fields because when performed within 8 cholecystectomy is available in world literature. 8 days it can restore vision and consciousness.8

Pituitary apoplexy may be the first presentation of a previ- Conclusion ously undiagnosed pituitary adenoma. Pituitary apoplexy High degree of suspicion in the diagnosis of pituitary apoplexy occurring after surgery is a rare but life-threatening acute is required in a patient who deteriorates in consciousness or clinical situation following extensive haemorrhage or necrosis symptoms of headache and visual disturbances developing within a pituitary adenoma. Although many mechanisms of following any kind of surgery. pituitary apoplexy have been proposed in the literature, the 7 exact pathogenesis remains unclear. One of the patho- References physiological mechanisms that has been postulated is the 1. Berthelot JL, Rey A: Pituitary apoplexy. Presse Med 1995, 24 fall of arterial blood pressure, inducing ischaemia, followed (10): 501-3 by infarction of the pituitary gland.6 The other mechanism 2. Dubuisson AS, Beckers A, Stevenaert A: Classical pituitary is, outstrip of blood supply by the growing tumour, leading tumor apoplexy: clinical features, management and outcomes in a series of 24 patients. Clin Neurol Neurosurg. 2007, 109(1): to haemorrhagic infarct. 63-70 3. Gradišer M, Matovinović M, Vrkljan M: Decrease in growth Pituitary apoplexy is a well-known clinical syndrome char- hormone and insulin-like growth factor (IGF)-1 release and acterised by headache, meningeal irritation, visual loss, amelioration of acromegaly features after Rosiglitazone treat- 2 ment of type 2 diabetes mellitus in a patient with acromegaly. ophthalmoplegia and alterations in consciousness. The Croat Med J 2007, 48(1): 87-91 acute form results from massive intrapituitary bleeding, 4. Kalavalapalli S, Reid H, Kane J, Buckler H, Trainer P, Heald leading to headache, meningeal signs, impaired conscious- AH: Silent growth hormone secreting pituitary adenomas: IGF- ness and ophthalmology signs, and sometimes bilateral 1 is not sufficient to exclude growth hormone excess. Ann Clin Biochem 2007, 44: 89-93 blindness. Associated signs are frequent including paralysis 5. Kim JP, Park BJ, Kim SB, Lim YJ: Pituitary apoplexy due to of the oculomotor nerves, epilepsy seizure and hemiplegia, pituitary adenoma infarction. J Korean Neurosurg Soc 2008,

120 PAN ARAB JOURNAL OF NEUROSURGERY PITUITARY APOPLEXY • Nayak & Rao

43(5): 246-249 JA: Classical pituitary apoplexy: clinical features, management 6. Liberale G, Bruninx G, Vanderkelen B, Dubois E, Vandueren E, and outcome. Clin Endocrinol 1999, 51(2): 181-8 Verhelst G: Pituitary apoplexy after aortic abdominal aneurysm 9. Wang HF, Huang CC, Chen YF, Ho DM, Lin HD: Pituitary surgery: a case report. Acta Chir Belg 2006, 106(1): 77-80 apoplexy after thyrotropin-releasing hormone stimulation test in 7. Liu JK, Nwagwu C, Pikus HJ, Couldwell WT: Laparoscopic a patient with pituitary macroadenoma. J Chin Med Assoc anterior lumbar interbody fusion precipitating pituitary apoplexy. 2007, 70(9): 392-5 Acta Neurochir (Wien) 2001, 143(3): 303-306; Discussion 306- 10. Yahagi N, Nishikawa A, Matsui S, Komoda Y, Sai Y, Amakata 307 Y: Pituitary apoplexy following cholecystectomy. Anaesthesia 8. Randeva HS, Schoebel J, Byrne J, Esiri M, Adams CB, Wass 1992, 47(3): 234-6

VOLUME 14, NO. 2, OCTOBER 2010 121 Review Article

Microsurgical management of craniopharyngiomas

Mohmammad-Yashar S Kalani, Kaith K Almefty, Alim Mitha, Peter Nakaji, Robert F Spetzler

Abstract: Despite recent advances in microsurgery and skull base techniques and the availability of refined imaging and guidance modalities, craniopharyngiomas pose a major challenge to surgeons. The decision to attempt gross total resection versus subtotal resection combined with adjuvant therapies is an area of intense discourse. Herein, we illustrate the application of the orbitozygomatic, interhemispheric, transnasal and endoscopic transsphenoidal approaches to the resection of several lesions treated at our institution. A step-by-step discussion covers the various approaches and pitfalls associated with the approach. Despite advances in radiosurgery techniques and other adjuvant therapies, surgery remains the gold standard for the treatment of craniopharyngiomas. Because any given lesion may be most suitable for any one of these techniques, surgeons should feel comfortable tackling these lesions with all the techniques presented. (p12-20)

Key words: Craniopharyngiomas, microsurgery, orbitozygomatic and transsphenoidal.

Introduction Craniopharyngiomas are World Health Organization (WHO) Histologically, the tumour consists of an adamantinomatous grade I, extraaxial epithelial tumours that arise from rem- form with nests of squamous epithelium bordered by radially nants of Rathke's pouch or the craniopharyngeal duct.16 arranged cells (Fig. 1a) or a form with a microscopic Craniopharyngiomas account for 1.2 - 4% of all primary papillary architecture. Computed tomography (CT) is usually brain tumours in the population and possibly as many as the initial imaging modality used to evaluate intracranial 10% of the primary brain tumours in the paediatric popula- lesions; in the case of craniopharyngiomas, CT often shows tion.1 The incidence of craniopharyngioma has been a calcified and/or cystic lesion and possible osseous estimated at about 1.5 cases per million people per year in changes in the skull base (Fig. 1b). Magnetic resonance the United States but it may be considerably higher among (MR) imaging remains the diagnostic gold standard for the members of specific ethnic groups such as the Japanese evaluation of craniopharyngiomas, providing surgeons where an incidence of 5.25 per million has been reported.1,20 detailed information about the relationship of the tumour to The age distribution for the presentation of these lesions is eloquent structures; involvement of the sella, ventricles, bimodal with peaks between 5 - 14 years of age and again posterior fossa, optic chiasm, and identification of cystic between 50 - 74 years of age.1 Because these lesions are components that may be amenable to drainage (Fig. 1c). located near the pituitary gland and hypothalamus, the most Preoperative evaluation of the vascular anatomy using MR common symptoms at presentation are visual disturbances angiography allows surgeons to select a corridor along the and endocrine deficiencies. Involvement and obstruction of path that will minimize risk to critical arterial and venous the third ventricle can also lead to signs and symptoms of structures. increased intracranial pressure from obstructive hydro- cephalus.

Barrow Neurological Institute Division of Neurological Surgery St. Joseph’s Hospital and Medical Center Phoenix USA

Correspondence: Prof. Robert F Spetzler Barrow Neurological Institute Division of Neurological Surgery St. Joseph’s Hospital and Medical Center Figure 1a - Haematoxylin and eosin (H&E) staining of a cra- Phoenix, AZ 85003 niopharyngioma sample illustrating the nests of squamous USA epithelium bordered by radially arranged cells. (Used with Email: [email protected] permission from Barrow Neurological Institute.)

12 PAN ARAB JOURNAL OF NEUROSURGERY MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al

reported series, the overall rate of recurrence-free survival has been 80 - 90% over a 5-year follow-up regardless of treatment modality.28,30 Surgical treatment of recurrences is significantly more challenging than the treatment of the primary lesion due to scarring from the primary surgery, radiotherapy or both.

Surgical treatment of craniopharyngiomas Surgery remains the gold standard treatment of cranio- Figures 1b - Sagittal T1-weighted contrast-enhanced MRI pharyngiomas, especially large lesions that may involve and showing a midline craniopharyngioma anterior to the pituitary compress the chiasm or . The location of stalk. (c) Coronal MRI of the same lesion showing the lesion the tumour with respect to the sella, chiasm and third medial to the internal carotid arteries. (Used with permission from Barrow Neurological Institute.) ventricle heavily influences the choice of surgical approach.

Craniopharyngiomas are noted for their adherence to sur- There are numerous options for intraoperative monitoring rounding structures. Overaggressive detachment of the during the resection of suprasellar tumours, including cranio- tumour capsule can result in severe morbidity from hypo- pharyngiomas, somatosensory evoked potentials (SSEPs), thalamic/pituitary dysfunction, including diabetes insipidus electroencephalography (EEG), visual evoked potentials and weight gain, as well as visual dysfunction, memory (VEPs), microneurography and laser Doppler flowmetry deficits and pseudoaneurysm formation. For this reason, for monitoring sympathetic nerve activity.22,24,26 However, our philosophy is that tumour capsule clearly adherent to these modalities have not been shown to improve patient adjacent structures should be left in place and a less than outcomes and their value in affecting intraoperative decision- gross total resection be accepted in exchange for less making is debatable. Use of these monitoring techniques morbidity from the procedure itself. therefore depends on the availability and the surgeon’s preference. At our institution, we routinely employ SSEPs Cystic components of craniopharyngiomas include choles- and EEG monitoring during supratentorial tumour cases. terol-rich and keratin-containing fluid. During aspiration and/or drainage, care is taken not to spill these contents into Several classification systems are used for craniopharyngio- the subarachnoid space. Doing so can irritate the brain and mas.27,34 Samii’s classification system, which is based on may result in chemical meningitis and cranial nerve deficits. the vertical projection of the lesion, is one of the most If spillage occurs, perioperative intravenous steroids and widely accepted and is used here (Table 1).29 thorough intraoperative irrigation of the subarachnoid space with a steroid-containing solution or copious amounts of Table 1 - Samii’s classification system for craniopharyngiomas.29 normal saline may help prevent symptoms and complications. Type Location I Intrasellar Frequently, craniopharyngiomas extend into the third ven- Infradiaphragmatic tricle. Craniopharyngiomas that partially extend into the II Extension into cistern ± intrasellar component third ventricle can be resected using the modified orbito- III Extension into lower half of third ventricle zygomatic , with the intraventricular component IV Extension into upper half of third ventricle being resected through a liberal opening in the lamina V Extension into septum pellucidum or lateral ventricles terminalis. In the rare instance of a craniopharyngioma that is entirely within the third ventricle, a subfrontal trans- laminar terminalis approach through a bifrontal craniotomy Although craniopharyngiomas are histologically benign, can be utilized. However, the endoscopic approach is their location and intimate relationship with surrounding perhaps even better suited for tumours in the anterior eloquent structures such as the hypothalamus, pituitary, portion of the third ventricle, especially in patients with carotids and the optic apparatus provides major surgical enlarged ventricles. challenges. Strategies for treatment of these lesions typically involve gross total resection or subtotal resection It is important for surgeons to consider the anatomy of the followed by radiation, chemotherapy or both. The 10-year tumour and the extent of extension on a case-by-case basis recurrence-free survival rates after gross total resection and to use an approach that is best suited to the patient. range from 74 - 81%.6,9,31 Subtotal resection without Below we discuss some of the most frequently used ap- adjuvant therapy is associated with a recurrence-free survival proaches to craniopharyngiomas and present case illus- rate of almost 40%.6,13,28 The addition of radiotherapy trations to highlight our technique for treating these improves outcomes to almost 90%.6,13,28 In recently challenging lesions.

VOLUME 14, NO. 2, OCTOBER 2010 13 MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al

Transsphenoidal approaches nasal septum. The sinonasal mucosa is prepared with Traditionally, the transsphenoidal approach was reserved topical vasoconstrictors and local anaesthetic injection. The for intrasellar and intrasuprasellar infradiaphragmatic cranio- appropriate trajectory should be confirmed with fluoro- pharyngiomas (Figs. 2a-f).19,25 The consensus was that a scopy or image guidance.8,10 transcranial approach was preferred for supradiaphragmatic and intraventricular lesions.8,14,19 However, in 1987 Weiss A Cottle elevator is used to make a vertical mucosal described good results treating supradiaphragmatic lesions incision, approximately 2 cm in length, at the junction of with a modified transsphenoidal approach that included the the keel of the sphenoid bone and posterior nasal septum posterior portion of the planum sphenoidale, commonly (Fig. 2a). The mucosa is elevated and reflected laterally. termed the transsphenoidal transtuberulum sellae approach.32 The posterior nasal septum is pushed off the midline with Since then, a number of series have been published with the speculum, and the contralateral mucosa is reflected to good results using similar approaches.15,18,23 further expose the sphenoid ostium. The handheld speculum is replaced with a low-profile, self-retaining speculum The transsphenoidal approach offers several advantages. abutted to the face of the sphenoid bone. Rongeurs or a No brain retraction is needed, manipulation of the optic high-speed drill are used to make and enlarge the sphenoid- apparatus is minimized and the patient is spared a scalp otomy (Fig. 2b). The face of the sellae turcica is identified, incision. Postoperative cerebrospinal fluid (CSF) leakage septations within the sinus are removed, and mucosa over and the risk of meningitis are the primary risks. Compared the sellae is removed. A high-speed drill or rongeurs are to a cranial approach8, advances in image guidance, surgical used also to open the sellar floor (Fig. 2c).7,33 technique and use of the endoscope have expanded the utility of the transsphenoidal approach in the management In the classic approach, bone is removed to the upper recess of suprasellar lesions.3,5,14,17 Currently, the standard trans- beneath the tuberculum sellae. For supradiaphragmatic sphenoidal route can be recommended for infradiaphrag- lesions, however, bone removal is extended to the posterior matic lesions. The transsphenoidal transtuberulum sellae edge of the planum sphenoidale.32 The optic canals and approach is suggested for midline suprasellar lesions that do carotid prominences limit the lateral extension of the bony not encase the main vascular structures and that have no opening. A microDoppler probe may be used to localize significant lateral extension beyond the supraclinoid carotid the intercavernous carotid arteries before the dura is arteries.19 Additional consideration should be given to the opened.7 The dura is opened with a cruciate, vertical, or Y- presumed relationship of the tumour to the pituitary stalk. shaped incision (Fig. 2d). An incision is made along the Lesions anterior to the stalk should direct surgeons to a diaphragm sellae allowing access to the supradiaphragmatic transsphenoidal approach while lesions posterior to the stalk portion of the tumour. The pituitary gland and stalk are suggest a cranial approach; however, a transsphenoidal ap- dissected from the capsule of the lesion (Fig. 2e).18 In proach to lesions posterior to the stalk has been described.17 general, a wider opening facilitates early identification of critical structures. The tumour is debulked internally (Fig. Transsphenoidal microscopic approach 2f) to improve visualization of the optic nerves and chiasm. The indications for the transsphenoidal microscopic approach Sharp dissection between the tumour and normal anatomy are discussed above. The microscopic and endoscopic trans- is usually necessary and improves preservation of delicate sphenoidal approaches are essentially interchangeable; a neurovascular structures. The decision about how far to combination of the two techniques is sometimes employed, pursue removal intraoperatively must be made on a case- depending on the need, comfort and experience of the by-case basis but in general minimal disruption of the surgeon. hypothalamus must be the overriding priority.14

The patient is placed in the supine position with the head in Multilayer reconstruction is essential to prevent a CSF leak. a horseshoe headholder or rigid pin fixation and angled 30° Abdominal fat is used for packing, and septal bone is used away from the surgeon. For purely sellar lesions, the head to repair defects in the sellar floor and planum sphenoidale may remain in the neutral plane but for suprasellar lesions it (Fig. 2g). Additional fat or a fibrin sealant may be left in should be extended 10 - 15°. The nostril contralateral to the the sphenoid sinus. The use of a posterior nasal artery- side of maximal tumour projection is selected as the main based septomucosal flap may aid in obtaining a durable route, with both nostrils available for bimanual work. watertight closure. Postoperatively, CSF drainage with a Alternatively, a sublabial approach may be used but is not lumbar drain is often necessary.18 preferred. The nostril is explored with the use of a handheld speculum, and the middle and inferior turbinate are Endoscopic endonasal approach identified. The speculum is placed in the trajectory of the As noted, the endoscopic endonasal approach is suitable for middle turbinate to the keel of the sphenoid and posterior any craniopharyngioma that would be approached through

14 PAN ARAB JOURNAL OF NEUROSURGERY MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al

A B C

D E F

Figures 2a - Vertical mucosal incision at the junction of the keel of the sphenoid bone and posterior nasal septum. (b) Rongeurs are used to make the sphenoidotomy. (c) Rongeurs or a (d) high-speed drill are used to open the floor of the sella. (e) The dura is opened in a Y-shaped incision. The pituitary gland and stalk are dissected from the capsule of the tumour, allowing visualization of the optic nerves and chiasm. (f) The tumour is internally debulked. (g) Fat is used to pack the defect and the incision is closed in layers so that it is water tight. (Used with permission from Barrow Neurological Institute.)

G the microscopic transsphenoidal technique. The operative middle turbinate is pushed laterally and the endoscope is technique is described in detail by de Divitiis et al, and is handled by the assistant or otolaryngological surgeon to performed in a similar manner with some variation.4 allow the primary surgeon to use both hands for dissec- tion.17 A sphenoidectomy is performed and must be wider An approximately 18 cm long, 0° rigid endoscope is the pri- than for standard sellar approaches.4 The need for a poste- mary optic instrument for the procedure; 30° and 45° endo- rior ethmoidectomy to achieve the sphenoidectomy varies scopes may be used as adjuncts but are seldom necessary. among surgeons.10,17 Septa within the sphenoid cavity are Image guidance is used universally by us for the approach then removed. and should be positioned so the surgeon can view the endoscopic and image guidance screens simultaneously.4,17 After the wide sphenoidotomy is completed and the sphe- noid septa are removed, the sellar face is removed. Next, The patient is placed in the supine position with the use of the tuberculum sella and planum sphenoidale are removed three-point Mayfield-Kees skeletal fixation, and the head is for 1 - 2 cm with the use of a high-speed diamond-bit drill extended 10 - 15° and rotated toward the surgeon 10 - and Kerrison rongeurs.3 The bone removal is limited 15°.2,17 The procedure begins with lateralization or resec- laterally by the presence of the optic nerve, which is at a tion of the middle turbinate on the right side followed by distance of 14 - 18 mm at the level of the tuberculum removal of the posterior nasal septum. The contralateral sellae.10 A horizontal dural incision is made in the sellar

VOLUME 14, NO. 2, OCTOBER 2010 15 MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al dura below the intracavernous sinus. A second horizontal exposure and trajectory. A 5 - 10 cm linear skin incision is incision is made in the dura of the planum sphenoidale placed in the forehead anterior to the hairline (Fig. 3a). The above the intracavernous sinus. The intracavernous sinus is bone flap should be positioned as low as possible while still controlled with clips or coagulation10,17, and the dura is cut. avoiding the frontal sinus. When the sinus cannot be The resulting two dural flaps are reflected laterally. The avoided, it should be identified early and obliterated. Next, diaphragm sella is also cut to widen the exposure. one or two burr holes are placed adjacent to the superior sagittal sinus and a craniotome is used to cut a bone flap The pituitary gland is gently pushed posteriorly to provide a (Fig. 3b). The dura is opened and reflected over the sagittal supraglandular route and to avoid the need to cut the pitui- sinus. The medial aspect of the frontal lobe and falx are tary stalk.3 A prechiasmatic suprasellar craniopharyngioma then retracted to enhance exposure; the use of fixed retrac- should be visible at the time that the dura is opened. After tors is avoided. internal debulking is completed, the tumour is removed with meticulous dissection, taking care around the optic chiasm, stalk and superior hypophysis.17 Preservation of the arachnoidal plane protects the anterior communicating artery complex, which is typically shielded by an arach- noidal sheet above the chiasm.3 Craniopharyngiomas that extensively involve the anterior communicating artery com- plex should be considered for a cranial approach.

The need for extensive exposure to approach craniopharyn- giomas has tested the cranial base reconstructive techniques of the endoscopic endonasal approach. A number of tech- niques have been used with acceptable results. Regardless of the specific technique used, the reconstruction should be multilayer and should include a packing layer to prevent graft migration.2,3,5,10,17 Hadad et al, recently described reconstruction with the use of a vascularised local flap.12 In this technique, a subdural graft is placed followed by an onlay graft in contact with a denuded osseous surface and sutured in place. Then, a nasal septal flap harvested during the exposure is positioned over the defect. Finally, a biological glue and Surgicel are placed, and the reconstruc- tion is secured with the balloon of a Foley catheter to Figures 3a - Skin incision and sites of burr hole placement for prevent migration. the interhemispheric approach. (b) The dura is opened and a retractor is placed to enhance exposure. (Used with permis- Interhemispheric approach sion from Barrow Neurological Institute.) The interhemispheric approach is used for tumours with large ventricular extensions. These patients frequently Transcallosal approach: The patient is placed in the present with obstructive hydrocephalus. The interhemi- supine position with a bump under one shoulder and the spheric approach may be further subdivided into precallosal head rotated until the superior sagittal sinus is parallel to the and transcallosal approaches, and each is used depending floor, and with the neck bent away from the floor to bring on the location and extent of ventricular spread. The the midsagittal plane 45° with respect to the floor. This precallosal approach is used for craniopharyngiomas that position allows the lower hemisphere to fall away from the have a large anterior and superior extension and that bow midline under gravity while the upper one is supported by forward between the hemispheres, often involving the optic the falx. Whichever side the tumour is biased toward is nerve complex. The transcallosal approach is preferred for placed on the uppermost side of the approach. The skin craniopharyngiomas that have a large superior extension incision is placed just anterior to the coronal suture in the into the third ventricle and lateral ventricles and are not in a midline. Intraoperative image guidance may be used to direct line for an extended transsphenoidal approach or do identify the location of the suture and sagittal sinus. Next, not have a substantial sellar component. one or two burr holes are placed adjacent to the superior sagittal sinus, and a craniotome is used to remove the bone Precallosal approach: The patient is placed supine with flap which is centered to be two-thirds in front and one- the head at 0° of rotation and extended to optimize the third behind the coronal suture. The dura is opened and

16 PAN ARAB JOURNAL OF NEUROSURGERY MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al secured on the edge of the sinus. Preoperative imaging is that extend anterior or lateral to the intradural carotid arter- used to identify the presence of large bridging veins to tailor ies. For tumours with significant third ventricular upward the opening to the path of least resistance, and the safest extension, this approach may need to be combined with conduit is selected between the bridging veins. Additional another approach, either at the same sitting or as a distinct exposure may be obtained by dissecting the arachnoid procedure. overlying bridging veins so they may be preserved. Self- retaining retractors are seldom required in this approach. Most craniopharyngiomas can be approached with an The space between the two pericallosal arteries on the top orbitozygomatic approach from the right side unless the of the corpus callosum is developed to provide a good tumour has a significant left-sided component, in which corridor into the ventricle. case a left-sided or bifrontal approach can be used. For most lesions treated at our institution, we prefer a modified Upon entry into the ventricle, the first landmark that the orbitozygomatic craniotomy, which improves access to the surgeon visualizes is the choroid plexus, which overlies the skull base and visualization of the superior aspect of the choroidal fissure. Next, the surgeon must find important tumour through an upward trajectory, and also requires less anatomical structures which include the choroid plexus, retraction of the frontal lobe.11,21 For this approach, the thalamostriate vein, anterior septal vein and anterior caudate patient is positioned supine with the head in a Mayfield vein (Fig. 3c). These structures are used to orient and holder and rotated 30 - 45° to the contralateral side. The identify the fornix which should be respected and positioning varies based on the extent of the lesion. The manipulated minimally to avoid damage to the memory. head is also extended slightly, with the malar eminence at The tumour is then identified and directly dissected and the highest point. This position allows the frontal lobe to debulked. It may be necessary to cauterize and remove fall away naturally from the skull base and prevents blood remnants attached to the ventricular walls. As with other from entering the operative field. The bed should be posi- approaches, a conservative approach must be taken to any tioned with 10° of incline in reverse Trendelenbeg, or the tumour that is strongly adherent to the hypothalamus, fornix patient can be flexed at the hips to improve venous drainage. or other delicate structures. After the tumour is resected, the dura is closed in the usual fashion. The bone flap is secured The skin incision begins 1 cm in front of the tragus at the with titanium microplates and screws. The bony defects are level of the zygomatic arch (Fig. 4a). Beginning the filled, prior to a layer-by-layer closure of the skin. incision this far posteriorly preserves the anterior branch of the superficial temporal artery and the frontotemporal branch of the facial nerve. The incision is carried superiorly and begins its curve anteriorly just above the level of the superior temporal line. It ends just medial to the midpu- pillary line and behind the hairline on the contralateral side.

Figure 4a - The skin incision used for the orbitozygomatic Figure 3c - When entering the ventricle, the surgeon must approach. (Used with per- make note of critical structures such as the choroid plexus, mission from Barrow Neuro- thalamostriate vein, anterior septal vein, and anterior caudate logical Institute.) vein when debulking the ventricular component of the tumour. (Used with permission from Barrow Neurological Institute.)

Orbitozygomatic and modified orbitozygomatic approaches The orbitozygomatic approach is the preferred subfrontal approach for tumours that are predominantly suprasellar The incision is made to the depth of the bone above the and have significant subarachnoid extension, particularly superior temporal line and through the temporalis fascia and involving neurovascular structures at the anterior skull base. muscle inferior to the superior temporal line. A rim of This includes tumours with direct connection to the anterior temporal muscle and fascia is left attached to bone to facili- cerebral artery or anterior communicating artery complex or tate anatomic apposition of the elevated muscle at closure.

VOLUME 14, NO. 2, OCTOBER 2010 17 MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al

Avoiding extensive cauterization on the temporalis fascia zygomatic suture and connects with the pterional cranio- and muscle helps to prevent shrinkage that can make tomy margin inferior to the pterion. The final cut is made closure more difficult and atrophy that can result in a poor through the posterior aspect of the lesser sphenoid wing into cosmetic result. The myocutaneous flap is retracted anteri- the superolateral aspect of the superior orbital fissure. Alter- orly using fish hooks attached to a Leyla bar. The periorbita natively, the orbitozygomatic osteotomy can be removed is dissected free from the overlying orbital roof and lateral after the first two cuts using an osteotome to propagate the orbital wall using a periosteal elevator, taking care not to fracture in the orbital roof. Rongeurs or a high-speed drill is breach the periorbital tissue. needed to resect additional bone from the lesser sphenoid wing up to the lateral aspect of the superior orbital fissure. Bone removal is performed as a one or two-piece proce- dure. The two-piece technique is described here. It begins After removal of the orbitozygomatic osteotomy, tenting with a pterional craniotomy followed by a limited orbitozy- sutures are placed through the outer layer of dura and into gomatic osteotomy (Fig. 4b). For the pterional craniotomy, the bone surrounding the craniotomy at about 2 cm two burr holes are placed: one at the keyhole and one intervals to prevent epidural blood from entering the field posteriorly in the temporal bone above the root of the and to prevent a postoperative epidural haematoma. The zygomatic arch. The bone is stripped of its underlying dura dura is separated from any remaining bone of the lateral in the region of the burr holes using the footplate attach- aspect of the lesser sphenoid wing, which then can be ment from the high-speed drill or a dural elevator. removed with a drill or rongeur to provide a flat trajectory to the suprasellar region. The dura itself is opened as an inferiorly based flap and is tented upward to the myo- cutaneous flap using 4 - 0 Nurulon sutures.

Under the operating microscope, the anterior clinoid process and ipsilateral optic nerve are first identified. The Figure 4b - In the modified orbitozygomatic approach, opticocarotid cistern is opened to drain CSF to provide the bone is removed in a brain relaxation. The arachnoid between the frontal lobe two-piece fashion. (Used and optic nerves is detached, and the lamina terminalis is with permission from Barrow identified and fenestrated to drain additional CSF. The Neurological Institute.) tumour itself is visualized through the opticocarotid cistern, and accessible cystic components are aspirated to permit immediate decompression and to facilitate dissection of the tumour from surrounding structures (Fig. 4c).

The burr holes are connected using a high-speed drill with the footplate attachment to create the pterional craniotomy. The frontal cut is made first, beginning in the keyhole and staying as low as possible just over the orbital rim. Drilling proceeds posteriorly in a curved fashion to then turns inferoposteriorly to the posterior burr hole above the root of the zygoma. The final cut proceeds anteriorly to the keyhole. Occasionally, the medial part of the frontal cut enters the lateral extent of the frontal sinus. If this occurs, the frontal sinus should be covered with a pericranial graft.

The modified orbitozygomatic osteotomy is created by three cuts of the sagittal saw, which are performed while protecting the globe and periorbital tissue. The first cut begins in the orbital roof, just lateral to the supraorbital notch and in line with the anterior extent of the pterional craniotomy. It is carried through the thickest part of the orbital rim and as far back along the orbital roof as possible, Figure 4c - The orbitozygomatic approach allows exposure of the ideally to the superior orbital fissure. The second cut is anterior cranial base and visualization of the tumour and visual appa- made through the lateral orbital wall along the fronto- ratus. (Used with permission from Barrow Neurological Institute.)

18 PAN ARAB JOURNAL OF NEUROSURGERY MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al

Often, part of the tumour can be accessed between the optic 3. de Divitiis E, Cappabianca P, Cavallo LM, et al: Extended nerves. Occasionally, however, a pre-fixed chiasm with endoscopic transsphenoidal approach for extrasellar cranio- short optic nerves can preclude this manoeuvre. Often pharyngiomas. Neurosurg 2007, 61(5 Suppl 2): 219-27; some combination of windows through the interoptic space, Discussion 228 4. de Divitiis E, Cavallo LM, Cappabianca P, Esposito F: Extended opticocarotid cistern, oculomotor triangle, lamina termi- endoscopic endonasal transsphenoidal approach for the nalis, and above the carotid bifurcation is used to access the removal of suprasellar tumors: Part 2. Neurosurg 2007, 60(1): tumour as needed. If the tumour has a significant solid 46-58; Discussion 58-9 component, the tumour should first be debulked internally 5. Dehdashti AR, Ganna A, Witterick I, Gentili F: Expanded endoscopic endonasal approach for anterior cranial base and using one or a combination of dissectors, pituitary rongeurs, suprasellar lesions: indications and limitations. Neurosurg CO2 laser, and ultrasonic aspirator. The capsule is carefully 2009, 64(4): 677-87; Discussion 687-9 dissected free from the surrounding tissue, during which the 6. Duff J, Meyer F, Ilstrup D, et al: Long-term outcomes for surgically pituitary infundibulum is identified. Care is taken to avoid resected craniopharyngiomas. Neurosurg 2000, 46(2): 291-305 7. Fatemi N, Dusick JR, de Paiva Neto MA, Kelly DF: The endo- excessive manipulation of important surrounding structures, nasal microscopic approach for pituitary adenomas and other including the hypothalamus, pituitary stalk, optic nerves, parasellar tumors: a 10-year experience. Neurosurg 2008, 63 medial basal frontal lobes and carotid arteries. As has been (4 Suppl 2): 244-56 emphasized for each previous approach, sharp dissection, 8. Fatemi N, Dusick JR, de Paiva Neto MA, Malkasian D, Kelly DF: Endonasal versus supraorbital keyhole removal of cranio- staying on the margin of the tumour and leaving tumour pharyngiomas and tuberculum sellae meningiomas. Neurosurg where it is densely adherent is recommended. Otherwise, 2009, 64(5 Suppl 2): 269-84; Discussion 284-6 severe morbidity can result, making complete macroscopic 9. Fischer EG, Welch K, Shillito J Jr, Winston KR, Tarbell NJ: removal a Pyrrhic victory at best. Craniopharyngiomas in children. Long-term effects of conser- vative surgical procedures combined with radiation therapy. J Neurosurg 1990, 73(4): 534-40 After the lesion is resected, careful attention is paid to 10. Frank G, Pasquini E, Doglietto F, et al: The endoscopic haemostasis. The resected area is inspected thoroughly and extended transsphenoidal approach for craniopharyngiomas. irrigated with normal saline. The dura is closed in a Neurosurg 2006, 59(1 Suppl 1): ONS75-ONS83 11. Gonzalez LF, Crawford NR, Horgan MA, et al: Working area watertight fashion using running 4 - 0 Nurulon sutures. If and angle of attack in three cranial base approaches: pterional, the dura has contracted or been torn, further closure can be orbitozygomatic, and maxillary extension of the orbitozygomatic obtained using an appropriate dural substitute. After the approach. Neurosurg 2002, 50(3): 550-5; Discussion 555-7 dura is closed, a central tenting suture is brought out 12. Hadad G, Bassagasteguy L, Carrau RL, et al: A novel recon- structive technique after endoscopic expanded endonasal ap- through two small holes created in the bone flap. Both the proaches: vascular pedicle nasoseptal flap. Laryngoscope orbital osteotomy and the pterional craniotomy flap are 2006, 116(10): 1882-6 reapproximated using microplates and screws, and the 13. Hetelekidis S, Barnes PD, Tao ML, et al: 20-year experience in central dural tenting suture is tied. The temporalis muscle childhood craniopharyngioma. Int J Radiat Oncol Biol Phys 1993, 27(2): 189-95 and fascia are sutured separately using interrupted 2 - 0 14. Honegger J, Tatagiba M: Craniopharyngioma surgery. Pituitary Vicryl sutures, and the galea is reapproximated using inter- 2008, 11(4): 361-73 rupted, inverted 2 - 0 Vicryl sutures. The skin is closed with 15. Kaptain GJ, Vincent DA, Sheehan JP, Laws ER Jr: Trans- running 3 - 0 nylon suture. sphenoidal approaches for the extracapsular resection of midline suprasellar and anterior cranial base lesions. Neurosurg 2008, 62(6 Suppl 3): 1264-71 Conclusions 16. Karavitaki N, Cudlip S, Adams CB, Wass JA: Craniopharyngio- Craniopharyngiomas remain a treatment challenge. The mas. Endocr Rev 2006, 27(4): 371-97 17. Kassam AB, Gardner PA, Snyderman CH, et al: Expanded advent of microsurgical and skull base techniques supple- endonasal approach, a fully endoscopic transnasal approach mented with novel imaging modalities has improved for the resection of midline suprasellar craniopharyngiomas: a approaches and treatment for these challenging lesions. It is new classification based on the infundibulum. J Neurosurg important that surgeons treating these lesions be comfort- 2008, 108(4): 715-28 18. Kato T, Sawamura Y, Abe H: Transsphenoidal-transtuberculum able with each of the approaches described above and work sellae approach for supradiaphragmatic tumors. No Shinkei with a multidisciplinary team of oncologists and radiation Geka 1998, 26(7): 583-8 experts to provide patients with the optimal treatment 19. Komotar RJ, Roguski M, Bruce JN: Surgical management of option for their particular lesion. craniopharyngiomas. J Neurooncol 2009, 92(3): 283-96 20. Kuratsu J, Ushio Y: Epidemiological study of primary intra- cranial tumors in childhood. A population-based survey in References Kumamoto Prefecture, Japan. Pediatr Neurosurg 1996, 25(5): 1. Bunin GR, Surawicz TS, Witman PA, et al: The descriptive 240-6; Discussion 247 epidemiology of craniopharyngioma. J Neurosurg 1998, 89(4): 21. Lemole GM Jr, Henn JS, Zabramski JM, Spetzler RF: Modi- 547-51 fications to the orbitozygomatic approach. Technical note. J 2. Cavallo LM, Prevedello DM, Solari D, et al: Extended endo- Neurosurg 2003, 99(5): 924-30 scopic endonasal transsphenoidal approach for residual or 22. Lin KL, Wang HS, Lui TN: Diagnosis and follow-up of cranio- recurrent craniopharyngiomas. J Neurosurg 2009, 111(3): 578-89 pharyngiomas with sonography. J

VOLUME 14, NO. 2, OCTOBER 2010 19 MICROSURGICAL MANAGEMENT OF CRANIOPHARYNGIOMAS • Spetzler, et al

Ultrasound Med 2002, 21(7): 801-6 long-term results following limited surgery and radiotherapy. 23. Maira G, Anile C, Albanese A, et al: The role of transsphenoidal Radiother Oncol 1993, 26(1): 1-10 surgery in the treatment of craniopharyngiomas. J Neurosurg 29. Samii M, Tatagiba M: Surgical management of craniopharyn- 2004, 100(3): 445-51 giomas: a review. Neurol Med Chir (Tokyo) 1997, 37(2): 141-9 24. Nau HE, Bock WJ, Clar HE: Electroencephalographic investi- 30. Symon L, Sprich W: Radical excision of craniopharyngioma. gations in sellar tumours, with special regard to different Results in 20 patients. J Neurosurg 1985, 62(2): 174-81 methods of operative treatment. Acta Neurochir (Wien) 1978, 31. Van Effenterre R, Boch AL: Craniopharyngioma in adults and 44(3-4): 207-14 children: a study of 122 surgical cases. J Neurosurg 2002, 97 25. Norris JS, Pavaresh M, Afshar F: Primary transsphenoidal (1): 3-11 microsurgery in the treatment of craniopharyngiomas. Br J 32. Weiss MH: Transnasal transsphenoidal approach. In: Apuzzo Neurosurg 1998, 12(4): 305-12 MLJ (ed), Surgery of the Third Ventricle. Baltimore ML, 26. Pietrangeli A, Jandolo B, Occhipinti E, Carapella CM, Morace Williams & Wilkins 1987, pp 476-94 E: The VEP in evaluation of pituitary tumors. Electromyogr 33. Zada G, Kelly DF, Cohan P, Wang C, Swerdloff R: Endonasal Clin Neurophysiol 1991, 31(3): 163-5 transsphenoidal approach for pituitary adenomas and other 27. Puget S, Garnett M, Wray A, et al: Pediatric craniopharyngio- sellar lesions: an assessment of efficacy, safety, and patient mas: classification and treatment according to the degree of impressions. J Neurosurg 2003, 98(2): 350-8 hypothalamic involvement. J Neurosurg 2007, 106(1 Suppl): 3-12 34. Zhou L, You C: Craniopharyngioma classification. J Neurosurg 28. Rajan B, Ashley S, Gorman C, et al: Craniopharyngioma - a 2009, 111(1): 197-9

20 PAN ARAB JOURNAL OF NEUROSURGERY Neuropathological Feature

The 2007 WHO Classification of Tumours of the Central Nervous System, 4th Edition: Recent advances in Diagnosis and Classification

In the previous edition of this Journal, the features of meningioma with brain invasion, WHO Grade II, and the relationship of GBM to anaplastic mixed oligoastrocytomas were briefly outlined. Continuing with the rather less frequently observed lesions in the neurosurgical practice that we receive while briefly analyzing grading changes, new clinicopathological entities, variants and pattern entries, resumption of this examination in this Journal edition will take the reader to the other established entities that had received grading changes. These include: • Atypical choroid plexus papilloma, WHO grade II, • Pineal parenchymal tumours, WHO Grades I - IV • Ganglioglioma, WHO Grade I or III • Haemangiopericytoma, WHO Grade II and anaplastic haemangiopericytoma, WHO Grade III

Choroid Plexus Neoplasms, WHO Grades I–III A subset of papillomas with worrisome histologic features and designated as ‘‘atypical choroid plexus papilloma’’, was recognized by the previous WHO edition; this neoplasm was not assigned a grade or clinical correlation, and criteria for this designation were not well developed. Based on a recent clinicopathologic study of a large number of nonmalignant choroid plexus neoplasms analyzing multivariate parameters, the investigation included atypical histologic features of these neoplasms as they related to tumour recurrence, mitoses, increased cellularity, nuclear pleomorphism, solid growth, and necrosis. The sole diagnostic criterion that was significantly predictive of tumour recurrence was the presence of >2 mitoses per 10 HPFs; there was a trend toward an increased risk of recurrence observed if any of the mentioned atypical histologic features were present. Based on the above, ‘‘atypical choroid plexus papilloma, WHO grade II’’ as a diagnostic category and an indication that the presence of mitotic activity (2 or more mitoses per 10 HPFs) should be used to establish the diagnosis in a low grade papillary neoplasm by the WHO classification, thereby further defining this neoplasm’s grade and clinical classification.

Pineal Parenchymal Tumours, WHO Grades I - IV Two major grading changes relating to neoplasms of the pineal gland have been introduced in the 2008 WHO classification. The first, involves ‘‘pineocytoma’’, which characteristically has bland appearing mature neoplastic pineocytes on histological examination, is now considered to be a WHO grade I neoplasm. The second, detailed description of pineal parenchymal tumour of intermediate differentiation; that is based on careful clinicopathologic investigation of a large series of these uncommon tumours. Such intermediate differentiation neoplasms account for at least 20% of pineal parenchymal tumours, although in the experience of others a larger figure is more likely to represent this subset of neoplasms. Sheets or large lobules of uniform appearing cells with moderate nuclear atypia and low to moderate mitotic activity is the main intermediate power magnification that histologically defines these controversially elusive neoplasms. This is partly because transitions from rosette-bearing areas (more typical of pineocytoma) to diffuse, sheet-like growth pattern may vary heterogeneously in these neoplasms depending on sampling adequacy. By-and-large, < 2 mitoses per 10 high power fields (HPF), and Ki-67 / MIB-1 proliferation indices that range from 3% to 10% is a constant overall feature in these neoplasms. The 5-year overall survival of pineal parenchymal tumour of intermediate differentiation is wide (39% to 74%), and that is an intrinsic aspect of the tedious and intangible grading process. For hair splitters, this intermediate differentiation tumour further fall into 2 prognostic groups; in the poorer prognostic category, there are those whose tumours are associated with a short survival, and there are tumours that display a high mitotic index 6 or more per HPF, recognizable necrosis, and a lack of neurofilament protein (NFP) immunostaining. The current WHO classification has designated these tumours as WHO grade II or III but has not provided strict criteria to distinguish between the 2 grades.

Ganglioglioma, WHO Grade I or III Whereas gangliogliomas were classified as either WHO grade I or II, and anaplastic gangliogliomas were considered WHO grade III in the 3rd (previous) WHO classification edition, that classification schema allowed for the grading of

122 PAN ARAB JOURNAL OF NEUROSURGERY Neuropathological Feature gangliogliomas as grade I, II, or III, but specific criteria to distinguish among the grades were not provided. In the current 4th classification, the WHO grade II has been eliminated altogether from the classification schema. Thus, gangliogliomas are now designated only as WHO grade I, and anaplastic gangliogliomas remain as grade III. Most likely, in upcoming WHO classifications schema, grade II tumours will be re-included; this is based on one of the largest and most comprehensive investigations of supratentorial gangliogliomas, which clearly suggested that grade II tumours contain cellular atypia (increased cellularity, conspicuous pleomorphism), microvascular proliferation, or an elevated MIB-1 labeling index of 5% or more. Grade III neoplasms in this investigation included the additional findings of necrosis and an MIB-1 proliferation index of 10% or more. Hence, establishment of definite criteria for gangliogliomas grade separation.

Cerebellar Liponeurocytoma, WHO Grade II When compared to medulloblastoma, the rarely occurring cerebellar liponeurocytoma which is of strikingly low proliferative potential and a favourable outcome has to be clearly distinguished on morphologic grounds. This neoplasm is characterized by being a well-differentiated cerebellar neurocytic neoplasm of adulthood that comprises focal or regional lipomatous differentiation. Whereas in the 3rd edition the neoplasm was classified as a WHO grade I, this was changed to a grade II neoplasm, based on a literature review which has indicated that as many as 60% of these neoplasms will recur within periods ranging from 1 to 12 years after a longer clinical follow-up periods as in its investigation; it has become clear that these tumours have a rate of recurrence that is higher and not therefore anymore compatible with a grade I designation than previously thought.

Haemangiopericytoma, WHO Grade II and Anaplastic Haemangiopericytoma, WHO Grade III Whereas the WHO 3rd edition failed to provide clear-cut histological features that can distinguish between grades II and III of CNS haemangiopericytoma (HPC), the 4th edition has introduced criteria for the diagnosis of anaplastic HPC, WHO grade III, which include a high mitotic activity (> 5 per 10 HPFs) and/or necrosis, and 2 or more of the following features: haemorrhage, moderate to high nuclear atypia, and high cellularity. Prior studies have demonstrated that dura-based HPCs with these ‘‘anaplastic’’ features recurred earlier and more frequently than those that did not and also had higher rates of metastasis.

Dr. Ibrahim S Tillawi, MBBCh, FCAP, FASCP Consultant in Haematopatholgy and Neuropathology Department of Pathology Riyadh Military Hospital Saudi Arabia

VOLUME 14, NO. 2, OCTOBER 2010 123 Book Reviews

The Epilepsies: Seizures, Syndromes and Management

Edited by: CP Panayiotopoulos Published: 2005 Number of pages: 560 Number of illustrations: 115 Cost: Hardcover £95 / US$170 ISBN No.: 1-904218-34-2 Publisher: Bladon Medical Publishing, Oxford, UK

The epilepsies, seizures, syndromes and management, based on the ILAE classification and practice parameter guidelines is a single author book, of 560 pages accompanied with a CD Rom with patient videos and electroencephalograms. Although the book is relatively old, printed in 2005, the contents do cover most recent advances in epilepsy. Moreover, it is the latest work from one of the world's leading experts in the field of epileptology.

In fourteen chapters, Dr. Panayiotopoulos gives information on the precise details of each seizure type including newly described types and the epileptic syndrome.

Details of epileptic seizures and syndromes demography, clinical manifestations including differential diagnosis, aetiology, diagnostic procedures, prognosis and management are explored in detail. The author describes the reflex seizures and reflex epilepsies. Principles of treatment with recommendations based on the author’s personal observations and experience; the optimal use of electroencephalography, and epilepsy imaging have been extensively covered.

On the other hand, several topics related to the epilepsy treatment have not been given the required attention such as ketogenic diet, refractory epilepsy, preoperative evaluation and epilepsy surgery. Of course, some of these topics, after the book’s publication, have had more evidences, like the ketogenic randomized controlled study published in 2008.

Many writing errors could be observed through the text; some of them in famous epilepsy surgery methods. This book is of limited benefit in the field of epilepsy surgery with only the basics in short form presented which cannot be relied on in this specialty field, but if we look at the target group for this book, we will find that the book amply achieves its goals

The book is published in full color throughout and is complemented with a CD ROM with patient video-EEGs where the students and trainees will find various fits, faints, and funny turns with split screen video EEG interpretation; these illustrative case reports make the reading of this book very interesting and encourage motivation to read the book several times.

This book is a useful reference source for neurologists, clinical neurophysiologists, epileptologists, and clinician dealing with children and adults with epileptic disorders in day-to-day practice. This book is highly recommended for scholars and teachers of epilepsy.

Alaa Eldin Elsharkawy, MSc, MD Falk Opel, MD, PhD Bethel Epilepsy Centre Germany

124 PAN ARAB JOURNAL OF NEUROSURGERY Book Reviews

Advances and Technical Standards in Neurosurgery, Vol. 34

Edited by: JD Pickard, N Akalan, C Di Rocco, VV Dolenc, J Lobo Antunes, JJA Mooij, J Schramm, M Sindou Published: 2009 Number of pages: 210 Number of illustrations: 55 in colour Cost: Hardcover 234.95 € ISBN No.: 978-3-211-78740-3 Publisher: Austria, Springer-Verlag Wien

Advances and Technical Standards in Neurosurgery, Vol. 34 is the latest of a series sponsored by the European Association of Neurosurgical Societies, mainly intended for use by training neurosurgeons. However, the depth of knowledge in the reviewed topics makes the book useful for experienced neurosurgeons. The first part of this volume presents two topics on advances in neurosurgery while the second part presents technical details of four standard neurosurgical procedures.

The first chapter of the “Advances” section is entitled “Present and Potential Future Adjuvant Issues in High-Grade Astrocytic Glioma Treatment”. The chapter is a comprehensive review of the cellular and molecular biology of malignant glioma in which the mechanisms of how invasive glioma cells acquire resistance to apoptosis and hence to conventional pro-apoptotic chemotherapy and radiotherapy are discussed. The patterns of cell death (apoptosis, autophagy and necrosis) are compared with a focus on autophagy as the alternative mechanism that can partly overcome the resistance of many cancers to pro-apoptotic-related therapies. The authors also discuss the therapeutic benefits of temozolamide and the local therapies for glioblastoma including a mention of the new convection-enhanced delivery system. They also review many recent clinical glioblastoma management trials which used growth factor receptor inhibitors, mammalian target of rapamycin (mTOR) inhibitors, angiogenesis pathways inhibitors, cellular and vaccination therapies and gene therapy. In addition the authors examine the fact that reducing the levels of malignant glioma cell motility can restore pro-apoptotic drug sensitivity, the observation that inhibiting sodium pump activity reduces both glioma cell proliferation and migration and the concept of targeting brain tumour stem cells as a complement to conventional treatment. This chapter is fairly specialized and would prove useful for neurosurgeons and neuro-oncologists interested in malignant glioma. It has a limited number of illustrations (4 figures) but well-referenced (136).

The second chapter of the “Advances” section is entitled “Deep Brain Stimulation for Psychiatric Disorders - State of the Art”. The chapter covers the use of deep brain stimulation (DBS) for therapy-resistant patients with major depression and obsessive-compulsive disorder (OCD). The neurobiology of depression and OCD, historical treatment, principles of DBS and the basis for the DBS target selection in both diseases are discussed. The chapter also includes a review of a limited number of publications that studied the use of DBS in depression and OCD in a small group of patients. The ethical aspect of DBS in neuropsychiatric disorders is reviewed. The authors conclude with the message that this technique is still in need of more clinical data and mandatory standards for patient and target selection and study protocol. This chapter is specialized and would prove useful for functional neurosurgeons. It is illustrated with 3 figures, 4 tables and has 66 references.

The first chapter of the “Technical Standards” section is entitled “High Flow Extracranial to Intracranial Vascular Bypass Procedure for Giant Aneurysms: Indications, Surgical Technique, Complications and Outcome”. The chapter provides detailed review of how the authors perform a high flow extracranial to intracranial bypass procedure. It includes a technical description of the cranial exposure, the cervical exposure, the saphenous vein exposure, the anastomoses and the postoperative care. In addition, the authors provide a useful checklist of the measures that should be considered to optimize surgical success and graft survival. They also compare the results of their small series (8 patients) with others with regards to indications, bypass type, graft patency and complications. This technique is highly specialized and only a number of

VOLUME 14, NO. 2, OCTOBER 2010 125 Book Reviews vascular neurosurgeons are trained to perform it. Nevertheless, the chapter is informative to all neurosurgeons as it draws their attention to when the technique is indicated and its limitations. The chapter ends with a clear statement that for this technique to succeed it must be approached in a stereotyped and meticulous fashion with multiple quality control checks at each phase of the procedure. The chapter is illustrated with 5 figures, 3 tables and has 38 references.

The second chapter of the “Technical Standards” section is entitled “Decompression for Chiari-I Malformation (with or without syringomyelia) by Extreme Lateral Foramen Magnum Opening and Expansile Duroplasty with Arachnoid Preservation: Comparison with Other Technical Modalities (Literature Review)”. The chapter is a fairly comprehensive review of the pathophysiology and imaging modalities used in Chiari-I Malformation (CM-1). The authors’ modification of the standard technique was by adding the “Extreme Lateral Foramen Magnum Opening”. The technique is described and a personal series of 44 cases is presented and compared to cases that had other surgical techniques. The authors did not report any vertebral artery injury during the extreme lateral exposure in their patients. They conclude that the most effective treatment for CM-1 with or without syringomyelia, is foramen magnum decompression with extreme lateral rim resection followed by dural enlargement with arachnoid preservation. This technique may be interesting but the argument for adding the extreme lateral foramen magnum opening with its potential risks, to the standard technique was not convincing. Hence the benefits of such addition remain questionable. The chapter is illustrated with 3 figures, 5 tables and has 77 references.

The third chapter of the “Technical Standards” section is entitled “Vagal Nerve Stimulation - a 15 Year Survey of an Established Treatment Modality in Epilepsy Surgery”. It is a comprehensive review of the use of vagal nerve stimulation (VNS) for refractory epilepsy. The mechanism of action of VNS, clinical efficacy, safety, side effects and tolerability are well covered. In addition, the relevant clinical trials published up to 2007 are reviewed. The chapter concludes with a clear message regarding limited efficacy of this technique and how it could be improved. This chapter is specialized and would prove useful for functional neurosurgeons. It is illustrated with 2 figures, 4 tables and has 170 references.

The fourth chapter of the “Technical Standards” section is entitled “Surgical Anatomy and Surgical Approaches to the Lateral Ventricle”. The chapter is fairly comprehensive review covering the embryology and anatomy of the lateral ventricles along with their arterial and venous vasculature, their relationships with the eloquent cortical areas and cortical sulci and their relationships with white matter fascicles especially the optic radiation. In addition, it covers technical details of the various surgical approaches that can be used for tumours in the frontal horn, the ventricular atrium and the temporal horn. The chapter is well illustrated with diagrams and images but lacks operative photographs. Complications and how to avoid them are discussed but the article lacks reference to results of surgery. This chapter will prove a useful read for the general neurosurgeon and the trainees. It is illustrated with 37 figures, 1 table and has 45 references.

In summary, this volume of the Advances and Technical Standards in Neurosurgery series covers 6 interesting specialized neurosurgical topics. The book is well referenced and apart from the last chapter, it is not heavily illustrated. As many of the authors are not native English speakers some of the passages are tedious to read. On the whole the book contains substantial information and would prove useful for trainees preparing for their final board examination. Practicing neurosurgeons may be interested in reading the topic that is related to their subspecialty.

Prof. Abdulhakim B Jamjoom, FRCS (SN) King Khalid National Guards Hospital Jeddah Saudi Arabia

126 PAN ARAB JOURNAL OF NEUROSURGERY Forthcoming Events

October, 2010 Grand Hyatt Bali, Jakarta, Bali, Indonesia Email: [email protected] 1 - 4 www.aosbs10bali.org/ 6th Int. Symposium on Neuroprotection & Neurorepair 22 - 24 Rostock, Germany 8th Asian Congress of Neurological Surgeons www.neurorepair-2010.de/ (ACNS) 2010 Kuala Lumpar, Malaysia 10 - 11 Email: [email protected] 4th Indian Japanese Friendship Neurosurgery www.ACNS2010.org Conference Cochin, Kerala, India 26 - 29 Email: [email protected] VIIIth Pan Arab Neurosurgical Society (PANS) Congress 10 - 13 Algiers, Algeria First Integrative Course on Spasticity Management Tel/Fax: (213) 21 52 49 31 Bodrum, Turkey Email: [email protected] www.vitalmedbodrum.com www.pans-algiers2010.com

27 - 30 6th European Cerebral Revascularization and December, 2010 Endovascular Stroke Treatment Course Institute of Pathology, University of Bern, Switzerland 1 - 2 Email: [email protected] Rome Spine 2010 Mtg. www.aesculap-akademie.ch Crowne Plaza St. Peter’s Hotel, Rome, Italy Email: [email protected] 27 - 30 www.romespine.org / www.formazionesostenibile.it 59th Annual Mtg. Italian Society for Neurosurgery Joint Mtg. with Neurological Society of India 1 - 4 Milan, Italy 15th Instructional Course & 38th Annual Mtg. www.sinch.it / www.csrcongressi.com/2010x5/ Cervical Spine Research Society The Westin Charlotte, North Carolina, USA www.csrs.org November, 2010 5 - 7 4 - 6 4th American-Kuwaiti Neurosurgical Mtg. 5th Int. Symposium on Microneurosurgical Anatomy Salmiya, Kuwait Istanbul, Turkey Email: [email protected] www.isma2010.org 9 - 11 5 - 7 19th Pan African Association of Neurological Int. Spinal Cord Society 49th Annual Scientific Sciences (PAANS) Congress Mtg. 2010 Tripoli, Libya New Delhi, India Email: [email protected] www.iscos.org.uk/ www.paanstripoli.ly

6 - 8 9 - 11 4th Int. Congress of Emirates Neuroscience Society Syringomyelia 2010 Int. Symposium Dubai, UAE Berlin, Germany Email: [email protected] Email: [email protected] www.syringomyelia2010.org 8 - 13 WFNS Skull Base Course / Asian-Australasian 16 - 19 Society of Neurological Surgeons (AASNS) Joint Mtg. of 59th Annual Conference of Course / 10th Asian-Oceanian Int. Congress on Neurological Society of India (NSI) & Congress of Skull Base Surgery (AOSBS) World Academy of Neurological Surgeons (CNS) Neurological Surgery (WANS) Mtg. Jaipur, India

VOLUME 14, NO. 2, OCTOBER 2010 127 Forthcoming Events

Email: [email protected] / September, 2011 [email protected] www.neurocon2010.com 14 - 17 XIV Interim Mtg. World Federation of Neurosurgical Societies XV Congress of March, 2011 Continuous Edition of Brazilian Society of Neurosurgery 2 - 4 Porto de Galinhas, Recife, Brazil American Society of Functional Neuroradiology Email: [email protected] (ASFNR) 5th Annual Mtg. 2011 Phoenix, USA www.asnr.org October, 2011

8 - 11 April, 2011 11th Congress of the World Federation of Interventional Neuroradiology (WFITN) 2011 9 - 13 Cape Town, South Africa 78th American Association of Neurological www.wfitn2011.org Surgeons’ (AANS) Annual Mtg. 2011 Denver, USA 9 - 14 www.aans.org European Association of Neurological Societies (EANS) Congress 2011 Palazzo dei Congressi, Rome, Italy May, 2011 www2.kenes.com/eans/Pages/home.aspx

8 - 12 19 - 22 10th Biennial Congress & Exhibition of the Int. EuroSpine 2011 Stereotactic Radiosurgery Society Milan, Italy Paris, France www.eurospine.org www.ISRScongress.org

128 PAN ARAB JOURNAL OF NEUROSURGERY Instructions to Authors

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VOLUME 14, NO. 2, OCTOBER 2010 129 Instructions to Authors

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VOLUME 14, NO. 2, OCTOBER 2010 131 Review Article

Current management of traumatic intracranial hypertension: A systematic approach

Mohammed H Bangash, Saleh S Baeesa

Abstract Background: Traumatic brain injury (TBI) is a major cause of mortality and morbidity in Arab countries in general, and in Saudi Arabia in particular. The efficacy and safety of most of the interventions used in the management of patients with TBI lack class I evidence and remain unproven.

Methods: A search was established using a subject heading “head injury”, “traumatic brain injury” and combined headings “brain injury and Saudi Arabia” and “brain injury and middle east”.

Results: The authors used the national library of medicine database to analyze these treatment options and the therapeutic goals of lowering intracranial pressure.

Conclusions: This review describes the systematic approach and current management practices including surgical interventions and various treatment modalities for traumatic intracranial hypertension. The results provide recommendations for intensivists and emergency physicians in Arab countries managing head injured patients. Details of these advocated treatments can be found through the literature references. (p21-28)

Key words: Head injury, intracranial hypertension and guidelines.

Introduction Traumatic brain injury (TBI) is the leading cause of death accident victims (68%).21 These statistics underscore the among young adults in Saudi Arabia. The exact incidence devastating consequences of head injury in today’s society. is not known but review of regional data by Al-Moutaery In Saudi Arabia, there is a seasonal variation regarding and Akhdar (1998) revealed that 80% of the deaths in trauma with a higher frequency during the Muslim pilgrim- Ministry of Health Hospitals are due to road traffic acci- age (Hajj) period.5 Although some advances have been dents.7 They found at that time, that TBI in Saudi Arabia made in the prevention of initial injuries after implementing resulting in death is about 17.4%, which is double that in seat belt laws a few years ago. Still the drivers’ lack of the USA (8.3%). The annual incidence of TBI in USA is safety knowledge, driving behaviours, tyre condition and approximately 200 injuries per 100,000 people with a peak timing of the emergency medical response remain impor- incidence among male subjects aged 15 - 30 years.9,20 tant factors affecting the trauma care in Saudi Arabia.4,6,51 Traumatic brain injury is the leading cause of death among The greatest impact that physicians can make on this is with people less than 24 years of age.20,41 The most frequent a quick and accurate assessment of primary insults and cause of head trauma is motor vehicle accidents (45%), ensuring prevention of secondary complications.38,62 which is also the main cause of death among motor vehicle General evaluation and stabilization Head injury exists along a continuum ranging from minor Division of Neurological Surgery scalp lacerations and abrasions to severe intracranial Faculty of Medicine King Abdulaziz University injuries. Many evaluation scales have been formulated to Jeddah assess the clinical risk condition of patients with head Saudi Arabia injury. The Glasgow Coma Scale (GCS) is the most widely Correspondence: utilised and reproducible scale used among health care Dr. Saleh S Baeesa Division of Neurological Surgery personnel (Fig. 1). In the presence of cerebral contusion, King Abdulaziz University Hospital haemorrhage or intracranial haematoma, these criteria PO Box 80215 prognosticate a worse prognosis with a mortality rate of up Jeddah 21589 36,37,42,47 Saudi Arabia to 35%. The GCS thus provides in the non-sedated Fax: (966 2) 640 8469 patient an easily reproducible assessment of the patient’s Email: [email protected] clinical condition with minimal inter-observer variability.

VOLUME 14, NO. 2, OCTOBER 2010 21 CURRENT MANAGEMENT OF SEVERE TRAUMATIC BRAIN INJURY • Bangash & Baeesa

The GCS is a tool which sums up the best scores for 3 should be intubated to maintain adequate ventilation and specific assessments: eye opening, verbal response and motor oxygenation. Prior to that, an assessment should be made response. Rapid assessment and initiation of treatment are of the presence of spontaneous respiration and respiratory of paramount importance for a patient with severe head patterns. A Cheyne-Stokes pattern of respiration may indicate injury. Drugs, hypoxia, hypothermia or hypotension may bilateral cortical involvement, while central neurogenic depress the initial GCS scores. A GCS score of 8 or less hyperventilation, ataxic "Biot" breathing, or hypoven- indicates a severe head injury and is associated with higher tilation/apnoea may be signs of midbrain, pontine and probability of increased intracranial pressure (ICP). medullary lesions, respectively. Oral endotracheal intubation is the preferred route for stabilizing the airway although a Figure 1 - Glasgow Coma Scale. nasal route may be employed. Nasotracheal intubation, Eye opening requires more time for placement, adds the additional risks of aspiration, traumatic epistaxis and obscuration of the None 1 = Even to supra-orbital pressure vocal cords and may lead to increased risk of paranasal To pain 2 = Pain from sternum/limb/supra-orbital sinusitis.8,23 If tracheal intubation is not possible due to pressure significant orofacial or airway trauma, a cricothyrotomy or To speech 3 = Non-specific response, not tracheostomy should be performed. Intubation should be of necessarily to command rapid sequence and the patient should be pre-oxygenated Spontaneous 4 = Eyes open, not necessarily aware with 100% oxygen. If necessary, morphine or midazolam (0.015 - 0.3 mg/kg) may be used for sedation at this stage. Motor response When succinylcholine is used for intubation (1.5 mg/kg), a None 1 = To any pain; limbs remain flaccid priming dose of either pancuronium (0.1 mg/kg) or mini Extension 2 = Shoulder abducted and shoulder and dose succinylcholine (0.1 mg/kg) should be given to forearm internally rotated decrease the incidence of fasciculations, which may elevate 8,23,29 Flexor response 3 = Withdrawal response or assumption ICP and also lead to emesis with aspiration. Lidocaine of hemiplegic posture (1.0 - 1.5 mg/kg) given 2 minutes before intubation has been shown to blunt the rise of ICP associated with tracheal Withdrawal 4 = Arm withdraws to pain, shoulder 8 abducts intubation. At all times cervical spine manipulation should Localises pain 5 = Arm attempts to remove supra-orbital/ be minimised. Once intubated, an arterial blood gas sample chest pressure should - if possible - be obtained to ensure a pO2 more than 70 mmHg.64 Obeys commands 6 = Follows simple commands

The other critical factor in maintaining cerebral metabolism Verbal response is the systemic blood pressure (SBP) as hypotension corre- None 1 = No verbalization of any type lates with increased morbidity and mortality.65 Adequate Incomprehensible 2 = Moans/groans, no speech intravascular access must be obtained and the SBP should Inappropriate 3 = Intelligible, no sustained sentences be maintained above 90 mmHg. It must be emphasised here Confused 4 = Converses but confused, disoriented that thorough neurological examination is meaningless as long as the patient is hypotensive.34 Head trauma, as such, Oriented 5 = Converses and oriented among adults is rarely the cause of hypotension unless massive scalp bleeding is present. Most instances of General systemic resuscitation should commence prior to hypotension in the trauma setting are due to haemorrhage observations of neurologic decline from these secondary outside the nervous system. brain injuries. More than half of all patients with severe head injuries have concomitant secondary trauma injuries, Brain herniation may be associated with hypertension and that may all increase the secondary brain lesions. Evalua- bradycardia as a terminal event (Cushing’s reflex) and tion using standard guidelines such as those provided by the cervical spinal cord injury may be the cause of hypotension advanced trauma life support protocol (ABC) should associated with bradycardia. therefore always be started immediately.44 Attention to the ABC’s of a trauma victim serves not only to stabilize vital Fluid resuscitation should be aimed at obtaining normo- cardiopulmonary function but also preserves cerebral blood volaemia through the use of lactated Ringer’s solution or flow (CBF) to a compromised brain. hypertonic (3%) saline solution, which has been shown to produce less cerebral oedema and is accompanied by lower Adequate airway must be ascertained as the first priority. ICP values.25 Recent critical reviews revealed insufficient All patients with a clinical severe head injury (GCS < 8) evidence at present to justify the use of hypertonic saline as

22 PAN ARAB JOURNAL OF NEUROSURGERY CURRENT MANAGEMENT OF SEVERE TRAUMATIC BRAIN INJURY • Bangash & Baeesa resuscitation fluid in patients with severe head injury.27 severe head injury and consequent decreased cerebral compliance can be very detrimental, as it will increase the When the patient is stabilized, attention should be directed cerebral metabolic rate (CMR) and ICP. However there is towards the thorough neurologic examination. The patient’s no statistical evidence that the use of anticonvulsants is level of consciousness is clearly the best indicator of the beneficial. Many neurosurgeons will, by tradition, extent of neurologic injury and risk for increased ICP at first administer phenytoin (18 mg/kg loading dose: 100 mg q 8 investigation. The GCS has limitations in providing an hrs maintenance dose) to avoid this complication. Although accurate assessment of neurologic complications and other anticonvulsants will not decrease the later development of features of the neurologic exam should be included in the epilepsy, phenytoin has been shown to exert a beneficial clinical examination. The GCS is solely an estimate of effect in reducing seizures during the first week after severe prognosis, and tells nothing about clinical details. A unilat- head injury.61 eral dilated pupil may indicate an oculomotor palsy due to uncal herniation. Bilateral dilated pupils may indicate Once the neurological exam has been carried out and the midbrain compression, while pinpoint reactive pupils may patient is stabilised, a head computed tomography (CT) indicate pontine involvement. Conjugate deviation of the scan should be obtained prior to completion of a formal eyes is indicative of ipsilateral frontal or contralateral secondary survey. On CT blood lesions are easily seen. If pontine lesions. Unilateral Horner’s pupil is seen occa- necessary, short acting narcotics, benzodiazepines and non- sionally with brainstem lesions, in the trauma setting, depolarising muscle relaxant drugs may be used to prevent attention should be given to the possibility of a disrupted patient movement during the scanning process as long as efferent sympathetic pathway (sympaticganglial lesion) at the airway is controlled and a satisfactory SBP is main- the apex of the lung, base of the neck or carotid sheath. tained. The CT scan in conjunction with the initial and Bilaterally fixed and dilated pupils may be the result of serial neurological exams will determine which further inadequate cerebral vascular perfusion (brain-death) often neurosurgical intervention may be required. Most patients caused by long-lasting systemic hypotension. Return of the with severe head injuries will require intensive care pupillary response may occur promptly after restoration of management with ICP monitoring. Operative indications blood pressure and thereby CBF. Oculocephalic (Doll’s are beyond the scope of this article. (Open depressed skull eyes) is a sign of midbrain lesions but testing-manoeuvres fracture or a closed depressed fracture with inward should not be performed until the status of the cervical displacement of bone greater than the width of the skull spine is known. Oculovestibular reflexes can yield similar table will usually be taken to the operating room for information but the time required to perform this test limits elevation, debridement and dural repair. The management its usefulness in the acute setting. Corneal responses and of intraparenchymal haematomas is a subject of debate in gag/cough reflexes provide also information about the the neurosurgical literature and many factors are involved in integrity of the brainstem. Absence of brainstem reflexes the decisions regarding evacuation). and abnormal posture movements indicate a more severe brain injury with a worsened prognosis. Neurological intensive care management The most reliable measure of the patient’s condition and A general head examination should be performed along response to therapy is their neurological examination. Serial with neurological examination. Lacerations should be GCS scores and brainstem responses should be measured at palpated for underlying skull fractures that may be frequent intervals during the first 48 hours or until the ICP indicative of underlying haematomas. Evidence of skull stabilises. If a patient worsens clinically, action should be base fractures; e.g., Battle’s and Raccoon’s eyes signs and taken to find the cause and treat it appropriately. Apart from haemotympanum should increase the clinician’s suspicion cerebral causes such as blossoming of contusions or of skull fractures with epidural/acute subdural haematomas cerebral oedema, hypoxia, hypotension and disturbances of and for possible non-recognisable otorrhea or rhinorrhea. electrolytes and glucose can have detrimental effects on These latter may become potential sources of meningitis. If cerebral function. documented, they should be properly treated with slight head elevation and CSF diversionary procedures. Prophy- Intracranial pressure monitoring lactic use of antibiotics has never been proven to be Intracranial pressure, in normal adults, will range from 5 - effective in prevention of meningitis and could be potentially 15 mmHg. Increased ICP adversely affects cerebral perfu- dangerous by giving more virulent bacteria possibility to sion pressure (CPP) and subsequently the delivery of grow.55 energy producing substrates to the brain. There are several mechanisms for monitoring ICP in the intensive care unit Another concern, which may arise in the initial evaluation, (ICU) and each has its own risks versus benefits. The gold is the use of anticonvulsants. A seizure in a patient with standard of ICP monitoring is the catheter.

VOLUME 14, NO. 2, OCTOBER 2010 23 CURRENT MANAGEMENT OF SEVERE TRAUMATIC BRAIN INJURY • Bangash & Baeesa

The catheter is placed through a burr or twist drill hole into A number of other modalities exist which tailored to the lateral ventricle and the proximal end is tunnelled individual patients provide more insight into the physiology beneath the skin before connecting it to a closed drainage of the injured brain. Jugular vein oxygen samples are ob- system. This technique offers the benefit of direct ICP tained by inserting a catheter via the internal jugular vein in monitoring via a fluid-coupled pressure transducer and a retrograde fashion to the jugular bulb.59 Samples of allows CSF drainage from lowering of ICP when necessary. cerebral venous blood allow for assessment of the arterial- Obvious risks include the danger of blindly passing a venous oxygen difference (A-VO2) and lactic acid pro- catheter through the brain with the possibility of creating a duction across the brain. Global cerebral hyperaemia is haematoma or CSF infection. Sometimes ventricular indicated when jugular venous oxygen saturation (SjvO2) catheters are difficult to place if the ventricles are small or exceeds 75% increased oxygen extraction as SjvO2 falls shifted from midline. Other systems include the fibreoptic below 55%, and cerebral ischemia when SjvO2 is less than intraparenchymal monitoring system and the subarachnoid 50%.43 Threshold values for hyperaemia and ischemia bolt, which is a fluid-coupled system. Both are less correspond, respectively, to an A-VO2 less than 4 or more invasive, simpler to place and have a lower incidence of than 9 ml O2/dL.43 Increased lactic acid production is infection. Infections, though rare, usually occur at the associated with cerebral ischemia or infarction.52 Informa- placement site. The fibreoptic system is more fragile and tion obtained from jugular venous oxygen sampling may be can be damaged easily while the subarachnoid bolt can beneficial in defining therapeutic approaches to intracranial become obstructed by herniated brain or debris and has a hypertension. However, the content of each jugular bulb tendency to become less accurate as the ICP increases.30 may differ as they receive different amounts of cerebral The use of a particular system is primarily determined by blood outflow. A recent study demonstrated discrepancies the indications for its use; i.e., monitoring versus treating between samples obtained from a particular jugular bulb ICP and the neurosurgeon’s familiarity. There are 2 major and clearly more studies are indicated to determine how this possible complications of ICP monitoring; haemorrhage (1 intervention will impact upon overall cerebral protection.31 - 2%) and ventriculitis (1 - 10%). Transcranial Doppler ultrasonography (TCD) permits non- Patients with severe head injury will in 50% develop a invasive measurements of blood flow velocity in the basal significant increase in ICP during the first post-traumatic 72 cerebral arteries. While originally used to evaluate vaso- hours.14 Findings on CT imaging may also be predictive of spasm in patients with subarachnoid haemorrhage, it is now problems of increased ICP. These CT findings are: a mass being utilised more and more among patients with severe lesion, midline shift, dilatation of the contralateral ventricle, head injuries. An increase of TCD pulsatility index has obliteration of the third ventricle or effacement of the been shown to identify states of decreased CPP.12 As TCD mesencephalic cisterns.48 Even patients with GCS scores of equipment improves this diagnostic adjunct may play a 8 or less and initial CT scans without these abnormalities greater role in the optimal management of ICP in the future. have at least a 10 - 15% risk of developing increased ICP.18 Other modalities are available for measuring CBF Once the ICP is obtained, measures should be taken to including: xenon-enhanced CT technique, thermal diffusion reduce it to 20 mmHg or less.54 Transient elevations of ICP method and laser Doppler method. above this value for less than 5 minutes are generally acceptable. The CPP should be maintained above 70 Electroencephalography (EEG) and use of evoked mmHg to sustain cerebral metabolism and prevent potentials are techniques infrequently used in intensive care ischemia.12 Cerebral perfusion pressure below this value settings. The complexity of performing and interpreting may be detrimental in patients with dysautoregulation these procedures limit their use to specific cases. Auto- secondary to severe head injury. Conversely, if the CPP is mated trend analysis techniques may provide early warning artificially elevated to too high values, ICP can be worsened signs of neurologic change at the physiologic level.57 due to increasing cerebral oedema in areas where vaso- Electroencephalography monitoring is most useful for paralysis is present. The CPP can be artificially elevated detecting subclinical seizure activity and may provide clues without deleterious ICP effects with judicious use of regarding changes in neurologic function and prognosis. pressors and careful adjustment of fluid therapy and Evoked potentials may be of benefit in identifying par- transfusions to maintain adequate vascular expansion ticular areas of neurologic dysfunction. As technology without excessive increases in total body water.53 becomes more advanced, these procedures may play more important roles in the ICU. Adjunctive monitoring Standard intensive care monitoring techniques; e.g., arterial Treatment of elevated ICP access lines, central venous and pulmonary artery catheters A. General measures are mandatory in the ICU. Various medical interventions exist for ICP control besides

24 PAN ARAB JOURNAL OF NEUROSURGERY CURRENT MANAGEMENT OF SEVERE TRAUMATIC BRAIN INJURY • Bangash & Baeesa withdrawal of CSF. this effect may be overcome by the addition of THAM.46 It is also concluded, in this study, that prophylactic hyper- A few simple manoeuvres can be performed to help lower ventilation is deleterious in head injured patients with GCS ICP. Positioning the patient with the head in a neutral motor scores of 4 - 5. position elevated to 30° will facilitate venous drainage via the jugular veins to decrease intracranial volume. Condi- C. Osmotherapy tions known to affect and increase ICP include agitation, Cerebral dehydration as a means for decreasing brain pain and Valsalva effects incurred while coughing. Efforts oedema and controlling high ICP is a traditional and impor- to reduce ICP elevation in response to these systemic tant part of the neurosurgical armamentarium. Intracranial stimulations include the use of sedation and pharmacologic pressure monitoring is essential during use of osmotherapy paralysis with the significant drawback of removing the as a measure of its effectiveness. Lack of monitoring would ability to examine the patient clinically. Outcome studies be akin to treating hypertension without measuring blood have also shown that routine paralysis is associated with pressure. Various agents such as: diuretics, hypertonic increased risk of pneumonia and longer ICU stays without feedings and fluid restriction have been used to increase necessarily reducing overall ICP.56 serum osmolarity in order to create an osmotic gradient for extraction of water from the brain. The most frequently Narcotics such as morphine (2 - 4 mg/hr) and benzo- utilised drug is mannitol. Mannitol is a diuretic, which diazepines such as midazolam (1 - 2 mg/hr) have been used draws fluid into the vasculature from subsequent clearance as they are short acting and can be reversed with antagonist by the kidney resulting in contraction of the vascular drugs to allow neurologic assessment. Narcotics do not volume and dehydration. It has been shown to decrease alter cerebrovascular resistence and will not raise ICP as blood viscosity and increase CBF without raising ICP in long as pO2 and pCO2 are not altered.57 Benzodiazepines some patients.11,28,45 Reduction of ICP usually occurs in 10 may reduce CBF and CMR to a modest degree, theoreti- - 20 minutes and the duration of effect may last for 2 - 6 cally lowering ICP. A newer agent being used more hours.40 For this reason, mannitol appears to be most frequently in the ICU setting is propofol. Propofol is a effective when given in 3 hour intervals for uncontrolled sedative-hypnotic, which also lowers CBF and CMR and intracranial hypertension. reduces ICP in patients with head trauma.13 It has the added benefit of a shortened half-life, which allows rapid reversal Urea has also been shown effective in reducing ICP of sedation one the intravenous infusion has been stopped, although it is not as widely used.24,32 Whenever osmo- thus allowing easier serial neurological examinations. One therapy is used for ICP control, careful attention towards drawback of propofol is that it can cause systemic hypo- fluid balance and electrolytes should be maintained. tension and fatty liver infiltration if used for a long period.63 Central venous pressure and/or pulmonary capillary wedge If patient agitation could not be controlled with sedation, pressures should be monitored when the volume status is in then pharmacologic paralysis can be utilised.26 Non- question. Maintenance fluids are reduced to one half daily depolarising muscle relaxants, such as vecuronium and requirements and free water is restricted in maintenance pancuronium, are given via continuous infusion to a level fluids by administering isotonic electrolyte solutions.9 sufficient to prevent coughing and patient movement. Dextrose in water (5%) should be avoided as this will exacerbate cerebral oedema and raise ICP. Withdrawal of B. Hyperventilation osmotherapy should proceed in a steady regressive pattern. It is generally agreed that prophylactic hyperventilation Once ICP is controlled, the dosages of osmotic and loop therapy should be avoided during the first 5 days after diuretics can be weaned slowly to allow the serum sodium severe TBI and particularly during the first 24 hrs.60 Hyper- and osmolarity to normalise. ventilation causes cerebral vasoconstriction and reversal of brain and CSF acidosis and only a short-lived effect on CSF D. Corticosteroids pH with a loss of HCO2- buffer from CSF. Possible disad- The use of glucocorticosteroids has been studied exten- vantages include severe cerebral vasoconstriction causing sively in the past in head injured patients.10,15 A recent cerebral ischemia. Hyperventilation and use of tro- randomised controlled trial of the effect of corticosteroids methamine (THAM) - a safe and low toxicity agent - which on death and disability after head injury revealed that the ameliorates the deleterious effect of prolonged hyper- risk of death was higher in the corticosteroid group than in ventilation in major part by causing CSF alkalosis.35 The the placebo.17 Corticosteroids should not be used routinely effect of THAM plus hyperventilation was studied in a in the treatment of head injury. randomised controlled trial and the authors found that a favourable outcome at 3 and 6 months was significantly E. Barbiturates less in those treated with hyperventilation alone although When ICP control is refractory to standard medical and

VOLUME 14, NO. 2, OCTOBER 2010 25 CURRENT MANAGEMENT OF SEVERE TRAUMATIC BRAIN INJURY • Bangash & Baeesa surgical treatments, barbiturate therapy may constitute an References acceptable alternative approach.49 The use of a barbiturate 1. Aarabi B, Hesdorffer DC, Simard JM, Ahn ES, Aresco C, coma has been debated in terms of its efficacy relative to Eisenberg HM, McCunn M, Scalea T: Comparative study of decompressive craniectomy after mass lesion evacuation in the risks involved and the time/skill required employing the severe head injury. Neurosurg 2009, 64(5): 927-39; Discussion technique successfully. High dose barbiturate therapy has 939-40 been shown to reduce the CMR and CBF by as much as 2. Albanèse J, Leone M, Alliez JR, et al: Decompressive craniec- 50%.16,19 Rapid weaning may cause withdrawal seizures or tomy for severe traumatic brain injury: Evaluation of the effects at one year. Crit Care Med 2003, 31(10): 2535-2538 rebound intracranial hypertension. The possible side effects 3. Alderson P, Gadkary C, Signorini DF: Therapeutic hypothermia include hypotension, respiratory complications, myocardial for head injury. Cochrane Database of Syst Rev 2004, 18(4): depression, increased infection rate from leukocytes depres- CD001048 sion, and electrolyte and liver function disturbances.56 4. Al-Ghamdi AS: Emergency medical service rescue times in Riyadh. Accid Anal Prev 2002, 34(4): 499-505 5. Al-Harthi AS, Al-Harbi M: Accidental injuries during muslim F. Hypothermia pilgrimage. Saudi Med J 2001, 22(6): 523-5 Recent studies have examined the use of mild and moderate 6. Ali Aba Hussein N, El-Zobeir AK: Road traffic knowledge and hypothermia as potential adjunct treatments of intracranial behaviour of drivers in the Eastern Province of Saudi Arabia. East Mediterr Health J 2007, 13(2): 364-75 hypertension. It is known by clinical observations and 7. Al-Moutaery K, Akhdar F: Implications of road accidents in experimental data that conventional hypothermia (< 30°C) Saudi Arabia. Pan Arab J Neurosurg 1998, 2(2): 7-8 reduces CMR, CBF and ICP.33,58 However, deep hypo- 8. Ampel L, Holt KA, Siclaff GW, Sloan TB: An approach to thermia may also invoke cardiovascular instability and airway management in the acutely head-injured patient. J Emerg Med 1988, 6(1): 1-7 coagulation abnormalities. Recent meta-analysis review 9. Borel C, Hanley D, Diringer MN, Rogers MC: Intensive man- showed that there is no evidence that hypothermia is agement of severe head injury. Chest 1990, 98(1): 180-189 beneficial in treatment of head injury.3 Moreover, hypo- 10. Braakman R, Schouten HJ, Blaauw-van Dishoeck M, thermia increases the risk of pneumonia and has other Minderhoud JM: Megadose steroids in severe head injury. Results of a prospective double-blind clinical trial. J Neurosurg potentially harmful side effects. 1983, 58(3): 326-330 11. Burke AM, Quest DO, Chien S, Cerri C: The effects of mannitol G. Decompressive craniectomy (DC) on blood viscosity. J Neurosurg 1981, 55(4): 550-553 There is a general agreement that aggressive management 12. Chan KH, Miller JD, Dearden NM, Andrews PJ, Midgley S: The effect of changes in cerebral perfusion pressure upon and monitoring of the patient with closed head injury with middle cerebral artery blood flow velocity and jugular bulb control of ICP will improve patient survival and eventual venous oxygen saturation after severe head injury. J Neurosurg outcome. However, there is little agreement on the value of 1992, 77(1): 55-61 DC for increasing intracranial volume and subsequently 13. Cornfield DN, Tegtmeyer K, Nelson MD, Milla CE, Sweeney M: Continuous propofol infusion in 142 critically ill children. decreasing ICP in these same patients although subtemporal Pediatrics 2002, 110(6): 1177-1181 decompression can be beneficial in patients with medically 14. Dearden NM: Management of raised intracranial pressure after intractable intracranial hypertension.22,24,39,50 Among adults, severe head injury. Br J Hosp Med 1986, 36(2): 94-103 there is no evidence to support the routine use of DC to 15. Dearden NM, Gibson JS, McDowall DG, Gibson RM, Cameron improve mortality and quality of life in brain injured adults MM: Effect of high-dose dexamethasone on outcome from 1,2,66 severe head injury. J Neurosurg 1986, 64(1): 81-88 with high ICP. 16. Donnegan JH, Traystman RJ, Koehler RC, Jones MD Jr, Rogers MC: Cerebrovascular hypoxic and autoregulatory Summary response during reduced brain metabolism. Am J Physiol 1985, 249: 421-429 Among patients with severe head injuries, the major deter- 17. Edwards P, Arango M, Balica L, Cottingham R, El-Sayed H, minant of morbidity and mortality is the severity of the Farrell B, Fernandes J, et al: Final results of MRC CRASH, a initial cerebral insult. Outcome from the initial injury is randomised placebo-controlled trial of intravenous cortico- difficult to ascertain at first inspection as confounding steroid in adults with head injury-outcomes at 6 months. Lancet 2005, 365(9475): 1957-9 issues; e.g., hypotension, drug intoxication, acidosis, may 18. Eisenberg HM, Gary HE Jr, Aldrich EF, et al: Initial CT findings be present. Brainstem indices are good prognosticators in 753 patients with severe head injury. A report from the NIH with the most important finding on examination being the Traumatic Coma Data Bank. J Neurosurg 1990, 73(5): 688-698 pupillary light response. In Arab countries, a great 19. Eisenberg HM, Frankowski RF, Contant CF, Marshall LF, Walker MD: High-dose barbiturate control of elevated intra- emphasis should be directed to pre-hospital care and cranial pressure in patients with severe head injury. J transportation. Severe closed head injury patients should be Neurosurg 1988, 69(1): 15-23 transferred and aggressively managed in specialised trauma 20. Frankowski RF, Annegers JF, Whitman S: Epidemiology and centres. The development and implementation of national, descriptive studies: Part I. The descriptive epidemiology of head trauma in the United States. In: Becker DP, Povlishock preferably Pan Arab, standards and guidelines for manage- JT (eds), Central Nervous System Trauma Status Report. ment of severely head injured patients may improve the Richmond, William Byrd Press 1985, pp 33-43 outcome and prognosis. 21. Gennarelli TA, Champion HR, Sacco WJ, et al: Mortality of

26 PAN ARAB JOURNAL OF NEUROSURGERY CURRENT MANAGEMENT OF SEVERE TRAUMATIC BRAIN INJURY • Bangash & Baeesa

patients with head injury and extracranial injury treated in trauma Am 1994, 5(4): 661-670 centers. J Trauma 1989, 29: 1193-1201; Discussion 1201-1202 44. Miller JD, Sweet RC, Narayan R, Becker DP: Early insults to 22. Gower DJ, Lee KS, McWhorter JM: Role of subtemporal the injured brain. JAMA 1978, 240(5): 439-442 decompression in severe closed head injury. Neurosurg 1988, 45. Muizelaar JP, Lutz HA 3rd, Becker DP: Effect of mannitol on ICP 23(4): 417-422 and CBF and correlation with pressure autoregulation in 23. Grindlinger GA, Niehoff J, Hughes SL, Humphrey MA, Simpson severely head-injured patients. J Neurosurg 1984, 61(4): 700-706 G: Acute paranasal sinusitis related to nasotracheal intubation 46. Muizelaar JP, Marmarou A, Ward JD, et al: Adverse effects of of head-injured patients. Crit Care Med 1987, 15(3): 214-217 prolonged hyperventilation in patients with severe head injury: 24. Guerra WK, Gaab MR, Dietz H, et al: Surgical decompression a randomized clinical trial. J Neurosurg 1991, 75(5): 731-739 for traumatic brain swelling: indications and results. J Neurosurg 47. Ng I, Lew TW, Yeo TT, Seow WT, Tan KK, Ong PL, San WM: 1999, 90(2): 187-196 Outcome of patients with traumatic brain injury managed on a 25. Gunnar W, Jonasson O, Merlotti G, Stone J, Barrett J: Head standardised head injury protocol. Ann Acad Med Singapore Injury and hemorrhagic stock: studies of the blood brain barrier 1998, 27(3): 332-9 and intracranial pressure after resuscitation with normal saline 48. O’Sullivan MG, Statham PF, Jones PA, et al: Role of intra- solution, 3% saline solution, and dextran-40. Surg 1988, 103 cranial pressure monitoring in severely head-injured patients (4): 398-407 without signs of intracranial hypertension on initial computer- 26. Hsiang J, Chesnut RM, Crips CB, et al: Early, routine paralysis ized tomography. J Neurosurg 1994, 80(1): 46-50 for ICP control in severe head injury: Is it necessary? Crit Care 49. Piek J: Barbiturate coma in patients with severe head injuries: Med 1994, 22: 1471-1476 long-term outcome in 79 patients. Adv Neurosurg 1993, 21: 27. Jackson R, Butler J: Best evidence topic reports. Hypertonic or 178-183 isotonic saline in hypotensive patients with severe head injury. 50. Polin R, Shaffrey M, Bogaev C, et al: Decompressive bifrontal Emerg Med J 2004, 21(1): 80-1 craniectomy in the treatment of severe refractory posttraumatic 28. Jafar JJ, Johns LM, Mullan SF: The effect of mannitol on cerebral edema. Neurosurg 1997, 41(1): 84-92; Discussion cerebral blood flow. J Neurosurg 1986, 64(5): 754-759 92-94 29. Koenig KL: Rapid-sequence intubation of head trauma patients: 51. Ratrout NT: Tire condition and drivers' practice in maintaining prevention of fasciculations with pancuronium versus minidose tires in Saudi Arabia. Accid Anal Prev 2005, 37(1): 201-6 succinylcholine. Ann Emer Med 1992, 21(8): 929-932 52. Robertson CS, Grossman RG, Goodman JC, Narayan RK: 30. Lang EW, Chesnut RM: Intracranial pressure. Monitoring and The predictive value of cerebral anaerobic metabolism with management. Neurosurg Clin N Am 1994, 5(4): 573-605 cerebral infarction after head injury. J Neurosurg 1987, 67(3): 31. Latronico N, Beindorf AE, Rasulo FA, et al: Limits of intermit- 361-368 tent jugular bulb oxygen saturation monitoring in the manage- 53. Rosner MJ, Daughton S: Cerebral perfusion pressure manage- ment of severe head trauma patients. Neurosurg 2000, 46(5): ment in head injury. J Trauma 1990, 30(8): 933-940; Discussion 1131-8; Discussion 1138-9 940-1 32. Levin AB, Duff TA, Javid MJ: Treatment of increased intra- 54. Saul TG, Ducker TB: Effect of intracranial pressure monitoring cranial pressure: a comparison of different hyperosmotic and aggressive treatment in mortality in severe head injury. J agents and the use of thiopental. Neurosurg 1979, 5(5): 570-5 Neurosurg 1982, 56(4): 498-503 33. Marion DW, Obrist WD, Carlier PM, et al: The use of moderate 55. Schaffer L, Kranzler LI, Siqueira EB: Aspects of evaluation and therapeutic hypothermia for patients with severe head injuries: treatment of head injury. Neurol Clin 1995, 3(2): 259-273 a preliminary report. J Neurosurg 1993, 79(3): 354-362 56. Schalén W, Messeter K, Nordström CH: Complications and 34. Marmarou A, Anderson RL, Ward JD, et al: Impact of ICP side effects during thiopentone therapy in patients with severe instability and hypotension on outcome in patients with severe head injuries. Acta Anaesthesiol Scand 1992, 36(4): 369-377 head trauma. J Neurosurg 1991, 75(Suppl 1): S59-S66 57. Shapiro HM: Anesthesia effects upon cerebral blood flow, 35. Marmarou A, Holdaway R, Ward JD, Yoshida K, Choi SC, cerebral metabolism, electroencephalogram, and evoked Muizelaar JP, Young HF: Traumatic brain tissue acidosis: potentials. In: Miller RD (ed), Anesthesia. New York, Churchill Experimental and clinical studies. Acta Neurochir Suppl (Wien) Livingstone 1986, pp 1249-1288 1993, 57: 160-4 58. Shiozaki T, Sugimoto H, Taneda M, et al: Effect of mild 36. Marshal LF, Gautille T, Klauber MR: The outcome of severe hypothermia on uncontrollable intracranial hypertension after closed head injury. J Neurosurg 1991 75(Suppl 1): S28-S36 severe head injury. J Neurosurg 1993, 79(3): 363-368 37. McGraw CP, Howard G: Effect of mannitol on increased 59. Stocchetti N, Paparella A, Bridelli F, et al: Cerebral venous intracranial pressure. Neurosurg 1983, 13(3): 269-271 oxygen saturation studied with bilateral samples in the internal 38. McHugh GS, Engel DC, Butcher I, Steyerberg EW, Lu J, jugular vein. Neurosurg 1994, 34(1): 38-43; Discussion 43-4 Mushkudiani N, Hernández AV, Marmarou A, Maas AI, Murray 60. The Brain Trauma Foundation, The American Association of GD: Prognostic value of secondary insults in traumatic brain Neurological Surgeons, The Joint Section on Neurotrauma injury: results from the IMPACT study. J Neurotrauma 2007, and Critical Care: Hyperventilation. J Neurotrauma 2000, 17 24(2): 287-93 (6-7): 513-20 39. Meier U, Gräwe A: The importance of decompressive craniec- 61. Temkin NR, Dikmen SS, Wilensky AJ, et al: A randomized, tomy for the management of severe head injuries. Acta double-blind study of phenytoin for the prevention of post- Neurochir Suppl 2003, 86: 367-71 traumatic seizures. N Engl J Med 1990, 323(8): 497-502 40. Mendelow AD, Teasdale GM, Russell T, et al: Effect of mannitol 62. Tisdall MM, Smith M: Multimodal monitoring in traumatic brain on cerebral blood flow and cerebral perfusion pressure in injury: current status and future directions. Br J Anaesth 2007, human head injury. J Neurosurg 1985, 63(1): 43-48 99(1): 61-7 41. Miller JD: Minor, moderate and severe head injury. Neurosurg 63. Vasile B, Rasulo F, Candiani A, Latronico N: The patho- Rev 1986, 9: 135-9 physiology of propofol infusion syndrome: a simple name for a 42. Miller JD, Jones PA, Dearden NM, Tocher JL: Progress in the complex syndrome. Intensive Care Med 2003, 29(9): 1417-25 management of head injury. Br J Surg 1992, 79(1): 60-64 64. White PF, Scholobohm RM, Pitts LH, Lindauer JM: A 43. Miller JD, Piper IR, Jones PA: Integrated multimodality monitor- randomized study of drugs for preventing increases in intra- ing in the neurosurgical intensive care unit. Neurosurg Clin N cranial pressure during endotracheal suctioning. Anesthesiol

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1982, 57(3): 242-244 66. Winter CD, Adamides A, Rosenfeld JV: The role of decom- 65. White RJ, Likavec MJ: The diagnosis and initial management pressive craniectomy in the management of traumatic brain of head injury. N Engl J Med 1992, 327(21): 1507-1511 injury: a critical review. J Clin Neurosci 2005, 12(6): 619-623

28 PAN ARAB JOURNAL OF NEUROSURGERY Review Article

Outcome predicting factors in intracranial aneurysms: Defining the complex aneurysms

Raj Kumar, Vivek Kumar Vaid, Samir Kumar Kalra, Sanjay Behari, Ashok Kumar Mahapatra

Abstract Objectives: The development of neurosciences has made the understanding and management of intracranial aneurysms better. It has been realized over the years that there may be a subgroup of patients who fare differently from the rest. The identification of factors which make the aneurysm different or complex may help in prognosticating patients. The aim of the current study was to identify such factors to assess how well they correlated with the outcome.

Methods: Three-hundred-fourteen consecutive cases of spontaneous subarachnoid haemorrhage with intracranial aneurysms were retrospectively analyzed. Sixteen independent factors broadly categorized into three categories viz., patient related, radiological factors and surgery related factors were analyzed and their correlation with outcome studied. Univariate and multivariate analysis was done using logistic regression analysis and P values, and predictive values were determined.

Results: Five factors viz., WFNS grade 4 - 5, clinical vasospasm, smoking ≥ 30 years, Fisher grade 3 - 4 and poste- rior circulation aneurysms were found to have highly significant association with poor outcome both using univariate and multivariate analysis.

Conclusions: The clinical factors predominate and have more significant association with the outcome. The presence of factors viz., WFNS grade 4 - 5, clinical vasospasm (DIND), smoking ≥ 30 years, Fisher grade 3 - 4 and posterior circulation aneurysms were found to correlate with poor outcome and any of these factors could lead to poor outcome and are sufficient to label the patient as having a complex aneurysm. (p29-37)

Key words: Complex aneurysms, Fisher grade, Glasgow Outcome Scale, intracranial aneurysms and subarachnoid haemorrhage.

Introduction The understanding and management of intracranial aneu- aim of the current study was to assess the effect of various rysms has progressively become better with the advent of factors; from the patient related parameters, clinico- recent neurosurgical techniques. The outcome of such pa- radiological profile and the surgical management with the tients varies differently and is dependent upon various outcome. The identification of those factors which corre- factors. The identification of such factors may help in better lated significantly with poor outcome was also performed. prognostication of the patients prior to surgery. Material and methods To predict the response following surgical treatment, con- Patient spectrum sideration of a multitude of factors may be needed. The Out of total 687 consecutive patients with spontaneous subarachnoid haemorrhage (SAH), and having intracranial aneurysms, 314 cases (mean age 51.65 years, range 65 ± Department of Neurosurgery Sanjay Gandhi Postgraduate Institute of Medical Sciences 14.225 [15 - 80 years], with female: male ratio of 58:42 Lucknow - UP [1.380]), who met the inclusion criteria were included. All India were examined, managed, followed-up and records were Correspondence: maintained in the departmental computer database during Prof. Raj Kumar Department of Neurosurgery the period 2001 - 2005 at Sanjay Gandhi Postgraduate Sanjay Gandhi Postgraduate Institute of Medical Sciences Institute of Medical Sciences, Lucknow, India were in- Lucknow - 226014, UP cluded and analyzed retrospectively. India Tel: (91 522) 266 8700 / 266 8800 Fax: (91 522) 266 8129 / 2668017 The inclusion criteria were: Email: [email protected] / [email protected] 1. Open surgery performed

VOLUME 14, NO. 2, OCTOBER 2010 29 COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al

2. A follow-up of at least 2 years Table 2 - Management protocol.

3. Spontaneous SAH with angiographically demonstrable SAH aneurysms Urgent DSA 4. Surgery within 48 hours of admission to our hospital and DSA negative DSA positive 5. Patients who underwent check digital subtraction angio- graphy (DSA) at 6 weeks following surgery. Conservative management Posted for surgery in next available and repeat DSA at 6 weeks OR with informed consent The exclusion criteria were: 1. Unruptured aneurysms 2. Traumatic aneurysms 3. Mycotic aneurysms and Intensive perioperative care; Not willing/unfit for 4. Patients managed with endovascular techniques. vasospasm, hydrocephalus surgery, managed managed conservatively The clinical profile of the included patients is presented as Table 1.

Discharge and check DSA Table 1 - Clinical spectrum. at 6 weeks No. Symptoms/ signs Number (n) 0 - 19 8 20 - 39 54 done using standard microsurgical techniques. Additional 1 Age group 40 - 59 142 wrapping (n = 6) of the fundus was also done in selective > 60 110 cases whenever required. Postoperatively, all patients were Male 132 2 Sex managed intensively and monitored closely and were Female 182 discharged on seventh postoperative day following suture No 28 3 Headache Yes 286 removal unless some co-morbidity forced us to delay their No 44 discharge. 4 Vomiting Yes 270 No 224 Follow-up evaluation 5 Seizures Yes 90 All cases were followed-up postoperatively at 6 weeks, 3 No 206 6 Loss of consciousness months, 12 months and then at yearly intervals thereafter, Yes 108 all follow-up records were maintained. A check DSA was No 228 7 Motor weakness done at 6 weeks to look for any residual aneurysms and Yes 86 position of clips. The mean follow-up was 33.38 ± 6.305 No 288 8 Sensory deficit Yes 26 months (range 24 - 59 months).

No 258 9 Cranial nerve involvement Yes 56 Analysis of factors and classification of results No 236 Sixteen independent factors were considered after extensive 10 Meningeal signs Yes 78 literature review and their presence in each patient was noted. They were categorized as patient related, radio- logical related and related to surgery. They were as follows: Management protocol All patients who underwent DSA and had intracranial Patient related factors: aneurysm were given option of endovascular or surgical 1. Age treatment. The risks, advantages, disadvantages and 2. World Federation of Neurological Surgeons (WFNS) complications of both procedures were explained in detail. grade at admission Those patients who opted for surgery were operated upon at 3. Clinical vasospasm or delayed ischemic neurological the earliest. The management scheme that was followed is deficit (DIND) mentioned in Table 2. 4. Ictus to surgery interval 5. Smoking Depending upon the location, multiplicity and the pattern of 6. Hypertension aneurysmal filling suitable craniotomy (n = 292)/ craniec- tomy (n = 22) was done. The aneurysms were dissected Radiological factors: out, the necks were defined and aneurysmal clippings were 1. Angiographic vasospasm

30 PAN ARAB JOURNAL OF NEUROSURGERY COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al

2. Giant aneurysm Smoking was another patient related factor to be considered 3. Multiple aneurysms and patients were divided into two groups; one with 4. Fisher grade on computed tomogram (CT) image smoking history of more than 30 years and the other 5. Presence of infarct including either non-smokers or those who had less than 30 6. Presence of hydrocephalus year duration of smoking.

Surgical factors: Hypertension was the last patient related factor considered 1. Posterior circulation aneurysms and patients were categorized into two groups; one with 2. Distal anterior cerebral artery (DACA) aneurysms history of hypertension less than 20 years and others who 3. Paraclinoid aneurysms had history of hypertension for more than 20 years duration. 4. Previous intracranial surgery Radiological factors: The factors were not mutually exclusive and there was The second category was based on the radiological parameters overlap of factors in some patients. and the first factor to be considered was the angiographic vasospasm which was characterized by arterial narrowing The statistical analysis was performed using the SPSS for with slowing of contrast filling. Here the patients were Windows software (version 13.0; SPSS, Inc., Chicago, IL). grouped into two groups, one where patient had angio- Univariate and multivariate analysis was done using logistic graphic vasospasm and another where the patients did not regression analysis and P values, and predictive values were have any such angiographic evidence. determined. Each factor was analyzed using both uni- variate and multivariate analysis and the factors which Another factor considered was presence of multiple aneu- showed statistical significance using both were identified. rysms and again patients were categorized into those who had 2 or more intracranial aneurysms and those who had Patient related factors: only a single aneurysm. Among the patient related factors, age was the first factor to be considered and patients were divided into two groups, Giant aneurysms were also included amongst the radio- less than 70 years and more than 70 years logical factors and patients were accordingly divided into those having giant aneurysms (size ≥ 25mm) and their non The next factor to be considered was WFNS grade and giant counterparts. patients were divided into two groups accordingly; grade 1 - 3 group and grade 4 - 5 group. The WFNS grading is The Fisher grading correlating with the amount of blood on mentioned in Table 3. CT image was another radiological factor to be considered and patients were then segregated into those with a grade of 1 - 2 and another group with a grade of 3 - 4. The Fisher Table 3 - World Federation of Neurological Surgeons grading. grading is mentioned in Table 4. WFNS grade GCS score Major focal deficit* 0+ 1 15 - Table 4 - Fisher grading system. 2 13 - 14 - Fisher group Blood on CT 3 13 - 14 + 1 No subarachnoid blood detected 4 7 - 12 + or - 2 Diffuse or vertical layers < 1 mm thick 5 3 - 6 + or - 3 Localized clot and/or vertical layer ≥ 1 mm Abbreviations: +intact aneurysm, *aphasia, hemiparesis or hemiplegia 4 Intracerebral or intraventricular clot with diffuse or no SAH

Clinical vasospasm or DIND was the next factor which was included and patients were divided into two groups The presence of radiological infarct and hydrocephalus according to its presence or absence. were also included and patients were divided accordingly into two categories based on their presence or absence. The interval between ictus to surgery was also considered as a patient related factor and patients were divided into two Surgical factors: groups accordingly; those undergoing surgery within 7 days Surgical factors were the next group to be included and the and those where surgery was performed after 7 days of presence of posterior circulation aneurysm was the first ictus. factor to be considered and patients were then accordingly

VOLUME 14, NO. 2, OCTOBER 2010 31 COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al grouped into those having them and those having in the Tables 6 and Table 7 respectively. more common anterior circulation.

Presence of DACA was also considered as a factor and Table 6a - Patient related factors (univariate analysis). Patient related Outcome P Exp 95% CI for patients were grouped into those having them and those not. No. factors Good Poor value (β) Exp (β) Paraclinoid aneurysms which involved drilling and mobili- 1 Age ≤ 70 years 260 194 66 .083 .578 .311 - 1.074 zation of optic nerve to define the neck and requiring a high > 70 years 54 34 20 2 WFNS grade 1 - 3 236 188 48 .000 .269 .156 - .464 level of expertise to deal with were also considered and 4 - 5 78 40 38 patients were grouped into two groups; one harbouring 3 Clinical No 204 166 38 .000 .296 .176 - .495 them and another not. vasospasm Yes 110 62 48 4 Ictus surgery ≤ 7 days 272 206 66 .002 .352 .181 - .686 The last factor to be considered was the presence of previous interval > 7 days 42 22 20 intracranial surgery and patients were categorized into two 5 Smoking No 294 224 70 .000 .078 .025 - .241 > 30 years Yes 20 4 16 groups where one group had underwent intracranial surgery 6 Hypertension No 260 194 66 .083 .578 .311 - 1.074 previously and another where patient were undergoing > 20 years Yes 54 34 20 surgery for the first time. Abbreviations: Exp: exponential, CI: confidence interval

Table 6b - Radiological factors (univariate analysis). The results were classified by the Glasgow Outcome Scale Radiological Outcome P Exp 95% C.I. for (GOS) and were divided into two groups; good outcome No. factors value (β) Exp (β) and poor outcome, where grade 4 and 5 were classified as Good Poor poor outcome and grade 1 - 3 as good outcome. The GOS 1 Angiographic No 258 216 42 .000 .053 .026 - .109 vasospasm Yes 56 12 44 is mentioned in Table 5. For statistical analysis, the result at 2 Giant No 288 24 64 .000 .052 .017 - .156 last follow-up was used and all the factors were analyzed in aneurysms Yes 26 4 22 relation to the outcome at this follow-up period. 3 Multiple No 260 182 78 .026 2.464 1.111 - .5.464 aneurysms Yes 54 46 8 4 Fisher grade 1-2 238 186 52 .000 .345 .200 - .597 Table 5 - Glasgow Outcome Scale. 3-4 76 42 34 Score Meaning 5 Infarct No 258 208 50 .000 .134 .071 - .250 5 Good recovery - resumption of normal life despite minor deficits Yes 56 20 36 (“return to work”) 6 Hydrocephalus No 260 216 44 .000 .058 .028 - .119 Yes 54 12 42 4 Moderate disability (disabled but independent) - travel by public transport, can work in sheltered setting (exceeds mere ability to perform “activities of daily living”) Table 6c - Surgical factors (univariate analysis). Surgical Outcome P Exp 95% C.I. 3 Severe disability (conscious but disabled) - dependent for No. daily support (may be institutionalized - but this is not a criteria) factors Good Poor value (β) for Exp (β) 2 Persistent vegetative state - unresponsive and speechless; 1 Aneurysms in No 276 210 66 .000 .283 .141 - .566 after 2 - 3 weeks, may open eyes and have sleep/wake cycles the posterior Yes 38 18 20 circulation 1 Death - most deaths ascribable to primary head injury occur 2 DACA No 294 212 82 .447 1.547 .502 - within 48 hours aneurysms Yes 20 16 4 4.765 3 Paraclinoid No 284 220 64 .000 .106 .045 - .249 aneurysms Yes 30 8 22 Results 4 Previous intra- No 300 222 78 .016 .264 .089 - .783 Patient’s spectrum: cranial surgery Yes 14 6 8 In our series the most common age group to which the Table 7 - Multivariate analysis. patients belonged was of 40 - 59 years with 45.20% cases Outcome P Exp 95% C.I. for No. Factors (n = 142), with next common age group being 60 and above Good Poor value (β) Exp (β) with 35% cases (n = 110). Fifty-eight-percent (n = 182) 1 WFNS grade 1 - 3 236 188 48 .000 5.444 2.414 - 12.277 patients were females. The most common presentation was 4 - 5 78 40 38 abrupt onset headache and vomiting with 91.10% and 86% 2 Clinical No 204 166 38 .000 4.567 2.219 - 9.402 vasospasm Yes 110 62 48 of patients presenting with these symptoms respectively. 3 Smoking No 294 224 70 .019 11.073 1.482 - 82.719 This was followed (in decreasing frequency) by loss of con- > 30 years Yes 20 4 16 sciousness, seizures, motor weakness, cranial nerve involve- 4 Fisher grade 1 - 2 238 186 52 .002 3.563 1.613 - 7.874 ment and sensory involvement. Meningeal signs were 3 - 4 76 42 34 5 Aneurysms in No 276 210 66 .000 6.131 2.233 - 16.833 elicited in 75.20% of the patients. The analysis of factors the posterior Yes 38 18 20 using univariate and multivariate analysis is presented in circulation

32 PAN ARAB JOURNAL OF NEUROSURGERY COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al

Analysis of factors: was present in 17.83% (n = 56) patients and was associated Patient related: with a poor outcome in 78.57% patients. In comparison, Age: In our series there were 17.19% (n = 54) patients with the patients without angiographic vasospasm had poor age ≥ 70 years and had poor outcome in 37.03% as com- outcome in 16.27% only. This was found to be statistically pared to those < 70 years who had a poor outcome in significant on univariate analysis (P value = 0.000) but not 25.38%. On univariate (P value = 0.083) and multivariate on multivariate analysis (P value = 0.999). analysis (P value = 0.294); this was not however found statis- tically significant. Giant aneurysms: In the series there were 8.28% (n = 26) patients who had a giant aneurysms (size more than 25 WFNS grade 4 - 5: There were 24.84% (n = 78) patients mm) and this group had a poor outcome in 84.61% patients. who had WFNS grade of 4-5 and had a poor outcome in The patients with smaller sized aneurysms had poor out- 48.71% as compared to grade 1-3 who had a poor outcome come in 22.22% in comparison. This was again found to be in 20.33%. This was found to be highly significant on statistically significant on univariate analysis (P value = univariate analysis (P value = 0.000) as well as on multi- 0.000) but not on multivariate analysis (P value = 0.496). variate analysis (P value = 0.000). Multiple aneurysms: There were 17.19% (n = 54) pa- Clinical vasospasm: In our series there were 35.03% (n tients who had multiple aneurysms and had poor outcome = 110) patients who developed clinical vasospasm prior to in 14.81% as compared to those patients who had a single surgery and were associated with a poor outcome in 43.63% aneurysm and had poor outcome in 30%. This was found to as compared to those patients who did not have clinical be statistically significant on univariate analysis (P value = vasospasm who had poor outcome in 18.02%. This also 0.026) but did not have statistical significance on multi- was found to be highly significant on univariate analysis (P variate analysis (P value = 0.096). value = 0.000), as well as on multivariate analysis (P value = 0.000). Fisher grade 3 - 4: The thickness of blood on CT scan significantly influenced outcome and there were 24.20% (n Ictus to surgery interval > 7 days: Patients who =76) patients who had Fisher grade of 3 - 4 and were underwent surgery after a period of more than 7 days after associated with a poor outcome in 44.73% as compared to ictus were 13.37% (n = 42) and were associated with a poor those with Fisher grade 1 - 2 who had poor outcome in outcome in 47.61%, as compared to those patients who 21.84%. This was found to be statistically highly signifi- underwent surgery earlier who had a poor outcome in cant on univariate analysis (P value = 0.000) and also on 24.26%. This was found to be statistically significant on multivariate analysis (P value = 0.002). univariate analysis (P value = 0.002); however on multi- variate analysis this was not found to be statistically Presence of infarct: There were 17.83% (n = 56) patients significant (P value = 0.131). who had radiological infarct diagnosed on imaging before

undergoing surgery and these were associated with a poor Smoking for more than 30 years: There were 20 outcome in 64.28% of the patients as compared to those (6.36%) patients in our series that had history of smoking of who did not have infarct and who had poor outcome in more than 30 years and had poor outcome in 80% as 19.37% only. This was highly significant on univariate compared to non-smokers or those who had smoked for analysis (P value = 0.000) but not on multivariate analysis less than 30 years who had a poor outcome in 23.80%. (P value = 0.077). This was found to be statistically significant not only on univariate analysis (P value = 0.000) but also on multi- Presence of hydrocephalus: In our series there were variate analysis (P value = 0.019). 17.19% (n = 54) patients who had hydrocephalus on imag- Hypertension more than 20 years: In our series there ing prior to surgery and these were associated with a poor were 17.19% (n = 54) patients with history of hypertension outcome in 77.77% as compared to those who did not have for more than 20 years and had a poor outcome in 37.03% hydrocephalus who had poor outcome in 16.92%. This was as compared to those patients who were non-hypertensive statistically significant on univariate analysis (P value = and were hypertensive for a period less than 20 years who 0.000) but did not have any significance on multivariate had a poor outcome in 25.38%. This factor was not found analysis (P value = 1.000). to be statistically significant on both univariate and multi- variate analysis. Surgery related: Aneurysm in the posterior circulation: In our series Radiology related: there were 12.10% (n = 38) patients with posterior circula- Angiographic vasospasm: Angiographic vasospasm tion aneurysms and these had poor outcome in 52.63% in

VOLUME 14, NO. 2, OCTOBER 2010 33 COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al comparison to others who had poor outcome in 23.91%. headings viz., patient related, radiology related and surgery This was found to be statistically highly significant on both related, and analyze their impact on outcome. univariate analysis (P value = 0.000) and multivariate analy- sis (P value = 0.000). Patient spectrum: In our series we found that 40 - 59 years age group was the Aneurysms of the DACA: Twenty patients (6.36%) had most common age group for the patients having intracranial DACA aneurysms and the poor outcome in these was 20% aneurysms in our setup. Majority of patients were females. as compared to the other group who had poor outcome in These are in concordance with the present literature.4,8 The 27.89%. This was found to be statistically insignificant on most common presentation was of headache and vomiting univariate (P value = 0.447) as well as multivariate analysis and meningeal signs were present in a significant number of (P value = 0.797). the patients.

Paraclinoid aneurysms: In our series there were 9.55% Analysis of factors: (n = 30) patients who had paraclinoid aneurysms and these Patient related factors: patients had poor outcome in 73.33% as compared to the The incidence of aneurysmal rupture gradually increases rest of the patients with aneurysms who had poor outcome with each decade of life and peaks in the sixth decade. An in 22.53%. This was statistically significant on univariate aging brain has a less than optimum response to initial analysis (P value = 0.000) but was statistically insignificant bleeding (also observed after traumatic brain injuries) on multivariate analysis. probably due to (i) time-dependent loss of structure and function; (ii) impaired accuracy of translation (codon Previous intracranial surgery: These were 4.45% (n = restriction theory of cellular aging); (iii) reduced number of 14) patients who had undergone previous intracranial sur- mitochondria in the neurons and increased susceptibility to gery. These included surgery for previous intracranial oxidative and excitatory amino acid damage; and (iv) slow aneurysm (n = 8), pituitary macroadenoma (n = 3), supraten- response of ‘reactive synaptogenesis’ or axonal sprouting. torial meningioma (n = 2) and chronic subdural haematoma Irrespective of the grade at surgery, patients above 70 years (n = 1). Patients who had undergone previous intracranial fare poorly.19,27,41,42 In our series we had categorized the surgery had poor outcome in 57.14% as compared to those patients into < 70 years and ≥ 70 years and the patient’s ≥ patients who were undergoing intracranial surgery for the 70 years understandably had poor outcome. This was first time where the incidence of poor outcome of 26%. significant by univariate analysis but however by This was found to be statistically significant on univariate multivariate analysis it was found to have no significant (P value = 0.016) but not on multivariate (P value = 0.059) bearing on the outcome of the included patients. analysis. World Federation of Neurological Surgeons grading was Factors with poor outcome (complex aneurysms): used in our series of patients to grade the patient at After statistical analysis using both univariate and multi- admission. World Federation of Neurological Surgeons variate logistic regression, there were 5 factors which were grade uses the Glasgow Coma Scale (GCS) to evaluate the statistically significant on both analyses; they are: WFNS level of consciousness, and uses the presence or absence of grade 4 - 5 at admission, clinical vasospasm, smoking for major focal neurological deficit to grade the patients. This more than 30 years, Fisher grade 3 - 4 on CT image and grading system is universally acceptable. Patients with aneurysm in the posterior circulation. poor grades at admission are associated with poor outcome and imply already established insult to the brain.9,11,29,35,37 The patients who had any one or more of these factors pre- In our series we also found that WFNS grade is a useful operatively were highly likely to have poor outcome follow- tool to predict poor outcome. World Federation of ing surgery. Based on the presence of these, we chose to Neurological Surgeons was found to have significant define the patients with these factors as those harbouring correlation to outcome by both univariate and multivariate complex aneurysms. analysis and thus a factor which would quantify an aneurysm as complex by its inclusion. Discussion Over the years, extensive research related to the factors Patients with DIND or clinical vasospasm are characterized associated with poor outcome of intracranial aneurysms has by newly developing neurological deficit that is not been done. A collation of all such factors is however scarce explainable by the haematoma, hydrocephalus, seizures or in available literature. After extensive literature review we metabolic derangements. Patients with DIND do poorly considered factors which had significant bearing on the postoperatively. It heralds the onset of vasospasm which outcome. We chose to categorize the factors into three requires not only dedicated intensive monitoring and

34 PAN ARAB JOURNAL OF NEUROSURGERY COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al medication but also prolongs the hospital stay.21,36,44 In our trapping with vascular bypass.5,7,23 In our study we found study we found that development of clinical vasospasm was that giant aneurysms were correlating with poor outcome associated with poor outcome and this factor was signifi- on univariate analysis but not on multivariate analysis. cantly associated with the outcome of both the univariate Patients with multiple aneurysms also pose surgical chal- and multivariate analysis and hence qualified for its inclu- lenge as these patients sometime require multiple surgeries sion in the complex category. and it is difficult to manage vasospasm after first surgery with remaining unclipped aneurysms as hyperdynamic ther- Ictus to surgery interval of more than 7 days is associated apy cannot be instituted.32,38 Though as a single factor with a poor outcome as day 4 - 10 following SAH is the multiple aneurysms were of significant importance to the high risk time period for a patient to develop vasospasm. poor outcome, it was not so on multivariate analysis. Also early surgery with removal of clot and other soluble substances, which predisposed to vasospasm along with The amount of blood in the subarachnoid space as reflected administration of aggressive hyperdynamic therapy once by the Fisher grade has a significant influence on the poor the aneurysm was clipped, helps in managing the outcome. This was found in our study as Fisher grades 3 vasospasm.30,37 In our patients we found that the patients and 4 were found to have a significant correlation with poor being operated late because of either delayed referral or any outcome; using both analytical methods. This may be due other reason i.e., > 7 days had a statistically significant to the fact that a higher amount of blood with release of association with poor outcome on univariate analysis but soluble degradation products increases the chances of clini- this factor did not have significance on multivariate analysis cal vasospasm.1,15,20 and hence was not included in the complex category. The presence of hydrocephalus was also found to be an On reviewing the literature we found that smoking not only important factor having a bearing on the outcome on uni- predisposed the formation of aneurysms but also had an variate but not on multivariate analysis. The reasons mainly impact on the development of vasospasm.4,6,8,16,22 It is a include surgical difficulty, delayed recovery, increased modifiable factor and patients should be educated regarding morbidity and often a second surgery in the form of ventri- the risks of smoking.8 In our study we found that patients culoperitoneal shunt.8,12,14,16,21,43 with history of prolonged smoking > 30 years had a strong correlation with poor outcome and this factor showed The presence of infarct also reflects on the poor outcome as statistical significance on both univariate and multivariate again it is an easily identifiable factor which not only analysis, hence, we included this factor in the complex reflects the insult to the brain but also correlates with patient variety. related factors to some extent.21,44

Hypertension was the factor which we studied and found Surgical factors: that it not only predisposes formation of aneurysms but also Posterior circulation aneurysms as a group in itself were leads to their rupture with significant cardiopulmonary found to have a strong influence on the outcome. It is morbidity.8,16 This factor was found to be significant on because the surgical approach needed to deal with this univariate analysis but did not have significance on multi- aneurysm is specific and associated vasospasm, if present, variate analysis and thus was not considered in the complex is lethal because of close relationship to the brainstem with variety. attendant vital structures.31,39 In our series 12.10% patients had aneurysm in the posterior circulation and these patients Radiological factors: had significant correlation with poor outcome both by Angiographic vasospasm demonstrated by spastic arteries univariate and multivariate analysis and were included as or decreased blood flow and metabolism does not always complex aneurysms. correlate with clinical vasospasm and though these subgroup of patients are kept under strict supervision, they Distal anterior circulation aneurysms are again approached often do not develop new deficits. It is an easily identifiable through special routes and are surgically demanding. and visible factor which alarms the possible DIND.25,26 In 10,28,34,40 These aneurysms were also found to have a strong our series we found that even though angiographic influence on the outcome; however not statistically signifi- vasospasm was having correlation with poor outcome, it cant in multivariate analysis. was not as significant as clinical vasospasm. Paraclinoid aneurysms as a group is found to have direct Patients with giant aneurysms do not fare well as the influence on the outcome as these often involve proximal surgical approach is challenging; the intraoperative course neck control of the internal carotid artery (ICA), temporary demanding and includes rupture, temporary clipping and clipping and drilling of the anterior clinoids so as to

VOLUME 14, NO. 2, OCTOBER 2010 35 COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al delineate the neck of the aneurysm, making their approach circulation aneurysms are significantly associated with poor surgically demanding.13,18,19,33,45 In our study we found that outcome. The clinical factors predominate and have more patient with paraclinoid aneurysms had significant correla- significant association with the outcome. The presence of tion with poor outcome as expected but on multivariate any of these factors hence could make the patient fall in analysis this factor did not have any significance. poor outcome group and are sufficient to label the patient having a complex aneurysm. The patients with previous intracranial surgery were in- cluded as they have specific management issues. It is References because of the adhesions encountered during microsurgical 1. Ahuja A, Guterman LR, Hopkins LN: Carotid cavernous fistula dissection of their aneurysms making their approach and false aneurysm of the cavernous carotid artery: compli- cumbersome and demanding.1 Intracranial surgery for any cations of the transsphenoidal surgery. Neurosurg 1992, 31 (4): 774-9; Discussion 778-9 other cause also makes the aneurysmal surgery more 2. Abbed KM, Ogilvy CS: Intracerebral hematoma from aneu- complicated by itself and these have specific management rysm rupture. Neurosurg Focus 2003, 15(4): E4 issues by themselves. In our series 4.45% (n = 14) patients 3. Andaluz N, Zuccarello M: Fenestration of the lamina terminalis had undergone previous intracranial surgery. Though this as a valuable adjunct in aneurysm surgery. Neurosurg 2004, 55(5): 1050-9 factor when analyzed by univariate regression had signifi- 4. Anderson CS, Feigin V, Bennett D, Lin RB, Hankey G, cance, it was not so with multivariate analysis. Jamrozik K, Australasian Cooperative Research on Subarach- noid Hemorrhage Study (ACROSS) Group: Active and passive Factor with poor outcome (complex aneurysms): After smoking and the risk of subarachnoid hemorrhage: an interna- tional population-based case-control study. Stroke 2004, 35 analyzing the results it was found that five factors were (3): 633-7 found to have a statistically significant association with the 5. Arnautovic KI, Al-Mefty O, Angtuaco E: A combined micro- poor outcome in both analysis and these were WFNS grade surgical skull-base and endovascular approach to giant and 4 - 5 at admission, clinical vasospasm, smoking > 30 years, large paraclinoid aneurysms. Surg Neurol 1998, 50(6): 504- 18; Discussion 518-20 Fisher grade 3 - 4 on CT image and aneurysm in the poste- 6. Ballard J, Kreiter KT, Claassen J, Kowalski RG, Connolly ES, rior circulation. Mayer SA: Risk factors for continued cigarette use after sub- arachnoid hemorrhage. Stroke 2003, 34(8): 1859-63 The presence of these factors in a patient was then used to 7. Biondi A, Jean B, Vivas E, Le Jean L, Boch AL, Chiras J, Van Effenterre R: Giant and large peripheral cerebral aneurysms: label these patients as ‘complex aneurysms’. Etiopathologic considerations, endovascular treatment, and long-term follow-up. Am J Neuroradiol 2006, 27(8): 1685-92 Various authors have tried to define aneurysm with com- 8. Broderick JP, Viscoli CM, Brott T, Kernan WN, Brass LM, plex issues as a need was always felt to prognosticate Feldmann E, Morgenstern LB, Wilterdink JL, Horwitz RI, 15 Hemorrhagic Stroke Project Investigators: Major risk factors for patients with SAH so as to apprise of their outcome. aneurysmal subarachnoid hemorrhage in the young are modifiable. Stroke 2003, 34(6): 1375-81 In the present study even though we have tried to include 9. Cavanagh SJ, Gordon VL: Grading scales used in the man- multiple factors to make a comprehensive analysis, there agement of aneurysmal subarachnoid hemorrhage: a critical review. J Neurosci Nurs 2002, 34(6): 288-95 still are other factors that may affect the outcome and have 10. Chhabra R, Gupta SK, Mohindra S, Mukherjee K, Bapuraj R, not been included. Moreover, the factors are interdependent Khandelwal N, Khosla VK: Distal anterior cerebral artery and one may directly or indirectly affect the other. These aneurysms: bifrontal basal anterior interhemispheric approach. were not mutually exclusive and there is a significant Surg Neurol 2005, 64(4): 315-9; Discussion 320 11. Chiang VL, Claus EB, Awad IA: Toward more rational overlap in patients. The study also suffers from being a prediction of outcome in patients with high-grade subarachnoid retrospective analysis, even though of a large number of hemorrhage. Neurosurg 2000, 46(1): 28-35; Discussion 35-6 patients. We however chose to analyze those that had 12. Dehdashti AR, Rilliet B, Rufenacht DA, de Tribolet N: Shunt- documented significant associations and chose to analyze in dependent hydrocephalus after rupture of intracranial aneu- rysms: a prospective study of the influence of treatment our study group to help in better prognosticate future modality. J Neurosurg 2004, 101(3): 402-7 patients. The introduction of term ‘complex aneurysm’ was 13. De Jesus O, Sekhar LN, Riedel CJ: Clinoid and paraclinoid done for easy identification and better record maintenance. aneurysms: surgical anatomy, operative techniques, and outcome. Surg Neurol 1999, 51(5): 477-87; Discussion 487-8 14. Dorai Z, Hynan LS, Kopitnik TA, Samson D: Factors related to Conclusions hydrocephalus after aneurysmal subarachnoid hemorrhage. Intracranial aneurysms present as spontaneous intracranial Neurosurg 2003, 52(4): 763-9; Discussion 769-71 haemorrhage most commonly in 5th and 6th decade. 15. Fisher CM, Kistler JP, Davis JM: Relation of cerebral vaso- Multiple factors either patient related, radiology related and spasm to subarachnoid hemorrhage visualized by computerized tomographic scanning. Neurosurg 1980, 6: 1-9 surgery related have a bearing on outcome. The presence of 16. Frontera JA, Fernandez A, Claassen J, Schmidt M, factors viz., WFNS grade 4 - 5, clinical vasospasm (DIND), Schumacher HC, Wartenberg K, Temes R, Parra A, smoking ≥ 30 years, Fisher grade 3 - 4, and posterior Ostapkovich ND, Mayer SA: Hyperglycemia after SAH: predic-

36 PAN ARAB JOURNAL OF NEUROSURGERY COMPLEX INTRACRANIAL ANEURYSMS • Kumar, et al

tors, associated complications, and impact on outcome. Roos YB, Tulleken CA, Vandertop WP, van Gijn J, Vos PE, Stroke 2006, 37(1): 199-203 Rinkel GJ: Timing of aneurysm surgery in subarachnoid 17. Hacein-Bey L, Connolly ES Jr, Mayer SA, Young WL, Pile- haemorrhage - an observational study in The Netherlands. Spellman J, Solomon RA: Complex intracranial aneurysms: Acta Neurochir (Wien) 2005, 147(8): 815-21 combined operative and endovascular approaches. Neurosurg 31. Ogilvy CS, Hoh BL, Singer RJ, Putman CM: Clinical and 1998, 43(6): 1304-12; Discussion 1312-3 radiographic outcome in the management of posterior 18. Hoh BL, Carter BS, Budzik RF, Putman CM, Ogilvy CS: Results circulation aneurysms by use of direct surgical or endovascular after surgical and endovascular treatment of paraclinoid techniques. Neurosurg 2002, 51(1): 14-21; Discussion 21-2 aneurysms by a combined neurovascular team. Neurosurg 32. 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Neurosurg 1999, 45(4): 827- outcome of patients with ruptured distal anterior cerebral artery 31; Discussion 831-2 aneurysms. Acta Neurochir (Wien) 2000, 142(11): 1241-6 44. Weir B, Grace M, Hansen J, Rothberg C: Time course of 29. Mocco J, Ransom ER, Komotar RJ, Schmidt JM, Sciacca RR, vasospasm in man. J Neurosurg 1978, 48(2): 173-8 Mayer SA, Connolly ES Jr: Preoperative prediction of long- 45. Zhao J, Wang S, Yang L, Zhao Y: Clinical experience of 153 term outcome in poor-grade aneurysmal subarachnoid hem- patients with posterior circulation aneurysms. J Clin Neurosci orrhage. Neurosurg 2006, 59(3): 529-38; Discussion 529-38 2005, 12(1): 17-20 30. Nieuwkamp DJ, de Gans K, Algra A, Albrecht KW, Boomstra S, Brouwers PJ, Groen RJ, Metzemaekers JD, Nijssen PC,

VOLUME 14, NO. 2, OCTOBER 2010 37 Education & Training

Distal median- and ulnar nerve compression syndromes

Jens Haase

Introduction Best understood in this context are the median- and ulnar nerve discussion elsewhere.15,17 compression syndromes.3-5,11-13,15-17,21,23,26-28,31,35,36,47 (p38-45) The median nerve may most often be compressed at the Most common among these is the median nerve compres- carpal tunnel distal to the wrist and the position of the sion in the carpal tunnel = carpal tunnel syndrome (CTS). median nerve under the TCL and can clearly be seen with Compression syndromes may be treated conservatively e.g. ultrasound imaging (Fig. 1). 14,17 by a splint or by steroid injections. Similarly they may be treated surgically.3,5,12,17,47

Most important and relevant to all kinds of surgery is the question ‘‘Will the actual procedure relieve symptoms caused by the disease and are the complication rates low and/or insignificant?’’. In Denmark with 5.4 million inhabitants a Hook of total of 5,000 hands with CTS are operated upon each year, hamate and equivalent figures are found in Sweden and US. These high numbers make it therefore relevant to discuss involved Pisiform surgical technique, as complications are possible with all surgical interventions. Transverse carpal ligament Figure 1 - Ultrasound image of the carpal tunnel. Most common surgical procedure since 1854, has been the open surgical release (OCTR) of the median nerve by cutting For a compressed median nerve an increase of the carpal the transverse carpal ligament (TCL).17 The first endoscope tunnel space can be obtained by cutting the TCL by two procedure (ECTR) for transecting the TCL was introduced different surgical methods e.g. 1) OCTR or 2) ECTR.17 in 1987 and many modifications have been described since then.1,8,22,32,40,49 A combination of open surgery and endoscope The major difference between an OCTR and an ECTR surgery has been introduced recently.24 Surgical treatment procedure is the way the TCL is cut. With the OCTR the of ulnar nerves has been performed since the19th century.5 surgeon cuts the TCL from outside the carpal tunnel thereby viewing all structures in - for the surgeon - a normal 3D Despite the fact that this procedure seems to be a rather fashion. This is in contrast to the ECTR where the TCL is trivial and simple operation, the problems of learning to cut from inside the carpal tunnel viewing it in a - for the 11,18,25 perform these operations requires thorough training. surgeon - new, and only 2D fashion.17 This endoscopic procedure is for many reasons much more difficult and Median nerve compression - CTS must be learned thoroughly through training and regular For diagnosing CTS the reader is referred to a more detailed practice.18

Through literature validation, both surgical methods seem Faculties of Engineering, Science & Medicine to lead to the same results but the complications of the Department of Health Science & Technology methods are different, apparently being more severe among Aalborg University 1,9,11,17,34,39, 46, 49,50 Denmark the endoscope method.

Correspondence: To carry out carpal tunnel release surgery the surgeon needs Prof. Jens Haase Faculties of Engineering, Science & Medicine anaesthesia, and anaesthetic complications must be added to Department of Health Science & Technology the surgical failures.6,17 Aalborg University Fredrik Bajers Vej 7, E-4 9220 Aalborg Anaesthesia for CTS treatment comprise of: Denmark 1. Local infiltration anaesthesia (LA)

38 PAN ARAB JOURNAL OF NEUROSURGERY DISTAL MEDIAN– AND ULNAR NERVE COMPRESSION SYNDROME • Haase

2. Local infiltration anaesthesia with use of tourniquet carpal tunnel from the proximal wrist crease. After 4 - 5 3. Intravenous anaesthesia with use of tourniquet and minutes the quality of sensation is tested with small pin 4. General anaesthesia. pricks at the anaesthetic area and outside. Thereby the patient will relax, learning the difference between normal The author prefers the first simple method (LA) where the painful areas and the operative analgesic area. The hand is skin is infiltrated in the hand by injection of lidocain 1% then placed on a well-bolstered separate arm table. If it is with adrenaline. Hand surgeons in many countries tradi- the right hand, the right-handed surgeon places himself at tionally use LA combined with extremity exsanguinations the ulnar side of the hand to be operated upon. Visa versa if to obtain a bloodless field. Whereas a bloodless field is the surgeon is left-handed. The reason being that the absolutely mandatory for dissecting tendons and synovial surgeon then, in all cases, will cut and dissect from proximal tissue, it is not the case for nerve dissection. The entrap- towards distal, whereby the risk of injuring nerve branches ment (CTS) cause cyanotic colour changes of the nerve that diverse from proximal to distal is reduced. and/or distended vessels on the nerves, which cannot be visualized in a bloodless field.19,30 Postoperative venous Before the operation, the surgeon prepares the microscope. haemorrhages in the operative field are also more common The surgeon must set the oculars and interpupillary distance following use of tourniquet. However, if one compares correctly and the microscope may be draped. He must also blood oozing by the use of tourniquet and simple local determine if his own glasses shall be used or not. The lidocain + adrenaline, the simple infiltration of skin with operative field is, with this type of surgery, rather stationary adrenalin is superior to tourniquet.6 so draping can be excluded as the hand can be moved to maintain focus. The operative microscope is now brought Intravenous anaesthesia combined with tourniquet is a to its place (Fig. 2). standard for many hand surgical procedures, but is more complicated than LA.

General anaesthesia may be indicated in cases of re- operations with significant scarring or if the patients are very nervous.

Surgical techniques Open carpal tunnel release Open surgical section of the TCL has been the gold standard surgical treatment for patients with CTS since 1854.12,17,21,23 Cutting the TCL with a scalpel under direct vision produces reliable symptom relief in the vast majority of cases.17 However, despite this high clinical success rate, transient postoperative symptoms such as ‘‘pillar pain’’, scar tenderness, or hand weaknesses are known to occur.22, Figure 2 - Operative microscope. 37,44 The author uses two separate pairs of gloves for the surgery Preoperatively the patient is carefully informed about how for prevention of infection from skin flora.45 A 3 - 4 cm the operation is carried out. It is thus described how local long incision is made with a 15-blade from the distal crease anaesthesia is used and that he/she can feel “something”, of the hand towards the interdigital space 3/4. All bleeding but no pain during the operation. During the operation vessels must be carefully occluded with bipolar coagulation, additional local anaesthetics can be applied, if necessary. with a low setting.29 Thereby postoperative blood oozing Instruments needed are simple: a surgical knife with a 15- in the wound is very seldom experienced. The small blade, a small retractor, that is used to hold the skin edges retractor is placed to hold the skin edges. With aid of the apart, and an operative microscope for better viewing and microscope the palmar aponeurosis is now visualized and light. Magnifying loupes may be used in case a microscope cut longitudinally. Eventual cutaneous nerve structures or is not available. The author always advocates use of some vessels are avoided. It is at this time exchange of the first kind of magnification. pair of gloves that are always contaminated with skin bacterial flora is done. The TCL with its white transverse The hand/arm is carefully prepped with hexidine alcohol or fibres is now visualized and is opened by cutting with a 15- similar disinfection material. With a thin needle 4 - 5 cc blade in the middle, slightly ulnar to the midline. When the lidocain 1% with adrenaline is infiltrating the skin over the carpal tunnel contents are encountered, the incision is

VOLUME 14, NO. 2, OCTOBER 2010 39 DISTAL MEDIAN– AND ULNAR NERVE COMPRESSION SYNDROME • Haase carried further distally to the rim of the TCL until the above systolic pressure are often necessary to obtain a normal yellow fat is visualized. Then the proximal part of bloodless field. The introduction of the endoscope into the the ligament is cut and eventually part of the antebrachial carpal tunnel may increase pressure on the median nerve fascia, again keeping ulnar (Fig. 3). and cause unpleasant sensations for the patient if carried out in LA.17 Even with perfectly planned endoscope surgery the surgeon must be prepared to change to an open type of surgery if anatomical landmark identification is not possible.

Single-portal techniques are those in which a single skin incision is made in the proximal wrist crease.1,22 Dual- portal techniques are those in which a second supple- mentary small incision is made in the palm when the endoscope/obturator has reached this area.8,40 Both methods require some degree of hyperextension and fixation of the hand during surgery. This hyperextension decreases the volume of the carpal tunnel, as does the introduction of the endoscope equipment. Figure 3 - Median nerve inside the carpal tunnel. 1) ECTR single-port technique: A small incision is The palmar motor branch of the median nerve is usually made in the distal hand crease on the ulnar side of the long never seen with this approach. The median nerve is palmar muscle tendon. Through this an obturator is intro- visualized in the tunnel and hourglass shape and eventual duced blindly into the carpal tunnel developing a channel 17 cyanosis indicating the compression site is seen (Fig. 4). for the endoscope. The endoscope sheet is then inserted directly in the carpal tunnel through this channel followed by introduction of the endoscope. A window near the tip of the system angled upwards makes it possible to continu- ously view the undersurface of the TCL through the endoscope. A hook knife cutting blade is then inserted via the endoscope and cutting of the TCL takes place often from distal to proximal viewing the ligament, but not the median nerve.1,17

2) ECTR dual-port technique: With the two portal techniques the introducer/obturator is also passed blindly through the carpal tunnel through a similar small transverse cut in the distal skin crease. When the obturator reaches the Figure 4 - Hourglass shape of compressed median nerve. palm a supplementary contra incision is made here and the tip of this introducer is thereafter pushed out through the Movements of the tendons and the median nerve are skin (Fig. 5). obtained by pulling the fingers and secures that the contents in the canal is free. The TCL edges are coagulated with Loop around median nerve bipolar coagulation and the skin closed in one layer with Long single 5 - 0 sutures. The wound is covered with a band-aid flexors and the hand bolstered leaving the fingers free for active tendons movements immediately after surgery. The hand is kept high for the first day and skin sutures removed after 12 - 14 days. Decreased wrist movements are common after two weeks (relative immobilization) and should be treated by active movements after the skin sutures have been removed.

Endoscopic carpal tunnel release techniques Visualization of anatomical structure is of course of para- mount importance when performing endoscope procedures.20 Blood obscures vision and extremity exsanguinations with TCL - cut edges an Esmarch bandage followed by inflation of tourniquet Figure 5 - Two-portal endoscope technique.

40 PAN ARAB JOURNAL OF NEUROSURGERY DISTAL MEDIAN– AND ULNAR NERVE COMPRESSION SYNDROME • Haase

The endoscope is then passed into the introducer from the incisions must take aim of visualization of the ligament and proximal end of the carpal tunnel and the channel is then the contents of the carpal tunnel. Lack of magnifi- inspected for contents. The custom designed instrumen- cation and light both disturb the possibilities of viewing the tation protects the median nerve and flexor tendons, and carpal ligament sufficiently. Many open cases are rather positioning of the slotted cannula through the two portals closed as a “Mickey”-probe is introduced into the canal and ensures a stable surgical environment.8,40 The surgeon the TCL thereafter cut on this probe that is supposed to inserts a hook knife via the proximal port and advances it prevent lesions of the median nerve. Similarly, some behind the distal end of the TCL. The TCL is caught by the surgeons use a pair of scissors to cut the ligament with e.g. knife and the ligament is cut with a backwards pull. introducing one branch blindly into the canal. They cannot see the distal cut of the TCL. “To see” for the author, is the Complications due to operative treatments: first and most important factor for reducing complications Nowadays, complications range from 0 - 24%.17 This huge in OCTR.17 With the technique described here, the median variation documents the influence of the surgical learning nerve is clearly viewed due to magnification and excellent curve/ and or surgical competence. According to recent light in the operative field provided by the operative studies, the overall complication rate should be in the range microscope. Performing the skin incision the larger cuta- of 1 - 2% in experienced hands for both ECTR and OCTR neous nerves may be seen and protected. The white surgery.2,10,13,34,41,42,44,49-51 transverse fibres of the TCL are also easily visualized with the microscope. Another important point is that the author The most common complication due to OCTR and ECTR never uses a tourniquet, thereby all degrees of nerve is inadequate cutting of the distal part of the TCL.17 These compression including colour changes and vessels stasis are patients will not experience the immediate normal relief of clearly shown. The motor branch of the median nerve is the painful hand paraesthesia after the operation. So, if the never found during this dissection and thus never sectioned patient still complains of painful paraesthesia after 2 - 3 because you see all necessary details of the procedure - days the surgeon must consider this complication. nothing is blind. For teaching / learning activities it is also possible to preserve the whole operation on a DVD or as Other complications are direct surgical lesion of the median slides in a PowerPoint presentation for validation (Möller- nerve including its motor branch and compression neuro- Wedelc). pathy due to pressure by the endoscope. A lesion of the palmar cutaneous branch of median nerve may often lead to Most series published are "personal" and thus not suited for a complex regional pain syndrome.17 Hypertrophic generalization - which is still done. The surgical learning hypersensitive skin scar is only seen if the skin incision has curve is important and because one great endoscope surgeon been carried proximal to the distal wrist crease, whereas or micro-neurosurgeon can carry out CTS operations with slight pillar pain is common in the first weeks after OCTR minimal complications this does not invariably indicate that surgery. Injury to the superficial vascular arch distal in the all surgeons will accomplish the same.17 Endoscope tech- hand and wound infection are rarely encountered. De- niques has many proponents who cite the potential benefits creased grip strength is common with both methods for the of faster patient recovery time, less incision pain and first 2 - 3 postoperative months, but will normally gradually improved grip strength recuperation.1,10,11,22,31,32,34,40-43 No disappear. Erroneous decompression of the ulnar nerve in controlled randomized series exist to prove these state- Guyon´s canal instead of the median nerve may be the ments. Application of endoscopy techniques has not result of lack of experience, both in OCTR and in decreased operative expenses, nor increased operative effi- ECTR.17,30 ciency, or improved intraoperative visualization (compared with conventional OCTR).42 Results of surgical treatment of CTS Carpal tunnel syndrome must be graded in different Evans stated in an editorial in Journal of Handsurgery: “The stages.17,39 In the early stages of CTS, total relief of pain serious complications (… to CTS operations) must be and nightly paraesthesia is obtained in close to 96% of all regarded as the result of: careless or inexperienced surgery cases within 24 - 48 hours. In later stages of CTS, full and the established principle of surgery under direct vision 11 return of sensation and muscle power cannot be anticipated, has provided reliable protection against disaster". as regeneration of the axonal injured nerve fibres will take up to several years. The influence of postoperative training Proper formal training in both open and endoscope tech- 18 - or “remodeling the brain” - is very important.38 niques must be obtained.

One of the main causes for surgical mistakes in OCTR is Ulnar nerve entrapments improperly placed incisions.12,21 Hands are different and Ulnar nerve anatomy: Many variations in the ulnar

VOLUME 14, NO. 2, OCTOBER 2010 41 DISTAL MEDIAN– AND ULNAR NERVE COMPRESSION SYNDROME • Haase nerve anatomy exist. The ulnar nerve follows the medial cranon is infiltrated with 5 cc of local anaesthetics. The head of the triceps muscle and enters the retrocondylar skin is then opened from the middle of the line between the groove behind the olecranon heavily protected in a fibro- olecranon and the epicondyle distal to the groove. The osseous tunnel. Distally the nerve gives off sensory muscle aponeurosis over the cubital tunnel is opened but the branches to the elbow joint and skin at the olecranon and retrocondylar groove is left untouched. After cutting the motor branches to the ulnar carpal flexor- and the medial muscle-aponeurosis the Osborne compartment is opened half of the deep flexor muscles.2,5,23,24,35,36 The space just and the ulnar nerve is located and dissected beneath the two outside and distal to the groove created by an arcuate heads of the ulnar carpal flexor muscle. The author always aponeurosis (“Osborne ligament”) and the two heads of the uses operative microscope at this stage. When the fascia ulnar carpal flexor muscle is also called the Osborne has been opened, the arm is moved to see how the nerve is compartment.33 The ulnar nerve digs down and continues sliding. The author, does not open the fibro-osseous tunnel into the forearm, lying between the ulnar carpal flexor (retrocondylar groove) unless compression here is sus- muscle and the deep long flexor muscle closely joined with pected by inspection. If opened, it is only the distal half of the ulnar artery. Distal at the wrist it enters Guyon´s canal the fibro-osseous tunnel that is cut to allow release of the where it divides into sensory branches to the volar ulnar ulnar nerve. Other authors do open the whole fibro-osseous part of the hand and 4th and 5th fingers, and superficial and tunnel routinely releasing the nerve and leaving it to lie deep motor branches to the small hand muscles.19 Nerve freely only covered by connective tissue. The patient needs anastomosis between the ulnar- and median nerves exists in to have a deep condylar groove in these cases. The risk of a the forearm and distally in the palm in many patients. A new postoperative chronic nerve irritation may easily gliding movement of up to 4.7 cm of the only 2 - 4 fascicles develop if the released nerve slides over the epicondyle of the ulnar nerve is taking place during flexion/extension with elbow flexion. The skin is closed in two layers. The of the elbow in the average person.47 The fibro-osseous patient is urged to move the elbow freely immediately after canal size decreases during flexion and increases during surgery. At night an elbow bandage is used to prevent extension. In contrast, the Osborne compartment space maximum elbow flexion for the next 2 - 3 weeks. Skin decreases with elbow extension. Therefore, we may antici- sutures are removed after 14 days.16,33 pate two different types of entrapment and subsequently two different operative treatments. The strain at elbow Open surgery is still the standard but cubital tunnel release flexion is maximal directly behind the medial epicondyle with endoscope assistance has been advocated with a new with pressures that increases up to 3 times by elbow flexion. micro/endo version in 2006.24 The author has no personal The ulnar nerve may very rarely be compressed proximal to experience with this method. the sulcus. Most common it is found in younger patients compressed at the level of medial epicondyle due to recent 2) Epicondylectomy: Medial epicondylectomy is another elbow fractures. If the fibro-osseous tunnel size is reduced hypothetical way to release pressure on the ulnar nerve at by trauma, the ulnar nerve may easily be entrapped here. In the elbow. This operation demands general anaesthesia. the cubital compartment an elbow flexion causes stretching Excision of the proper amount of bone is critical to the of the ligament and the compartment flattens leading to success of this procedure. If too much bone is excised pressure on the ulnar nerve.27,33,36 damage to the medial collateral ligament of the elbow, deep in the groove, may lead to a valgus position of the elbow Conservative treatment: Involves prevention of bend- joint and painful instability of the medial elbow. Osteo- ing the elbow or compressing the nerve by sitting with the myelitis is another severe complication to this operation. elbow on the table, which is the most common treatment.17 Heterotope ossification may be the result of osteotomy and thereby continuous minor trauma to the nerve may occur as Surgical treatment must be reserved for long-lasting it is now unprotected.23,36,37 The author has also never symptoms and thorough documentation. For indications to carried out this operation. surgery, the readers are kindly asked to read the more detailed discussion.5,7,27,48 For the simple “Osborne” decom- 3) Ulnar nerve decompression with transpositions pression LA is sufficient. Additional anaesthetics can be of the nerve: Surgical decompression of the ulnar nerve applied if necessary and no tourniquet is needed.2,17,33 With with subsequent anterior transposition to the medial transposition procedures general anaesthesia is preferred.2,36 epicondyle is often suggested. Hereby the position of the The minimal invasive technique by endo/ micro-surgery ulnar nerve should be shorter and tension of the nerve thus may have an advantage, but needs long-term validation.24 be relieved. Interfascicular gliding should similarly be improved. The ulnar nerve may be positioned subcutane- Simple decompression techniques ously above the muscle fascia or submuscular either under 1) “Osborne” operation: The skin distal to the ole- or inside the pronator teres muscle.2,5,23,24,27 Lesions of

42 PAN ARAB JOURNAL OF NEUROSURGERY DISTAL MEDIAN– AND ULNAR NERVE COMPRESSION SYNDROME • Haase cutaneous branches to the olecranon/ elbow joint may result technique exist. Postoperatively, the elbow is immobilized from transposition techniques and long dissection of mus- in 45 degrees of flexion in a post mould or cast for 3 - 4 cular and cutaneous branches may also be needed. weeks.2,23

A) The subcutaneous technique: An incision starts 4) Guyon´s canal - wrist - decompression: A surgical some 8 cm proximal to the medial epicondyle and contin- decompression is carried out most often from the volar side ues in front of this to 6 cm distal over the flexor carpi of the wrist with a straight incision with a Z- at the wrist ulnaris muscle. Branches of the medial antebrachial creases. Another approach is from the ulnar side of the cutaneous nerve are carefully protected to prevent lesions hand via a Z-shaped incision lateral along the hypothenar. and neuroma development. The ulnar nerve is found proxi- Hereafter the pisiform bone is removed whereby the deep mally and then dissected distally. It is freed from all septa, motor branch is decompressed. Minor postoperative prob- Osborne ligament and flexor carpi ulnar fascia. The distal lems are the result of this latter procedure.2,19 medial intermuscular septum should also be cut protecting major vessels. The ulnar nerve is mobilized in front of the Results of surgical treatment of ulnar nerve medial epicondyle preserving the motor branches to the compression flexor muscles. If necessary, the articular branch to elbow The ulnar neuropathy is manifested by multiple patho- joint should be preserved too. The nerve now lies on the genesis factors. This manifests in different clinical situations fascia and the subcutaneous fat is sutured to the tip of the with similar symptoms. The acute ulnar nerve neuritis is a medial epicondyle with non-absorbable sutures, thereby a completely different entity then that of an ulnar nerve subcutaneous tunnel is created. It is ensured that the nerve neuropathy or a median nerve neuritis. We have no lies and moves freely (Fig. 6). controlled and validated information with regard to which surgical treatment to offer.2,7 We must accept this and be Ulnar nerve transposed cautious in our suggestions of what type of treatment the patients should receive. Prevention seems better than any surgical cure in the first stadium of the ulnar nerve diseases. It seems more than relevant to carry out a careful electrophysiological examination before surgery is decided upon.17,19

Conservative treatment is based primarily on preven- tion of compression. Keep the elbow as straight as possible if elbow flexions provoke symptoms. Use headsets instead of mobile telephone, adjust workspace if necessary and use elbow protectors if compression seems to be leading to symptoms. This will, in most cases lead to reduced symp- Osborne compartment toms. Only if these treatments fail, surgery can be consid- opened 2 Cutaneous branch Sulcus ered. Olecranon for olecranon Figure 6 - Ulnar nerve transposed subcutaneously. Basically, the operative treatments consist of the "simple" decompression with a minimum of complications. Simple decompression will, in the majority of cases, be the best Then the skin is sutured in two layers. Postoperatively, the 7 elbow is immobilized in a post mould or cast at 45 degrees choice of surgery. The technically, much more complicated nerve-transposition procedures of the ulnar nerve, leads to of flexion for 2 weeks. Active mobilization can start after 2,7,27,33 two weeks. This is the simplest transposition technique but many complications. Transposition is carried out in must be carried out meticulously to prevent later kinking of cases where medial dislocation of the nerve is a prominent the nerve both proximally and distally.2,7,27 feature and the choice of subcutaneous and submuscular transposition is not clear - the author favours the simplest B) Submuscular techniques: In submuscular trans- choice. position the initial dissection is as with the subcutaneous technique. The idea is to position the ulnar nerve deeper Complications inside the muscle tissue. Therefore the origin of the flexor- A kinking of the ulnar nerve can easily occur against the pronator muscle group is released and the nerve positioned medial intermuscular septum and under the aponeurosis under these lying on the brachial muscle. Then the flexor- arch between the two heads of the ulnar flexor carpi muscle. pronator muscle is reattached securely. Variations of this This happens if sufficient decompression is not carried

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2,7 out. Most common complications to operative treatment syndrome. BMJ 2006, 332: 1463-1464 are injury to the nerve while decompressing it or trans- 16. Grant GA, Goodkin R, Kliot M: Evaluation and surgical posing it and neuromata of the medial antebrachial cutaneous management of peripheral nerve problems. Neurosurg 1999, 36 44(4): 825-839; Discussion 839-840 nerve. 17. Haase J: Carpal tunnel syndrome - a comprehensive review. In: Pickard JD (ed), Advances and Technical Standards in Endoscope decompression has been described, but only Neurosurgery. Wien, Springer-Verlag 2007, Vol. 32, pp 178-249 lately with the combined endo-micro technique and it 18. Haase J: How to develop the surgical dexterity needed for seems to be a tool to be taken seriously in the future.24 endoscope neurosurgery? PAJNS 2009, 13(2): 1-8 19. Højer-Pedersen E, Haase J: The ulnar tunnel syndrome. Acta Neurochir (Wien) 1980, 52: 121-7 Conclusion 20. Hong JT, Lee SW, Han SH, Son BC, Sung JH, Park CK, Park Surgical treatment of median- and ulnar nerve compression CK, Kang JK, Kim MC: Anatomy of neurovascular structures around the carpal tunnel during dynamic wrist motion for syndromes are very rewarding. The operations demands endoscopic carpal tunnel release. Neurosurg 2006, 58(1 similar micro techniques as with aneurysm surgery. All Suppl): ONS127-33 techniques must be properly learned before being applied to 21. Hudson AR, Wissinger JP, Salazar JL, Kline DG, Yarzagaray L, our patients. Danoff D, Fernandez E, Field EM, Gainsburg DB, Fabi RA, Mackinnon SE: Carpal tunnel syndrome. Surg Neurol 1997, 47(2): 105-114 References 22. Jimenez DF, Gibbs SR, Clapper AT: Endoscopic treatment of 1. Agee JM, McCarroll HR Jr, Tortosa RD, Berry DA, Szabo RM, carpal tunnel syndrome: a critical review. J Neurosurg 1998, Peimer CA: Endoscopic release of the carpal tunnel: a 88(5): 817-826 randomized prospective multicenter study. J Hand Surg (Am) 23. Kline D, Hudson A (eds): Nerve Injuries. Philadelphia, WB 1992, 17(6): 987-995 Saunders, 1995 2. Assmus H, Antoniadis G (eds): Nervenkompressions-syndrome. 24. Krishnan KG, Pinzer T, Schackert G: A novel endoscopic Germany, Steinkopff Verlag 2008 technique in treating single nerve entrapment syndromes with 3. Assmus H, Antoniadis G, Bischoff C, Haussmann P, Martini special attention to ulnar nerve transposition and tarsal tunnel AK, Mascharka Z, Scheglmann K, Schwerdfeger K, Selbmann release: clinical application. Neurosurg 2006, 59(1 Suppl 1): HK, Towfigh H, Vogt T, Wessels KD, Wüstner-Hofmann M: ONS89-100 Diagnosis and therapy of carpal tunnel syndrome. Handchir 25. Long DM: Competency-based training in neurosurgery: the Mikrochir Plast Chir 2007, 39(4): 276-88 next revolution in medical education. Surg Neurol 2004, 61(1): 4. Atroshi I, Larsson GU, Ornstein E, Hofer M, Johnsson R, 5-14; Discussion 14-25 Ranstam J: Outcomes of endoscopic surgery compared with 26. 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J Bone Joint Surg American Society for Surgery of the Hand: results of a (Am) 1989, 71(5): 679-683 questionnaire. J Hand Surg (Am) 1987, 12(3): 384-391 33. Osborne G: Compression neuritis of the ulnar nerve at the 11. Evans D: Endoscopic carpal tunnel release - the hand doctor´s elbow. J Hand Surg (Eur) 1990, 2: 10-13 dilemma - Editorial. J Hand Surg (Br) 1994, 19(1): 3-4 34. Palmer AK, Toivonen DA: Complications of endoscopic and 12. Fernandez E, Pallini R, Lauretti L, Scogna A, La Marca F: open carpal tunnel release. J Hand Surg (Am) 1999, 24(3): Carpal tunnel syndrome. Surg Neurol 1997, 48(4): 323-325 561-565 13. Geoghegan JM, Clark DI, Bainbridge LC, Smith C, Hubbard R: 35. Pécina MM, Markiewitz AD, Krmpotic-Nemanic J (eds): Tunnel Risk factors in carpal tunnel syndrome. J Hand Surg (Eur) Syndromes: Peripheral Nerve Compression Syndromes, 3rd 2004, 29(4): 315-320 Ed. Florida, CRC Press 2001 14. Gerritsen AA, de Vet HC Scholten RJ, Bertelsmann FW, de 36. Posner MA: Compressive ulnar neuropathies at the elbow: I. Krom MC, Bouter LM: Splinting vs surgery in the treatment of Etiology and diagnosis. J Am Acad Orthop Surg 1998, 6(5): carpal tunnel syndrome: A randomized controlled trial. JAMA 282-8 2002, 288(10): 1245-1251 37. Rodner CM, Katarincic J: Open carpal tunnel release. 15. Graham B: The diagnosis and treatment of carpal tunnel Techniques in Orthopaedics 2006, 21(1): 3-11

44 PAN ARAB JOURNAL OF NEUROSURGERY DISTAL MEDIAN– AND ULNAR NERVE COMPRESSION SYNDROME • Haase

38. Rosén B, Lundborg G: Sensory re-education after nerve 45. Sørensen P, Ejlertsen T, Aaen D, Poulsen K: Bacterial repair: aspects of timing. Handchir Mikrochir Plast Chir 2004, contamination of surgeons gloves during shunt insertion: a pilot 36(1): 8-12 study. Br J Neurosurg 2008, 22(5): 675-77 39. Rotman MB, Enkvetchakul BV, Megerian JT, Gozani SN: Time 46. Stütz NM, Gohritz A, van Schoonhoven J, Lanz U: Revision course and predictors of median nerve conduction after carpal surgery after carpal tunnel release - Analysis of the pathology tunnel release. J Hand Surg (Am) 2004, 29(3): 367-372 in 200 cases during a 2 year period. J Hand Surg (Br) 2006, 40. Russell SM: Dual-portal endoscopic release of the transverse 31(1): 68-71 ligament in carpal tunnel syndrome: Results of 411 procedures 47. Sunderland S: Nerves and nerve injuries. Churchill Livingstone with special reference to technique, efficacy, and complications 1972 - Commentary. Neurosurg 2006, 59(2): 340 48. Tindall SC: Simple decompression to treat ulnar entrapment 41. Sanz J, Lizaur A, Sánchez Del Campo F: Postoperative changes within the cubital tunnel. In: Al-Mefty O, Origitano TC, Harkey of carpal canal pressure in carpal tunnel syndrome: A prospec- HL (eds): Controversies in Neurosurgery. New York, Thieme tive study with follow-up of 1 year. J Hand Surg (Br) 2005, 30 Medical Publishers 1996, pp 340-9 (6): 611-614 49. Trumble TE, Diao E, Abrams RA, Gilbert-Anderson MM: 42. Scholten RJ, Gerritsen AA, Uitdehaag BM, van Geldere D, de Single-portal endoscopic carpal tunnel releases compared with Vet HC, Bouter LM: Surgical treatment options for carpal open release: a prospective, randomized trial. J Bone J Surg tunnel syndrome. Cochrane Database Syst Rev 2004, 18(4): (Am) 2002, 84(7): 1107-1115 CD003905 50. Vasen AP, Kuntz KM, Simmons BP, Katz JN: Open versus 43. Schuind F: Canal pressures before, during, and after endo- endoscopic carpal tunnel release: A decision analysis. J Hand scopic release for idiopathic carpal tunnel syndrome. J Hand Surg (Am) 1999, 24(5): 1109-1117 Surg (Am) 2002, 27(6): 1019-1025 51. Wong KC, Hung LK, Ho PC, Wong JM: Carpal tunnel release. 44. Shapiro S: Microsurgical carpal tunnel release. Neurosurg A prospective, randomised study of endoscopic versus limited- 1995, 37(1): 66-70 open methods. J Bone Joint Surg (Br) 2003, 85(6): 863-868

VOLUME 14, NO. 2, OCTOBER 2010 45 Original Article

Experience with peripheral nerve injury in lower extremities

Hatem Badr, Mohammed Kassem, Ahmed Zaher, Mohammed Mansour, Ashraf Shaker

Abstract: This study reports the results of 43 operations performed on nerves of lower extremities of 43 patients during a period of 7 years from 1999 - 2005 in Mansoura University Hospital and Mansoura Emergency Hospital. There were 15 patients with isolated sciatic nerve injury, 24 with isolated peroneal nerve injury and 4 with isolated tibial nerve injury. All patients were treated with nerve exploration within 1 hour to 7 months after injury and were followed- up for 6 months to 4 years. There were 22 nerve lesions not in continuity (9 needed suture repair and 13 needed sural nerve graft repair), while 21 nerve lesions were in continuity (16 partial lesions needed neurolysis and 5 complete lesions needed neuroma excision and suture repair). Analysis of the outcome of surgical treatment was performed with respect to the following parameters: period between the injury and operation, patient age, type of injured nerve, mechanism of injury and type of surgical intervention. Overall significant outcome (≥ 3 Louisiana State University Health Science grade) was obtained in 53.5% (sciatic nerve 46%, peroneal nerve 54% and tibial nerve 75%). According to the type of intervention and lesion categories; lesions not in continuity had a significant outcome 41% (suture repair 55.5% while graft repair 31%), and lesions in continuity had a significant outcome 67% (lesions underwent neurolysis 75%, while lesions underwent suture repair 40%). Useful function was achieved in 3 (43%) of 7 patients with grafts less than 6 cm in length and in only 1 (16%) of 6 patients with grafts greater than 6 cm in length. The mean time to recovery in patients who underwent surgery was 18 months (range: 1 - 32).

In conclusion, the most favourable outcome was obtained with lesions that result in partial lesion in continuity. Considering the rate of spontaneous recovery of post-injection nerve injuries of the sciatic nerve and early onset of skeletal deformities, a closed nerve injury of the lower limb with no recovery within 3 months should always undergo surgery, even if complete functional outcome is not always guaranteed. (p46-50)

Introduction Peripheral nerve injuries are a major source of chronic Patients and methods disability. Advances in microsurgery and a better under- Between 2000 and 2006, we evaluated 43 patients with standing of nerve healing have greatly improved the peripheral nerve injuries in the lower limb at Mansoura outcomes of nerve repair in the past two decades.13 University Hospital and Mansoura Emergency Hospital to assess functional outcome following repair of the nerve Lower-extremity nerve injuries are relatively less common injuries in relation to variable factors. There were 15 patients than injuries to upper-extremity nerves.2,35 with isolated sciatic nerve injury, 24 with isolated peroneal nerve injury and 4 patients with isolated tibial nerve injury. Aim of the work was to assess functional outcome following surgical repair of the nerve injuries in lower limb All patients were preoperatively and postoperatively as- in relation to period between the injury and the operation, sessed both clinically and electrophysiologically, and were patient age, type of injured nerve, mechanism of injury and followed-up for a period that ranged from 6 months to 4 type of surgical intervention. years postoperatively.

Patients were treated with nerve exploration within 1 hour to 7 months after injury. Surgical procedures were done Department of Neurosurgery Mansoura University according to status of the nerve injury. If the nerve was in Egypt continuity, the procedure was either neurolysis or epineureal

Correspondence: circumferential suture with 6/0 proline suture after excision Dr. Mohamed Kassem of neuroma. If the nerve was not in continuity, the Department of Neurosurgery procedure was either epineureal circumferential suture with Mansoura University PO Box 35516-74 6/0 proline suture or sural nerve cable graft. Egypt Email: [email protected] Surgical exploration was conducted to patients with persistent

46 PAN ARAB JOURNAL OF NEUROSURGERY PERIPHERAL NERVE INJURY IN LOWER EXTREMITIES • Kassem, et al deficit showing no spontaneous clinical or electrophysio- There were 15 patients with isolated sciatic nerve injury logical improvement or in cut wounds with nerve known to (35%), 24 with isolated peroneal nerve injury (56%) and 4 be cut. with isolated tibial nerve injury (9%).

Preoperative and postoperative clinical evaluation was According to the type of injured nerve, there were 15 sciatic performed using Louisiana State University Health Science nerve injuries in different levels with only 7 cases (46%) (LSUHS), grading system in which grade 3 or more is con- showing significant outcome (> G3), 24 peroneal nerve sidered a favourable functional outcome (Tables 1 and 2).8 injuries showing significant outcome in 13 of them (54%) and 4 tibial nerve injuries with only 3 (75%) showing significant outcome (Table 3) (Fig. 1). Table 1 - The LSUHS grading system for motor and sensory function. Grade Evaluation Description Table 3 - Functional outcome in relation to type of nerve. Individual muscle grades Type of nerve No. of lesions Significant outcome (> G3) 0 Absent No contraction Sciatic 15 7 (46.6%) 1 Poor Trace of contraction Peroneal 24 13 (54%) 2 Fair Movement against gravity only Tibial 4 3 (75%) 3 Moderate Movement against gravity and some (mild) resistance 4 Good Movement against moderate resistance No. of lesions Significant outcome > G3 5 Excellent Movement against maximum resistance 25 Sensory grades 0 Absent No response to touch, pinprick or pressure 20 1 Poor Testing produces hypaesthesia or paraesthesia; deep pain recovery in autonomous zones 15 2 Fair Sensory response sufficient for grip and slow protection; sensory stimuli mislocalized 10 3 Moderate Response to touch and pinprick in autonomous zones; sensation mislocalized and not normal 5 4 Good Response to touch and pinprick in autonomous zones; response localized but sensation not normal 0 Sciatic Peroneal Tibial 5 Excellent Near normal response to touch and pinprick in entire field including autonomous zones Figure 1 - Functional outcome in relation to type of nerve.

Table 2 - The LSUHS criteria for grading nerve injury. Grade Description According to the level of injury (Table 4); in the buttock 9 0 (absent) No muscle contraction; absent sensation sciatic nerve injuries were managed with significant improve- 1 (poor) Proximal muscles contract but not against gravity; ment (> grade 3) in only 4 (44%), in the thigh 6 sciatic sensory grade is 1 or 0 nerve injuries were managed with significant outcome in 2 (fair) Proximal muscles contract against gravity and distal only 3 (50%) while in the knee 28 peroneal and tibial nerve intrinsic muscles do not contract; sensory grade, if injuries were reported with favourable outcome in only 16 applicable, is usually < 2 (59%) (Table 4). 3 (moderate) Proximal muscles contract against gravity with some resistance; some distal muscle contract against little resistance; sensory grade is usually 3 Table 4 - Functional outcome in relation to level of injury. 4 (good) All proximal and some distal intrinsic muscles of the foot contract against pressure with some resistance; Level of injury No. of lesions Significant outcome (> G )3 sensory grade is > 3 Buttock 9 5 (excellent) All muscles, including intrinsic muscles of the foot, • Sciatic 9 4 (44%) contract against moderate resistance; sensory grade is > 4 Thigh 6 • Sciatic 6 3 (50%) Knee 28 • Peroneal 24 13 (54%) Results • Tibial 4 3 (75%) In our series, there were 39 males and 4 females, mean age was 25 years ranging from (3 - 53) years.

VOLUME 14, NO. 2, OCTOBER 2010 47 PERIPHERAL NERVE INJURY IN LOWER EXTREMITIES • Kassem, et al

According to the period between injury and tion results which were similar in both groups.5 operation: Primary repair was performed in 6 lesions with significant outcome in 4 cases (66%), while secondary In our series, sciatic nerve showed the worst outcome repair and graft were performed in 21 lesions with signifi- (46%) in comparison to peroneal nerve (54%) and tibial cant outcome in only 7 (33%). All patients with a period of nerve (75%), this is confirmed in previous studies.10 more than 8 months between injury and operation (5 cases) Roganovic classified the peripheral nerves according to showed no satisfactory outcome (Table 5). intensity of regenerative potentials into 3 groups; excellent (radial, muscalocutaneous and femoral nerve), moderate (median, ulnar and tibial nerves) and poor regenerative Table 5 - Functional outcome in relation to the type of interven- 15 tion and lesion categories. potential (peroneal nerve).

Lesion category and No. of lesions Significant type of injury outcome (> 3) The level of nerve injury in the lower limb did not Not in continuity 22 9 (41%) significantly affect the final outcome, as with upper limb • 1 yr suture repair 6 4 (75%) the lower the level of injury the better the outcome. The • 2 yrs suture repair 3 1(33%) outcome among nerve injuries in the buttock was (44%), • graft repair 13 4 (31%) (50%) in the thigh while it was (57%) in the knee. Other In continuity 21 14 (67%) 3,14,15,17 • Partial (neurolysis) 16 12 (75%) series agreed with these findings. Roganovic found • Complete (suture repair) 5 2 (50%) that the level of repair significantly affect the final outcome only for nerves with moderate regenerative potentials (tibial), while for nerves with excellent (radial) and poor recovery According to the mechanism of injury: potentials (peroneal nerve), difference in outcome after • Buttock-level sciatic nerve injuries: injection injury high, intermediate and low level repair was not significant.15 was the most common injury mechanism (6 patients) followed by hip fracture (3 patients). Only 6 nerve injuries underwent primary repair within 72 • Thigh-level sciatic nerve injuries: the mechanism of hours of injury with good outcome in 4 (66.6%), in injury was femoral shaft fracture (3 patients) and sharp comparison to 37 nerve injuries subjected to secondary laceration (3 patients). repair with significant outcome in 19 (51.3%). The defi- • Knee-level peroneal nerve injuries: stretch/contusion cient number of primary repair is explained by the fact that without fracture/dislocation (15 patients) followed by most of our patients reached us late after receiving primary laceration with sharp object (9 patients). aid treatment of associated vascular or tendon injuries in • Knee-level tibial nerve injuries: the mechanism of their localities. injury was contusion with fracture (2 patients) and contusion without fracture (2 patients). Based on their experience with military related peripheral nerve injuries, early reports by Platt and Woods(14) and by According to the type of surgical interventions Zachary and Holmes(20) recommended delayed repair, how- and lesion categories: Surgical results were generally ever in 1975, Seddon(18) suggested that early repair of tran- better for lesions in continuity than those not in continuity; sected nerves resulted in a higher proportion of satisfactory 9 (40%) of 22 repaired lesions that were not in continuity outcomes. regained grade 3 or better function whereas 14 (67%) of 21 lesions in continuity that were surgically treated regained The recommendation of early surgical repair was further grade 3 or better function. Functional recoveries of grade 3 supported by many authors.2,11 Bigos and Coleman demon- or better were seen in 7 (50%) of 14 nerves that received strated that excellent surgical outcome following early suture repair (epineureal anastmosis) and in 12 (75%) of 16 repair of nerve injuries due to sharp transection could be that were treated with neurolysis alone. Only 4 (30%) of 13 achieved in significant proportion of patients (52 of 56). In lesions that were treated with sural nerve graft showed instances in which surgical repair was delayed, post- satisfactory results (Table 5). operative recovery of constructed nerve was less favourable than in those with immediate repair.2 Discussion In our series, the age of the patients (5 - 53 years) did not Lacerating injuries proved to have a better prognosis in influence significantly the final outcome, this is met within comparison to traction injuries, in which neurolysis and most of published series.1,13 Demuynck reported that there graft are not very effective due to the long segment of is marked superior sensory recovery in children, however affected nerve. he concluded that the difference in the clinical results of adults and children was not reflected in the nerve conduc- Injection remains the most frequent cause of serious injury

48 PAN ARAB JOURNAL OF NEUROSURGERY PERIPHERAL NERVE INJURY IN LOWER EXTREMITIES • Kassem, et al to the buttock-level sciatic nerve, (66.6%) of the patients reinnervation in innervated muscles before clinical evidence with buttock-level injuries in this series were injured in this of functional recovery, but the extent of functional recovery manner. Patients with poor gluteal covering, such as consti- was not well predicted.3 tutionally thin or chronically ill and debilitated individuals, are predisposed to this type of injury. Infants and young Nerve injuries of lower limbs are characterized by a much children as well as elderly individuals are also very fre- longer time to recovery compared with upper limbs (mean quently afflicted.7 18 months for lower limbs vs. 5 months for upper limbs). This could be due to the longer reinnervation pathway of Intraneural injection appears to be the typical cause of muscles. sciatic nerve injury, resulting in an almost immediate onset of pain, paraesthesias, and/or deficit.13 A less frequent Conclusion pattern, which occurs in approximately 10% of cases, is a Functional outcomes of peripheral nerve injuries following delayed onset, ranging from minutes to hours, of pain, surgical repair are extremely encouraging in spite of poorer paresthesias, and/or deficit. This may be related to place- outcome than upper limb. ment of the injection either adjacent to the nerve or into the epineurium. The type of surgical repair (neurolysis, primary or second- ary suture or graft repair) depends on type of lesion (in As observed in the literature and reflected in this series, the continuity or not in continuity) and whether the lesion is majority of patients with injection injuries recovered function partial or complete, lesions that result in partial lesion in spontaneously.16 continuity have the most favourable outcome.

In our series we explore 6 patients with post injection injury Considering the rate of spontaneous recovery of post- after failure of conservative measures for 3 months. Only 1 injection nerve injuries of the sciatic nerve and early onset of 6 patients required excision of neuroma incontinuity and of skeletal deformities, a closed nerve injury of the lower repair. Unfortunately, there is currently no means within the limb with no recovery within 3 months should always early months after injury other than intraoperative evalua- undergo surgery, even if complete functional outcome is not tion to segregate the majority of patients who do not require always guaranteed excision of the lesion from the minority who do. We therefore continue to recommend surgical exploration in References those patients who do not exhibit spontaneous recovery on 1. Aldea PA, Shaw WA: Lower extremity nerve injuries. Clin clinical or electromyographic examination by 3 months Plast Surg 1986, 13(4): 691-699 from onset of the injection palsy. 2. Bigos SJ, Coleman SS: Foot deformities secondary to gluteal injections in infancy. J Pediatr Orthop 1984, 4(5): 560-3 3. Clawson DK, Seddon HJ: The late consequences of sciatic Outcome is much better for lesions in continuity (66.6%) nerve injury. J Bone Joint Surg Br 1960, 42B: 213-225 than for lesions not in continuity (40.9%) and to our 4. Cozen L: Management of foot drop in adults after permanent knowledge no published series argue this suggestion. peroneal nerve loss. Clin Orthop Relat Res 1969, 67: 151-8 Functional recoveries of grade 3 or better were seen in 5. Demuynck M, Zuker RM: The peroneal nerve: is repair worthwhile? J Reconstr Microsurg 1987, 3(3): 193-7 (50%) of nerves that received suture repair and (75%) of 6. Garozzo D, Ferraresi S, Buffatti P: Surgical treatment of nerves treated with neurolysis alone while only (30.7%) of common peroneal nerve injuries: Indications and results - A lesions that were treated with sural nerve graft showed series of 62 cases. J Neurosurg Sci 2004, 48(3): 105-112 functional recoveries, most probably because all of them 7. Gentili F, Hudson AR, Midha R: Peripheral nerve injuries: types, causes, and grading. In: Wilkins RH, Rengachary SS exceeded 7 cm length. (eds), Neurosurgery, 2nd Ed. New York, McGraw-Hill 1996, pp 3105-3114 Kim et al, stated that functional recovery after graft repair 8. Kim DH, Kline DG: Management and results of peroneal nerve was largely dependent on the length of the grafts (75%) lesions. Neurosurg 1996, 39(2): 312-9; Discussion 319-20 whose injuries were repaired with grafts < 6 cm in length 9. Kim DH, Murovic JA, Tiel RL, Kline DG: Management and outcomes in 318 operative common peroneal nerve lesions at recovering function to grade 3 or better and (35%) with the Louisiana State University Health Sciences Center. graft lengths of 6 - 12 cm. Good functional recovery could Neurosurg 2004, 54(6): 1421-8; Discussion 1428-9 not be expected with graft length > 12 cm.8 10. Kim DH, Murovic JA, Tiel R, Kline DG: Management and outcomes in 353 surgically treated sciatic nerve lesions. J Clinical assessment of recovery beyond the nerve injury site Neurosurg 2004, 101(1): 8-17 11. Kim DH, Ryu S, Tiel RL, Kline DG: Surgical management and was often difficult and involved a significant delay before results of 135 tibial nerve lesions at the Louisiana State any intrinsic muscular recovery could be detected. University Health Sciences Center. Neurosurg 2003, 53(5): 1114-24; Discussion 1124-5 We used EMG studies which often produced evidence of 12. Lundborg G: A 25-year perspective of peripheral nerve

VOLUME 14, NO. 2, OCTOBER 2010 49 PERIPHERAL NERVE INJURY IN LOWER EXTREMITIES • Kassem, et al

surgery: evolving neuroscientific concepts and clinical signifi- 17. Rosen B, Lundborg G: A new model instrument for outcome cance. J Hand Surg Am 2000, 25(3): 391-414 after nerve repair. J Hand Clin 2003, 19(3): 463-70 13. Mayer M, Romain O: Sciatic paralysis after buttock intra- 18. Seddon H (ed): Surgical disorders of the peripheral nerves, muscular injection in children: an outgoing risk factor. Arch 2nd Ed. Edinburgh, Churchill Livingstone 1975, pp 212-223 Pediatr 2001, 8: 321-23 19. Senes FM, Campus R, Becchetti F: Peripheral nerve injuries in 14. Platt H, Woods RS: Discussion on injuries of peripheral nerves. developmental age. In: Germann G (ed), Reconstructive Micro- Proc R Soc Med 1937, 30(7): 863-874 surgery (Proceedings of the II Congress of the World Society 15. Roganovic Z: Factor influencing the outcome of nerve repair. for Reconstructive Microsurgery). Bologna, Monduzzi Ed 2003, Vojnosanit Pregl 1998, 55(2): 119-31; Abstract pp 275-282 16. Rosen B, Lundborg G: The long term recovery curve in adults 20. Zachary RB, Holmes W: Primary sutures of nerves. Surg after median or ulnar nerve repair: a reference interval. J Hand Gynecol Obstet 1946, 82: 632-651 Surg Br 2001, 26(3): 196-200

50 PAN ARAB JOURNAL OF NEUROSURGERY Original Article

Percutaneous image guided lumbar disc nucleoplasty: A minimal invasive technique for lumbar disc decompression

Khaled Saeed Ebrahim, Amr AlShehaby, Mohamed A AlWardany, Ahmed Darwish, Mohamed Awad

Abstract Introduction: Nucleoplasty is a minimally invasive, percutaneous procedure that uses radiofrequency energy to ablate nuclear material and create small channels within the disc.

Aim of the study: To evaluate the efficacy of nucleoplasty technique in patients with leg pain caused by radicular encroachment.

Patients and methods: This study was performed on 29 patients (23 males and 6 females) with lumbar disc prolapse causing unilateral sciatica with or without lower back pain for duration more than 3 months with no response to conservative treatment (in the form of medications, bed rest, and physiotherapy) in the period from November 2006 to November 2008. The Perc-D Spine Wand with 1 mm diameter and bipolar tip was used for coblation and the coagulation on the disc utilizing both radiofrequency coblation technology and thermal technology using a radiofrequency Arthrocare® generator system 2000 (Arthrocare Corporation®, Sunnyvale, CA) to generate coblation and coagulation energy.

Results: The mean visual analogue score (VAS) for the treated patients preoperative was 8.3 and there was significant reduction in VAS in follow-up visits with the mean VAS = 3.4, 3.2, 2.5, 3.1, 3.5 at 1 week, 1 month, 3 months, 6 months, and 1 year duration respectively. All patients were satisfied with the procedure and the degree of pain relief at all follow-up visits.

Conclusion: Percutaneous image guided lumbar disc decompression using nucleoplasty technique seems to be an effective, safe, simple and minimal invasive procedure for relief of sciatica due to lumbar disc prolapse in well selected cases. Nonetheless a longer follow-up period and a larger number of patients is needed to assess the long-term efficacy of this procedure. (p51-55)

Key words: Lumbar disc, nucleoplasty, coblation and sciatica.

Introduction Low back pain is probably the most common condition low back pain associated with radicular syndromes.7 affecting the lumbar spine and is a leading cause for seeking professional medical assistance and absence from work. It Conventional open , is considered the standard is reported that approximately 80% of the population in the treatment for leg pain caused by radiculopathy from disc western countries will experience at least one episode of herniation. In recent years there has been a general trend in 12 low back pain in their lifetime and that 55% will suffer from spinal surgery toward reductionism and minimalization.

For this purpose, minimally invasive intradiscal techniques that provide a percutaneous approach to the disc have been Department of Neurosurgery developed. Percutaneous procedures are minimally inva- Ain Shams Faculty of Medicine Cairo sive, requiring only a short hospital stay. These techniques Egypt also eliminate the risks of postoperative scarring, linked to

Correspondence: surgery, which are often responsible for recurrence of pain. Dr. Khaled Saeed Ebrahim They can be repeated in the same patient without preclud- Department of Neurosurgery ing recourse to traditional surgery if they should fail.5 Ain Shams Faculty of Medicine Cairo Egypt Percutaneous techniques work by partial removal of the nu- Email: [email protected] cleus, decompressing herniated discs and relieving pressure

VOLUME 14, NO. 2, OCTOBER 2010 51 MINIMAL INVASIVE TECHNIQUE FOR LUMBAR DISC DECOMPRESSION • Ebrahim, et al on the nerve roots. In addition, they have been shown to 6. Spinal stenosis or spondylolithesis. reduce the intradiscal pressure. The interventional procedures 7. Coagulopathy. incorporating this approach are chemonucleolysis, auto- mated percutaneous lumbar discectomy, intradiscal laser discectomy, intradiscal electrothermal therapy, and most recently percutaneous nucleoplasty.4

What is nucleoplasty? Nucleoplasty is a minimally invasive procedure that uses radiofrequency energy to ablate nucleus pulposus tissue in a controlled manner. This leads to a reduction of pressure on compressed nerve roots, decreased intradiscal pressure. A patented coblation technology is applied through a Perc-D Spine Wand. Through the wand, one alternates power and Figure 1 - Example of prolapsed lumbar disc L4 (bulge is less voltage for two modes of actions: coblation at 125 V and than 6 mm and disc is contained). coagulation at 65 V. Coblation is a non-heat driven process, in which radiofrequency energy is applied to a conductive All 29 patients had undergone complete neurological medium (saline) to generate a highly focused plasma field examination and preoperative 0 - 10 visual analogue score around the electrode at the tip of the Perc-D Spine Wand.6 (VAS) was recorded.

This plasma field contains sufficient energy to cleave molec- Complete informed consent was signed by all the patients. ular bonds at low temperatures (40 - 70°C) into various elementary molecules and low molecular weight gases, e.g. Preoperative recent MRI lumbar spine and plain x-rays oxygen, nitrogen, hydrogen, and carbon dioxide. These were done for all patients included in the study. gases escape through the introducer needle. A series of six channels is created within the disc, removing a portion of Equipment the nucleus pulposus. Coagulation mode is then applied to The Perc-D Spine Wand with 1 mm diameter and bipolar thermally seal the channels. This further decompresses the tip was used for coblation and the coagulation on the disc intervertebral disc. Approximately 1 cc of nuclear tissue (or utilizing both radiofrequency coblation technology and 10% of the nucleus pulposus) is removed.6 thermal technology using a radiofrequency Arthrocare® generator system 2000 (Arthrocare Corporation®, Patients and methods Sunnyvale, CA) to generate coblation and coagulation This study was performed on 29 patients (23 males and 6 energy. females) with lumbar disc prolapse causing unilateral sciatica with or without lower back pain for duration of Technique more than 3 months with no response to conservative Patients were admitted on the same day of operation with treatment (in the form of medications, bed rest and NPO instructions for at least 6 hours prior to the procedure. physiotherapy) in the period from November 2006 to November 2008. Patient lies on the prone position on radiolucent operating table with IV line inserted and D5-normal saline infusion is Inclusion criteria: given. Oxygen nasal catheter and ECG leads are connected 1. Patient with unilateral sciatica with or without LBP for together with oxygen saturation monitoring as the patient more than 3 months duration not responsive to con- usually requires mild IV sedation and analgesia (e.g. IV servative treatment. fentanyl). The C-arm is brought into the operative field 2. Contained disc herniation shown on MRI. draped with sterile towels and the exact level of disc 3. Unilateral disc bulge not more than 6 mm in MRI (Fig. prolapse is identified in AP view (Fig. 2). 1). Sterilization and draping are done with betadine 5% in the Exclusion criteria usual manner and C-arm is brought to the proper oblique 1. Disc space narrowing more than 50%. position 30 - 35 degrees to visualize the needle entry zone at 2. Extruded or sequestrated disc. the Kambin’s triangle. 3. Disc bulge more than 6 mm. 4. Disc space infection. Using the same approach used for discography a Crawford 5. Spinal fracture or tumour. 17 G needle is advanced under x-ray guidance to penetrate

52 PAN ARAB JOURNAL OF NEUROSURGERY MINIMAL INVASIVE TECHNIQUE FOR LUMBAR DISC DECOMPRESSION • Ebrahim, et al the posterolateral annulus in the safe Kambin’s triangle to junction between the annulus and the nucleus pulposes avoid root injury (Figs. 3 and 4). (touching the vertical imaginary line crossing the inner border of the pedicles shadows).

The trocar is removed and the Perc-D Spine Wand is introduced to the needle and distal limit of the wand is marked under C-arm guidance as it touches the imaginary line crossing the contra lateral pedicles and the distal limit is marked by the stopper of the wand (Figs. 5a and b).

Figure 2 - The C-arm in AP position with patient lying prone after sterilization and draping. Figure 5a - Spine wand inside cannula approximately placed and distal limit is marked by stopper.

Figure 5b - Wand inside cannula in the AP view under image midway in the working channel.

Figure 3 - The safe Kambin’s triangle (axial view). Six channels are made in the substance of the nucleus at 2, 4, 6, 8, 10 and 12 o’clock directions as the wand is intro- duced slowly in the coblation mode (125 V) in about 10 seconds duration till it reaches the distal limit and is then retracted in the coagulation mode (65 V), in the same period of time.

The six channels made in the nucleus is sufficient to decrease the nucleus volume by 1 cc = 10% of the nucleus volume and coagulation further seals the bleeding points and contracts the surrounding tissues. Figure 4 - Cannula is introduced into the safe Kambin's tringle under C-arm guidance. Single injection of 3rd generation cephalosporins as prophy- lactic antibiotic is given and the patient is kept under observation for 2 - 3 hours till resolution of sedative effect Once the annulus is felt to be penetrated the C-arm is and then discharged with specific instructions for follow-up brought to the AP position and the needle is advanced to the and post procedure instructions.

VOLUME 14, NO. 2, OCTOBER 2010 53 MINIMAL INVASIVE TECHNIQUE FOR LUMBAR DISC DECOMPRESSION • Ebrahim, et al

Post procedure instructions All patients were satisfied with the procedure and the • Rest for 2 days with limited sitting and walking per degree of pain relief at all follow-up intervals. All stated day (to decrease the back soreness quickly after the they would do the procedure again, if needed. procedure, due to large bore needle puncture and muscle and annulus trauma). Discussion • No driving for at least 3 days. Basic scientific studies show that nucleoplasty can lead to a • Limit lifting of no more than 5 kgs, no bending and no safe percutaneous decompression of the disc. Disc histology twisting of the lower back. post nucleoplasty reveals that one can achieve safe volu- • Weeks number 2 and 3: physical activity is allowed, metric removal of nucleus pulposus.2 walking, swimming and driving for short distances, and return to work without severe exertion. The coblation channel shows clear coagulation border with- • Formal physiotherapy started 3 weeks postoperatively out necrosis of nucleus. There is no disruption or necrosis to (annulus is nearly healed completely) for back and the surrounding vital structures including annulus, endplate, abdominal muscles programmes and weight reduction spinal cord and nerve roots.2 if needed. • After discharge from physical therapy, simple home A thermal mapping study in percutaneous disc decompres- exercise programme to be performed on a daily basis. sion in the porcine model confirms a steep temperature drop off from the tip of Perc-D Spine Wand.1 Intradiscal pres- Follow-up visits to assess pain relief and brief neurological sure studies were performed in human cadaver spine examination for any complications, and also patient satis- segments and a significant drop of pressure was noted faction with the procedure are scheduled at 1 week, 1 following the creation of six channels.2 month, 3 months, 6 months and 1 year intervals. Visual analogue score is taken from patient’s own words and The first report of the clinical efficacy of nucleoplasty was plotted against the preoperative pain score. presented at the Florida Pain Society meeting in 2001. This study examined the safety and clinical benefit of the newest We only encountered 2 complications with this procedure, method of percutaneous disc decompression. The cohort with new onset of sensory symptoms in the form of brief was followed for 3 months following the procedure and and temporary paraesthesia in the dermatomal distribution with this group several conclusions were reached that were of the root affected by the treated disc. Fortunately, these confirmed in later, published studies with larger groups of paraesthesia resolved completely after 1 month. patients. No complications were noted from the procedure; Results improvements in VAS ratings, percentage of symptom relief and percentage of patients with 50% or greater symp- The mean VAS for the treated patients preoperatively was 11 8.3 and there was significant reduction in VAS in follow-up tom reduction were statistically and clinically significant. visits with the mean VAS = 3.4, 3.2, 2.5, 3.1, 3.5 at 1 week, 1 month, 3 months, 6 months and 1 year duration respect- In a clinical study on 45 patients who underwent percutane- tively (Fig. 6). ous disc decompression using nucleoplasty. Nine of the 45 patients had previous fusion, percutaneous discectomy or laminectomy. The mean age of the patients was 39 years. 9 A total of 45 patients were followed for 1 month, 33 for 3 8 months, 23 for 6 months, and 2 for 1 year. The author 7 showed a two-point reduction in VAS, satisfied patients, 6 and a reduction in narcotic usage. This author concludes 5 that an overall success rate of 78% was achieved. The 4 success rate for patients who had not undergone surgery 3 was 81%, for patients who had undergone previous surgery, 9 2 it was 67%. 1 0 Another nucleoplasty study was performed on 56 patients. h Forty-four of the patients were followed for more than 3 e nt tiv eek months. The mean age of the patients was 42 years. The raative w e 1 year p 1 1 mo o 3 months 6 months patients were divided up into three groups: back pain only, re Ppreo back and leg pain, and leg pain only. Forty-five percent of Figure 6 - The VAS values at preoperative and postoperative the patients were using narcotics at the time of the study. visits. Nineteen-percent of the patients were on worker’s compen-

54 PAN ARAB JOURNAL OF NEUROSURGERY MINIMAL INVASIVE TECHNIQUE FOR LUMBAR DISC DECOMPRESSION • Ebrahim, et al sation and 64% were not working. There was an improve- patients did not have any clinical resolution at 6 months, ment in the sitting and walking time over 3 months. A and 4 (8%) had no resolution at the latest follow-up.8 We greater percentage of patients were able to sit and walk for believe that the pessimistic results of Mirzai et al, are 60 - 120 minutes. The average pain relief was greater in directly related to patient selection which is the detrimental patients with leg pain compared with back pain. At the end factor contributing to the post procedure results. of the 3-month period, there was no major difference in the percentage of patients with greater than 50% pain relief Conclusion between the non-worker’s and the worker’s compensation Percutaneous image guided lumbar disc decompression group. No complications were noted in the patients who using nucleoplasty technique seems to be an effective, safe, had undergone nucleoplasty.11 simple, and minimal invasive procedure for relief of sciatica due to lumbar disc prolapse in well selected cases. Yet Chen et al, performed a similar study. Thirty patients (19 longer follow-up and larger number of patients is needed to males, 11 females) underwent nucleoplasty and were assess the long term efficacy of this procedure. followed for 6 months. The mean age was 37.6 years with a range between 22 and 56 years. The patients were divided References in three groups: axial discogenic low back pain, axial 1. Chen Y, Lee S, Chen D: Experimental thermomapping study, discogenic low back pain with radicular symptoms, and in percutaneous disc decompression, nucleoplasty. North pure radiculopathy without neurological deficits. Twenty- American Spine Society/South American Spine Society Meeting of the Americas. New York 2001 three patients were treated at 1 level, 4 at 2 levels, and 3 at 3 2. Chen Y, Lee S, Chen D: Histology of disc, endplate and neural levels. Overall, the mean pain VAS reduction was 3.14. elements post coblation of nucleus: An experimental nucleo- Sixty-nine-percent of the patients had total resolution of leg plasty study. North American Spine Society/South American pain. Eighty-six percent did not require narcotics after 6 Spine Society Meeting of the Americas. New York 2002 3. Chen Y, Lee S, Lau E: Percutaneous disc decompression. months. Eighty-nine-percent of the patients were satisfied Nucleoplasty for chronic discogenic pain with or without with their results after 6 months. No complications were sciatica. North American Spine Society/South American Spine noted in patients who had undergone nucleoplasty.3 Society Meeting of the Americas. New York 2002 4. Erdine S, Ozyalcin NS, Cimen A: Percutaneous lumber nucleoplasty. Agri 2005, 17(2): 17-22 Another clinical study conducted by Welch and Gerszten on 5. Gangi A, Dietemann JL, Mortazavi R, Pfleger D, Kauff C, Roy 25 patients with lower back pain and radiculopathy yielded C: CT-guided interventional procedures for pain management very good to excellent results with patients with diffuse disc in the lumbosacral spine. RadioGraphics 1998, 18(3): 621-33 protrusion and radicular symptoms and they stated that pain 6. Kim PS: Nucleoplasty. Tech Reg Anes Pain Manage 2004, 8 (1): 46-52 relief had occurred almost immediately after the proce- 7. Long MD: Decision making in lumbar disc disease. Clin 12 dure. Neurosurg 1991, 39: 36-51 8. Mirzai H, Tekin I, Yaman O, Bursali A: The results of nucleo- In the most recent study conducted by Mirzai et al, which plasty in patients with lumbar herniated disc: A prospective clinical study of 52 consecutive patients. Spine J 2007, 7(1): included 52 patients, 34 had one disc treated and 18 had 88-92; Discussion 92-93 two discs treated. Of the total 70 discs treated, 41 were at 9. Sharps LS, Isaac Z: Percutaneous disc decompression using L4 - L5, 17 were at L5 - S1, and 12 were at L3 - L4. All nucleoplasty. Pain Physician 2002, 5(2): 121-126 procedures were considered technically successful, with the 10. Singh V, Piryani C, Liao K, Nieschulz S: Percutaneous disc decompression using coblation (nucleoplasty) in the treatment full treatment protocol carried out to completion. There of chronic discogenic pain. Pain Physician 2002, 5(3): 250- were no complications associated with the procedure during 259 the follow-up periods. The mean follow-up period was 11. Singh V: Percutaneous disc decompression using nucleo- 12.1 months (10 -15 months). Three patients were lost to plasty. Annual Meeting of Florida Pain Society, Miami, Florida 2001 follow-up after the 6-month examination. There was 12. Welch WC, Gerszten PC: Alternative strategies for lumbar dis- complete resolution of symptoms in 40 (77%) cases at 6 cectomy: Intradiscal electrothermy and nucleoplasty. Neurosurg months and in 41 (84%) at the latest follow-up. Eight (15%) Focus 2002, 13(2): Article 7

VOLUME 14, NO. 2, OCTOBER 2010 55 Clinical Study

Microsurgical excision of anterior and anterolateral intradural lesions of the foramen magnum

Hisham Aboul-Enein

Abstract: The most important factors that influence the surgical outcome in foramen magnum lesions are compre- hensive and three-dimensional understanding of the specific anatomy of the region, tailoring the exposure of the underlying lesion, maintaining a nearly bloodless surgical field intradurally, and applying an adequate microsurgical technique.

Objective: This study describes in detail the far lateral approach for intradural tumours in the craniocervical region, with special emphasis placed on the management of the vertebral artery and on the anatomic variations encountered in this region.

Material and methods: The study included 31 patients with different pathologies at the lateral foramen magnum. All were operated using the far lateral approach to access the lateral angle of the foramen magnum after mobilizing the vertebral artery.

Results: Total resection was achieved in 17 cases whereas subtotal resection with superadded foramen magnum decompression was done for 14 cases. In the study there have been no mortalities, yet 3 cases of CSF leakage were encountered, and another patient suffered from intradural vertebral artery injury.

Conclusions: Far lateral approach can provide a good working angle to the anterior of the brainstem without extra drilling of the occipital condyle. The aim of surgery at the foramen magnum should be directed at brainstem decom- pression rather than unsafe total excision of the tumour. The more the vertebral artery is well prepared and mobilized extradurally the less the incidence of its intradural injury. (56-62)

Key words: Foramen magnum, vertebral artery, transcondylar and brainstem.

Introduction Surgery of intradural lesions at the craniocervical junction Material and methods requires thorough knowledge of the regional anatomy within The present study included 31 patients suffering from the foramen magnum.20,21 Lateral approaches are used to various intradural pathologies at the craniocervical junction. reach lesions anterior and anterolateral to the medulla The present study was conducted starting from January oblongata. The primary goal of foramen magnum surgery is 2002 to July 2008. All patients were symptomatic, showing to decompress the vital neural structures without compro- a wide range of complaints and neurological deficits. mising the neurological function or the atlantooccipital stability. This goal implies that radical excision of tumour is Diagnosis No. patients 9 not always considered the best solution. Meningioma 15 Lower cranial nerve schwanoma 6 Brainstem glioma 8 Cavernous angioma 1 PICA aneurysm 1

Department of Neurosurgery Faculty of Medicine Alexandria Surgical technique: Egypt Positioning and skin incision: All patients were oper- ated in the lateral park bench position. The patient's head Correspondence: Dr. Hisham Aboul-Enein was slightly flexed, rotated 45 degrees to the ipsilateral side, Department of Neurosurgery and tilted to the contralateral side to obtain a wide angle of Alexandria Medical School Alexandria vision toward the anterolateral craniovertebral junction area. Egypt The head and upper body was elevated to reduce epidural Email: [email protected] venous bleeding. A longitudinal skin incision with slightly

56 PAN ARAB JOURNAL OF NEUROSURGERY MICROSURGICAL EXCISION OF LESIONS AT THE FORAMEN MAGNUM • Abdoul-Enein curved ends was preferred in the present study, allowing a the VA is first identified ventral to the C2 nerve in the C1 direct exposure of the lateral craniocervical junction and its C2 inter-transverse space. Then the V3 segment is identified important anatomical bony and vascular landmarks (Fig. 1). within the sulcus arteriosus after detaching the small muscles and subperiostal retraction of these muscles from medial to Vertebral artery mobilization: The vertebral artery (VA) lateral. The artery can then be visualized so that the course is a key structure and important anatomic landmark. The of the artery becomes clearly visible up to the point where atlantal arch is first identified by palpating the posterior the artery pierces the atlantooccipital membrane and the tubercle, posterior arch and transverse process of C1 within dura mater (Fig. 3). the deep muscular triangle (Fig. 2). The vertical portion of

Figure 1a - Skin incision used during far lateral approach: curvilinear skin lying midway between the mastoid process and the midline. The incision starts about 5 cms above the anatomical landmark of the transverse sinus till the level of the fourth cervical spine. b) The patient's head was slightly flexed, rotated 45 degrees to the ipsi- lateral side, and tilted to the contralateral side to obtain a wide angle of vision toward the dorsolateral cranioverte- bral junction area.

Figure 2a - Cadaver illustration of the right sub occipital triangle. b) Showing the vertebral artery in the sulcus arte- riosus where it could be easily palpated.

Figure 3a - Showing the vertical part of vertebral artery with the surround- ing venous plexuses. b) Showing the vertebral artery as it enters the dura.

VOLUME 14, NO. 2, OCTOBER 2010 57 MICROSURGICAL EXCISION OF LESIONS AT THE FORAMEN MAGNUM • Abdoul-Enein

Bony removal: Suboccipital craniectomy was done with meningiomas, all were operated upon using the far lateral C1 hemilaminectomy to expose the whole anatomic fora- approach. Male to female ratio was 2:3, the age ranged men magnum. A lateral suboccipital craniectomy was done from 26 to 64 years, with mean age of 45.6. to all patients: the craniectomy extended superiorly to the level of the transverse sinus and lateral to the sigmoid sinus allowing superior retraction of the cerebellum to visualize the rootlets of cranial nerves IX through XI. C1 hemi- laminectomy was done to expose the dural entry of the vertebral artery. A small portion of the occipital condyle was drilled not exceeding one third of the condylar fossa. The part of the condyle removed gave access to antero- lateral meningiomas (Fig. 4).

Figure 5a - Showing the dural opening, the dura mater could be reflected laterally together with the proximal intradural VA and was sutured to the surrounding muscles. A brain retractor is used to lift the cerebellum superiorly thus exposing the whole foramen magnum. b) Cadaver illustration after opening Figure 4a - MRI of an 18-year-old female patient axial view showing the dura exposing the entry of the vertebral artery. a lower cranial nerve schwanoma. b) Axial postoperative CT scan showing total removal and amount of bone removed from the occipital condyle. c) Intraoperative photo showing the extent of bone removed, a small suboccipital craniectomy allowing superior retraction of the cerebellum together with a C1 hemilaminectomy.

Dural opening: The dura mater was opened longitu- dinally at the C1 level and in a Y shape at the level of the cerebellum. The dura mater was reflected laterally together with the proximal intradural VA and was sutured to the surrounding muscles. A wide opening of the surgical field medially and laterally to the dural entrance of the VA Figure 6a - MRI coronal view of 34-year-old female patient became available (Figs. 5 and 6). showing a right laterally based foreman magnum meningioma. b) Postoperative CT scan showing total excision and formation of a pseudo-meningocele with subsequent CSF leakage. Results Meningiomas The present study included 15 cases of foramen magnum As shown in Table 1, the vertebral artery was encased in all

58 PAN ARAB JOURNAL OF NEUROSURGERY MICROSURGICAL EXCISION OF LESIONS AT THE FORAMEN MAGNUM • Abdoul-Enein patients but with different degrees. The vertebral artery was the brainstem which necessitated more extradural bony partially encased in 12 cases, and showed a good line of work that was not achieved in this study (Table 3). cleavage between it and the tumour. These were the cases in which total excision was achieved. Yet in the remaining 3 Table 3 - Extent of removal of foramen magnum meningioma cases the tumour was located more anterior to the brainstem using the far lateral approach in the present study. with bilateral encasement of the vertebral arteries, thus Extent of removal Total Subtotal Partial rendering safe total excision impossible through this No. of patients 12 2 1 approach. Only subtotal excision was done for these 3 patients, and subsequent foramen magnum decompressions. Accessibility of approach; it is observed that for lateral and anterolateral based foramen magnum meningiomas the far Table 1 - Degree of encasement of the vertebral artery in men- ingiomas. lateral approach targeted the main goals of surgery. Yet, with for more anteriorly based meningioma involving the Degree of VA encasement Partial Complete Bilateral VA bilaterally the angle of vision was obscured and limited. No. of patients 12 2 1 Lower cranial nerve schwanoma Partial involvement of the vertebral artery was observed in The study included 6 cases of foramen magnum schwano- 12 patients, this made its dissection from tumour much mas. The male to female ratio was 1:3. The age ranged easier than in the rest of patients where in 2 cases the between 18 and 56 years. In 4 cases the origin of the tumour vertebral artery and the PICA were both embedded in the was lower cranial nerve, while 2 cases had a C1 neuro- tumour tissue. One patient had both VA embedded in the fibroma. All cases were accessible and totally removed tumour which would necessitate more extra-dural bony through the far lateral approach. In only one case with a work that was achieved in the present study. recurrent C1 neurofibroma the VA was injured intradurally, and the patient suffered from hemiplegia and lateral me- dullary syndrome. Dural attachment, as shown in Table 2; 8 patients had an anterolateral meningioma, whereas 6 patients had a laterally Intra-axial tumours based tumour, and only one patient showed a totally The study included 8 brainstem lesions, one of which was a anterior based meningioma. The more laterally placed the caveroma and showed itself through the external surface of meningioma and the more lateral its dural attachment was, the brainstem by its typical xanthochromic colouration. The the easier it is to devascularise the tumour extradurally and remaining patients were young adults with exophytic hindering the field less bloody and achieving total excision. brainstem glioma. The lesions were accessible through the

same approach, and subtotal tumour removal was achieved As regards tumour consistency; in only 2 cases the tumour safely. was soft, suckable and easy to remove from its dural base, whereas in the remaining 13 cases the tumour was tough The study included a laterally placed brainstem cavernoma and encasing the vertebral artery with a wide firm attach- which showed itself on the surface of the brainstem after the ment to the dura. approach was completed. This patient had several attacks of bleeding before being operated upon. This event created a Table 2 - Site and extend of dural attachment of foramen mag- cavity inside the brainstem and rendered the entry zone num meningiomas. safer inspite of the lack of evoked potentials in our depart- Site of dural attachment Anterior Lateral Anterolateral ment. No. of patients 1 6 8 Complications of approach As seen in Table 4, the far lateral approach to the foramen Anterior placed meningioma was only found in one patient magnum in the present study showed no mortalities, while in whom partial resection was done due to involvement of morbidity was 5 patients. Cerbrospinal fluid leakage was both VA in the tumour. found in 3 patients and this was managed through daily lumbar taps for three consecutive days, CSF leakage The extent of tumour removal depended on the consistency stopped in 2 patients, yet one patient needed a second dural of the tumour and adherence to the VA. Total tumour re- repair. As mentioned above one patient suffered injury of moval was achieved in 12 cases. Subtotal removal leaving a the main trunk of the VA. This patient needed a longer stay sheet of the tumour on the VA was achieved in 2 cases in the ICU and suffered from a dense hemipaeresis and while in one case the tumour was located entirely anterior to lower cranial nerve affection and aspiration. The approach

VOLUME 14, NO. 2, OCTOBER 2010 59 MICROSURGICAL EXCISION OF LESIONS AT THE FORAMEN MAGNUM • Abdoul-Enein was proved to be limited when the tumour was entirely There is continuing controversy in the literature concerning anterior to the medulla. These tumours need further drilling the necessity of resecting the occipital condyle large lateral of the occipital condyle and jugular tubercle to gain a more and anterolateral tumours could be exposed easily without anterior view. extensively drilling the condyle.5,7,19,24 A different situation arises in anteriorly placed meningiomas or tumour portions Table 4 - Complications of far lateral approach experienced in located near the midline encasing the contralateral VA. In this series. order to visualize such lesions from a lateral direction it is Complication No. of patients required not only to resect the medial rim of the foramen magnum which corresponds to the medial portion of the CSF leakage 3 condyle but also of the jugular tubercle and, depending on VA injury 1 the caudal extension of the tumour, of the medial portion of Inadequate exposure 1 the lateral mass of the atlas.2,7,27

Discussion It was found that most meningiomas included in the present The surgical aspects to the foramen magnum have a unique study were tough in consistency and adherent to their dural consideration, due to its important neurovascular anatomy. attachment. The main target of surgical removal of menin- Different intradural pathologies may present at the cranio- giomas is attacking the vascular supply of the tumour first cervical junction. Various surgical approaches have been and this could only be achieved by disinsertion of the described to reach the anterolateral portion of the foramen tumour from its dural attachment. Meningiomas have magnum. The posterolateral transcondylar approach provides variable consistency and vascularity. Some tumours are a good viewing access ventral to the brainstem and lower fragile, of soft consistency but containing an abundant network of pathologic and fragile vessels. Such tumours clivus. In addition, the far lateral approach does not affect 3,23 the craniocervical stability when done with minimal removal cannot be cauterized adequately in their periphery. An of the occipital condyle.20,26 early devascularization of such tumours proved to be the best strategy, and this was possible only at the site of tumour insertion to the dura, requiring a so-called extreme The far lateral approach provides an adequate exposure and 7,11,33 viewing angle to the ventral brainstem with minimal retrac- lateral exposure. tion of important neurovascular structures in the region. The viewing angle is satisfactory even without mobilisation of The foramen magnum region concentrates many important the VA and its branches could be safely managed without neurovascular structures within a narrow field. Medulla and jeopardizing the neurological status of the patient.17-19,22,32 spinal cord, rootlets of the lower cranial and upper spinal nerves, VA, PICA, and small perforating branches of these The far lateral approach is an extension of the standard arteries could not be seen in all instances at the beginning of suboccipital approach, designed to maximize exposure of surgery. Often these vital structures were hidden behind or the anterolateral craniocervical junction.18,24,34,38 inside a large tumour. When surgery of the tumour is performed in a fashion such that long-lasting bleeding, Bone removal involves C1 hemilaminectomy, a sub-occipi- either from the tumour itself or from the extradural space, tal craniotomy and removing the posterior portion of the constantly obscures the surgical field, the procedure occipital condyle. Mobilization of the vertebral artery is not becomes hazardous because the aforementioned anatomic a mandatory step and only done if needed whenever the structures cannot be clearly identified.1,4,8,10,12 lesion is entirely anterior to the brainstem.18,24 All patients included in this study were followed up for a George et al, classified foramen magnum meningiomas period ranging between 6 months to 6 years. None of the according to their zone of insertion and its relation to the patients showed any signs of instability of the cranio- midline and denticulate ligament into anterior, lateral, and cervical junction. Several reports in literature have assessed posterior lesions.18 It is very important to define the exact the stability of the craniocervical junction both preopera- site and extent of the dural attachment of foramen magnum tively and postoperatively.11,16,28,31,36,39 These reports came to meningiomas, and the degree of encasement of the VA.7 the conclusion that the far lateral approach can offer a good These are the main factors regarding the extent of removal avenue to the anterolateral portion of the foramen magnum and the outcome. In the present work it is found that the far without jeopardizing the craniocervical stability. Most lateral approach offers a good working angle for lateral and authors agree that far lateral approach could be carried out antero-lateral meningiomas. Yet, with an entirely anterior without the need of occipito-cervical stabilization.6,25,27,29,30 meningioma this approach needs more extensive extradural bony work and to access the anterior foramen magnum dura. The amount of occipital condyle removed is the main factor

60 PAN ARAB JOURNAL OF NEUROSURGERY MICROSURGICAL EXCISION OF LESIONS AT THE FORAMEN MAGNUM • Abdoul-Enein

for doing craniocervical stabilization at the end of surgery. Cranial Base Surgery. London, Churchill Livingstone 2000, pp The more the surgeon has to remove from the occipital 237-258 condyle the more the need for stabilization. The lateral 14. Bozbuga M, Ozturk A, Bayraktar B, et al: Surgical anatomy and morphometric analysis of the occipital condyles and fora- approach of the foramen magnum offers a good surgical men magnum. Okajimas Folia Anat Jpn 1999, 75(6): 329-334 trajectory to the ventral brainstem and upper cervical cord. 15. de Oliveira E, Rhoton AL Jr, Peace D: Microsurgical anatomy This approach can be modified whenever needed to reach of the region of the foramen magnum. Surg Neurol 1985, 24 lesions reaching the jugular tubercle or entirely anterior (3): 293-352 8,13,15,19 16. Dowd GC, Zeiller S, Awasthi D: Far lateral transcondylar ap- placed lesions. proach: Dimensional anatomy. Neurosurg 1999, 45(1): 95-99 17. George B, DematonS C, Cophignon J: Lateral approach to the Conclusions anterior portion of the foramen magnum. Application to surgical removal of 14 benign tumors: Technical note. Surg Neurol The far lateral approach could be done with great safety to 1988, 29(6): 484-490 reach antero-lateral foramen magnum lesions. Anteriorly 18. George B, Lot G: Anterolateral and posterolateral approaches based meningiomas require more drilling of the occipital to the foramen magnum: Technical description and experience condyle. The VA is a keystone during this approach and from 97 cases. Skull Base Surg 1995, 5(1): 9-19 19. George B, Lot G, Boissonnet H: Meningioma of the foramen care must be given to preserve its main trunk and the magnum: A series of 40 cases. Surg Neurol 1997, 47(4): 371- branches. Removal of the posterior portion of the occipital 379 condyle does not affect the craniocervical stability. 20. George B, Lot G: Foramen magnum meningiomas: A review from personal experience of 37 cases and from a cooperative study of 106 cases. Neurosurg Quart 1995, 5(3):149-167 References 21. George B, Lot G: Surgical approaches to the foramen 1. Acikbas SC, Tuncer R, Demirez I, et al: The effect of magnum. In: Robertson JT, Coakham HB, Robertson JH condylectomy on extreme lateral transcondylar approach to (eds), Cranial Base Surgery. London, Churchill Livingstone the anterior foramen magnum. Acta Neurochir (Wien) 1997, 2000, pp 259-277 139(6): 546-550 22. George B, Lot G, Velut S, et al: Tumors of the foramen 2. AI-Mefty O, Borba LAB, Aoki N, et al: The transcondylar magnum. Neurochirurgie 1993, 39(Suppl 1): 1-89 approach to extradural nonneoplastic lesions of the cranio- 23. Goel A, Desai K, Muzumdar D: Surgery on anterior foramen vertebral junction. J Neurosurg 1996, 84(1): 1-6 magnum meningiomas using a conventional posterior suboc- 3. Arnautovic KI, AI-Mefty O, Husain M: Ventral foramen magnum cipital approach: A report on an experience with 17 cases. meningiomas. J Neurosurg 2000, 92: 71-80 Neurosurg 2001, 49(1): 102-106 4. Banerji D, Behari S, Jain VK, et al: Extreme lateral trans- 24. Karam YR, Menezes AH, Traynelis VC: Posterolateral condylar approach to the skull base. Neurol India 1999, 47(1): approaches to the craniovertebral junction. Neurosurg 2010, 22-31 66(3 Suppl): 135-40 5. Babu RP, Sekhar LN, Wright DC: Extreme lateral trans- 25. Katsuta T, Matsushima T, Wen HT, Rhoton AL Jr: Trajectory of condylar approach: Technical improvements and lessons the hypoglossal nerve in the hypoglossal canal: Significance learned. J Neurosurg 1994, 81(1): 49-59 for the transcondylar approach. Neurol Med Chir (Tokyo) 2000, 6. Bejjani GK, Sekhar LN, Riedel CJ: Occipitocervical fusion 40: 206-209 following the extreme lateral transcondylar approach. Surg 26. Kawase T, Bertalanffy H, Otani M, et al: Surgical approaches Neuro 2000, 154(2): 109-116 for vertebro-basilar trunk aneurysms located in the midline. 7. Bertalanffy H, Benes L, Becker R, et al. Surgery of intradural Acta Neurochir (Wien) 1996, 138(4): 402-410 tumors at the foramen magnum level. Operative Techniques in 27. Lanzino G, Paolini S, Spetzler RF: Far-lateral approach to the Neurosurgery 2002, 5(1): 11-24 craniocervical junction. Neurosurg 2005, 57(4 Suppl): 367-71; 8. Bertalanffy H, Gilsbach JM, Mayfrank L, et al: Microsurgical Discussion 367-71 management of ventral and ventrolateral foramen magnum 28. Matsushima T, Natori Y, Katsuta T, et al: Microsurgical anatomy meningiomas. Acta Neurochir Suppl (Wien) 1996, 65: 82-85 for lateral approaches to the foramen magnum with special 9. Bertalanffy H, Gilsbach JM, Mayfrank L, et al: Planning and reference to transcondylar fossa (supracondylar transjugular surgical strategies for early management of vertebral artery tubercle) approach. Skull Base Surg 1998, 8(3): 119-125 and vertebrobasilar junction aneurysms. Acta Neurochir (Wien) 29. Menzes AH: Surgical approaches: postoperative care and 1995, 134: 60-65 complications "posterolateral-far lateral transcondylar ap- 10. Bertalanffy H, Gilsbach J, Seeger W, Toya S: Surgical anatomy proach to the ventral foramen magnum and upper cervical and clinical application of the transcondylar approach to the spinal canal". Childs Nerv Syst 2008, 24(10): 1203-7 lower clivus. In: Samii M (ed), Skull Base Surgery. First 30. Nanda A, Vincent DA, Vannemreddy PSSV, Baskaya MK, International Skull Base Congress, Hannover 1992. Basel, Chanda A: Far-lateral approach to intradural lesions of the Karger 1994, pp 1045-1048 foramen magnum without resection of the occipital condyle. J 11. Bertalanffy H, Kawase T, Seeger W, et al: Microsurgical Neurosurg 2002, 96(2): 302-9 anatomy of the transcondylar approach to the lower clivus and 31. Rhoton AL Jr: The far-lateral approach and its transcondylar, anterior craniocervical junction. In: Gibo H (ed), Surgical supracondylar, and paracondylar extensions. Neurosurg 2000, Anatomy for Microsurgery V. Tokyo, Sci Med Publications 47(3 Suppl): 195-209 1992, pp 167-175 32. Salas E, Sekhar LN, Ziyal IM, et al: Variations of the extreme- 12. Bertalanffy H, Seeger W: The dorsolateral, suboccipital, lateral craniocervical approach: Anatomical study and clinical transcondylar approach to the lower clivus and anterior portion analysis of 69 patients. J Neurosurg 1999 90(2 Suppl): 206-219 of the craniocervical junction. Neurosurg 1991, 29(6): 815-821 33. Samii M, Klekamp J, Carvalho J: Surgical results for 13. Bertalanffy H, Sure U: Surgical approaches to the jugular meningiomas of the craniocervical junction. Neurosurg 1996, foramen. In: Robertson JT, Coakham HB, Robertson JH (eds), 39(6): 1086-95

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34. Sen CN, Sekhar LN: An extreme lateral approach to intradural infratentorial approach for lesions of the petrous and clival lesions of the cervical spine and foramen magnum. Neurosurg regions: Experience with 46 cases. J Neurosurg 1992, 76(4): 1990, 27(2): 197-204 588-599 35. Sen CN, Sekhar LN: Surgical management of anterior placed 38. Wanebo JE, Chicoine MR: Quantitative analysis of the lesions at the craniocervical junction--an alternative approach. transcondylar approach to the foramen magnum. Neurosurg Acta Neurochir (Wien) 1991 (108): 70-77 2001, 49(4): 934-943 36. Spektor S, Anderson GJ, McMenomey SO, et al: Quantitative 39. Wen HT, Rhoton AL Jr, Katsuta T, de Oliveira E: Microsurgical description of the far-lateral transcondylar transtubercular anatomy of the transcondylar, supracondylar and paracondylar approach to the foramen magnum and clivus. J Neurosurg extensions of the far-lateral approach. J Neurosurg 1997, 87 2000, 92(5): 824-831 (4): 555-585 37. Spetzler RF, Daspit CP, Pappas CT: The combined supra- and

62 PAN ARAB JOURNAL OF NEUROSURGERY Clinical Study

Percutaneous vertebroplasty for osteoporotic fracture of dorsolumbar and lumbar vertebra: Surgical technique and early outcome

Ahmed Yehya, Abdelaziz El-Nekady

Abstract Introduction: Vertebroplasty is an image-guided, minimally invasive, nonsurgical procedure used to strengthen fractured spinal vertebrae that has been weakened by osteoporosis. A hollow needle (trocar) is passed into the fractured vertebral body and a cement mixture including polymethylmethacrylate (PMMA), barium powder and a solvent is injected. The cement mixture resembles toothpaste or epoxy. We monitor the entire procedure on a fluoroscopy imaging screen and make sure that the cement mixture does not enter into the spinal canal.

Vertebroplasty is highly effective because the cement fills the spaces and strengthens the bone; it is less likely to fracture again. After vertebroplasty, the cement stabilizes the fracture, and also provides pain relief. Patients regain mobility within 24 hours and are usually able to reduce, or even eliminate, their pain medications within a short time.

Vertebroplasty can increase the patient's functional abilities, allow a return to the previous level of activity, and prevent further vertebral collapse.

Aim of the work: The aim of the present work is to study the procedure of percutaneous vertebroplasty in regard to the surgical technique and the early outcome. If it is effective in relieving the pain caused by osteoporotic vertebral compression fracture of dorsolumbar and lumbar vertebra and also evaluate any complications that may occur during the procedure.

Material and methods: The study was conducted on patients admitted to the main Alexandria University Hospital from January 2006 to 2008. We did plain x-ray lumbosacral spine AP and lateral view preoperative and postoperative for all the patients, MRI or CT LSS preoperative and CT LSS postoperative. (p63-68)

Key words: Percutaneous vertebroplasty, vertebral compression fractures and minimal invasive.

Introduction Percutaneous vertebroplasty is a minimally invasive, outpatient tating, causing limited mobility and significant reduction in procedure used to treat the pain associated with vertebral quality of life.1,20,41 compression fractures (VCFs), caused by osteoporosis and tumoural lesions such as metastasis, myeloma, haeman- Treatment of VCFs was mainly in the form of external gioma, etc. The osteoporotic vertebra, weakened by the support, bed rest and potent analgesics. However, these disease can collapse suddenly under the force of normal conservative measures were ineffective in all cases and daily activity, it results in severe pain, that can be debili- severe pain could persist and needs to be treated with vertebroplasty.13,14,22,33

Percutaneous vertebroplasty (PVP) was first reported by Department of Neurosurgery Faculty of Medicine Galibert et al, in 1984 and initially involved the augmenta- Alexandria University tion of the vertebral body with polymethylmethacrylate Alexandria (PMMA) to treat a case of haemangioma.4,11,19 Percuta- Egypt neous vertebroplasty resulted in early appreciable pain relief Correspondence: and low complication rates. The mechanism of pain relief Dr. Ahmed Yehya Department of Neurosurgery is still unknown but it may be due to stabilization of the Faculty of Medicine fractured bone fragments by PMMA or by the analgesic Alexandria University Alexandria result from local chemical, vascular, or thermal effect of Egypt PMMA on nerve endings of surrounding tissues. This can Email: [email protected] be supported by lack of the concentration of cement and the

VOLUME 14, NO. 2, OCTOBER 2010 63 PERCUTANEOUS VERTEBROPLASTY FOR OSTEOPOROTIC FRACTURE • Yehya & El-Nekady degree of pain relief.2,4,15,18 form of dry powder polymer mixed with barium and tungsten to make it radio-opaque. This powder was mixed Materials and methods with liquid monomer of methylmethacrylate to a consis- The study was conducted on 16 patients with 24 osteo- tency similar to that of toothpaste. porotic VCFs with failed conservative treatment. The mean age was 66 years, range from 55 - 78 years; 10 were The opacified PMMA was injected percutaneously with females and 6 were males. The selection criteria of the high pressure torque handle syringe via a transpedicular patients include osteoporotic fracture less than 12-months- approach through a trocar to obtain adequate filling of the old, with no response to conservative treatment, with severe osteoporotic vertebra with care to avoid PMMA leakage pain and localized tenderness over the affected level. (Figs. 3, 4 and 5).

The contraindications of PVP include presence of spinal cord compression, or retropulsed fragment at the affected level, also if the fracture extended to the posterior vertebral cortex, absence of facilities to perform emergency decom- pressive surgery in the event of complications; coagulo- pathy, sepsis and extreme vertebral collapse more than 70% reduction in vertebral height.

Vertebral compression fractures were documented by clinical and radiological results in the form of plain x-ray Figure 3 - Lateral view shows intraoperative fluoroscopy after (AP and lateral films) MRI and CT of the affected levels. injection of the bone cement with the 2 needles still inside in Those patients were treated with PVP using the standard the same patient. operative techniques, under local anaesthesia with mild sedation. The patient lies in prone position, a targeting trocar directed into the appropriate pedicles under continu- ous biplanar fluoroscopic guidance (Figs. 1 and 2).

Figure 1 - AP view shows intraoperative fluoroscopy of trans- pedicular insertion of 2 needles inside osteoporotic fractures at L1 - L2 vertebra in 65-year-old female.

Figure 2 - Lateral view shows intraoperative fluoroscopy of transpedicular insertion of 2 needles in the same patient. Figure 4 - Plain x-ray, AP, and lateral view showing osteo- porotic fractures of L1 with lateral view post injection of the The cement was prepared under sterile conditions in the bone cement in 73-year-old female.

64 PAN ARAB JOURNAL OF NEUROSURGERY PERCUTANEOUS VERTEBROPLASTY FOR OSTEOPOROTIC FRACTURE • Yehya & El-Nekady

Figure 5 - CT lumbar spine showing osteoporotic fractures of L1 post injection of the bone cement.

Preceding PMMA injection, intraosseous venography was Figure 7 - X-ray lumbar spine AP and lateral views showing often used to determine the filling pattern, identify the sites post injection of the bone cement of the same patient. of potential PMMA leakage, outline the venous drainage pattern, and confirm the needle placement within the bony trabeculae and to delineate fractures in the bony cortex. The patients were followed-up for a minimum of 6 months. Plain x-ray (AP and lateral films) routinely performed after If opacification of the paraspinous veins was seen early 1 and 3 months post injection, and also CT on the affected while injecting the cement the injection was suspended for level was performed after 1 month following procedure. approximately 1 minute to allow the cement to harden within the vein then injection was resumed. Results Sixteen patients were operated with 24 osteoporotic VCFs, When the cement reaches both the superior and inferior documented by clinical and radiological findings. Ten endplates of the injected osteoporotic vertebra and extends patients were female and 6 patients were male, age range across the midline, this meant that approximately 80% of from 55 - 78 years, with a mean age of 66 years. the load-bearing benefit of a completely opacified vertebral body was achieved (Figs. 6 and 7). Ten patients presented with a single level of VCF; 4 patients showed 2 levels and 2 patients showed 3 levels of VCFs (Table 1). After approximately 10 minutes of injection of PMMA the cement solidified and became harder than the native bone, and then the patients were advised to remain supine for 1 Table 1- The relation between number of patients and the hour. Patients were discharged from the hospital with post- number of levels of VCFs. procedural medications such as NSAIDS and antibiotics. No. of patients Levels 10 1 4 2 2 3

The lumbar spine was the most commonly affected area in 16 levels, 1st lumbar vertebra in 10 patients, 2nd lumbar vertebra in 4 patients, 3rd and 4th lumbar vertebrae 1 patient in each. The dorsal spine was affected only in 8 levels, 12th dorsal vertebra in 5 patients, 11th dorsal vertebra in 2 patients, and 10th dorsal vertebra in 1 patient (Table 2).

Unilateral vertebroplasty was performed in most of the patients with a total of 20 levels of VCFs and bilateral injec- tion were done in only 4 levels of VCFs, when fluoroscopic Figure 6 - Plain x-ray lumbar spine AP and lateral views showing guidance during injection showed that the unilateral injection osteoporotic fractures of L1 in 68-year-old female. was not sufficient to fill most of the fractured vertebra.

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Table 2- The number of the patients in regard to each level of occur mainly in older females, as documented by many VCF. studies.6,21,30,33 Level of the fractured vertebra No. of patients The procedure of percutaneous vertebroplasty has the D 10 1advantage of providing bone strengthening and rapid pain 2,6,12,15,18,21,27 D11 2relief. Bone strengthening is due to the me- chanical properties of methylmethacrylate, which hardens D12 5by polymeralization. It prevents further vertebral collapse 10,21,24,26 L1 10 and is less invasive than conventional surgery.

L2 4Pain relief is the fundamental advantage of vertebroplasty L3 1for patients with VCFs as the pain is usually severe and resistant to conservative treatment.5,18,27,30-32 L4 1 In our study we found that good pain relief and improved mobility occurred in 14 patients out of a total of 16 (87.5%) The volume of the material injected ranged from 2.5 - 7 ml with nearly complete pain relief in 11 patients (68.7%) and for each level of VCFs. only 2 patients (12.5%) still having pain with no improve- ment according to the VAS. Jensen et al, in 1997 found that We injected 24 levels of VCFs and only 5 injections (20%) 26 patients of 29 (90%) reported significant pain relief and showed asymptomatic lateral or anterior extravasations of improved mobility while only 3 patients (10%) reported no the cement with no post injection radiculopathy or neurol- change in pain level.23,24 ogical deficit. These occurred in patients treated in the initial part of our study due to lack of adequate experience. Cotton et al, reported that 36 patients of 37 (97%) reported partial or complete pain relief and improved mobility(7-9) In our study, we found that good pain relief and improved while, Martin et al, in 1999, found that 24 patients of 34 mobility occurred in 14 of 16 patients (87.5%); as nearly (70%) reported complete pain relief and improved complete pain relief in 11 patients (68.7%) and mild pain mobility.31 Weill et al, in 1996, found that 24 patient of 33 relief in 3 patients (18.8%). Only 2 patients (12.5%) (73%) reported good pain relief and improved mobility, continued to complain of pain with no improvement while 7 patients (21%) reported moderate pain relief and 2 according to the Visual Analogue Scale (VAS). No patient patients (6%) reported no change in pain level.40 Cortet et developed post injection spinal compression or neurological al, in 1999 reported significant improvement in 78% with deficit or increased radiculopathy or back pain (Table 3). marked reduction in VAS (p < 0.005).6

The mechanism of pain relief after vertebroplasty is not Table 3 - Outcome according to the Visual Analogue Scale. known, it may be due to destruction of the sensitive nerve Outcome No. of patients % endings in the surrounding tissues that occurs in response to Complete pain relief 11 68.7 mechanical, vascular, chemical and thermal forces during injection of methylmethacrylate.2,15,18,27 The second mecha- Mild pain relief 3 18.8 nism of pain relief is possibly due to stabilization of the No pain relief 2 12.5 micro bone fragments and reduction of mechanical forces, 17,28,29,39 Worsening of pain 0 0 which has also been suggested.

One of the most serious complications of vertebroplasty is Discussion the leakage of bone cement into the spinal canal or neural Our study here was mainly focused on the treatment of foramen which leads to spinal or nerve root compression osteoporotic fractures of the dorsolumbar and lumbar spine with neurological deficit postoperatively. The risk of this with percutaneous vertebroplasty to show if this method of complication increased if there is a cortical defect in the treatment could relieve the severe pain caused by VCFs or posterior vertebral body.16,36,38 In our study, this complica- not. tion did not arise as we excluded the cases of posterior cortical defect, as it was one of the exclusion criteria for our This study was done on 16 patients with 24 osteoporotic patient selection. VCFs documented by clinical and radiological studies, mostly in older females, as most osteoporotic fractures In other studies such as Cotton et al, in 1996 reported 2 of

66 PAN ARAB JOURNAL OF NEUROSURGERY PERCUTANEOUS VERTEBROPLASTY FOR OSTEOPOROTIC FRACTURE • Yehya & El-Nekady

37 (5.4%) patients had leakage of bone cement with nerve fractures: An open prospective study. J Rheumatol 1999, 26 root compression(8), also Weill et al, in 1996 reported one of (10): 2222-8 33 (3%) patients had leakage.40 Jensen et al, Martin et al, 7. Cotten A, Boutry N, Cortet B, et al: Percutaneous vertebro- plasty: state of the art. RadioGraphics 1998, 18: 311-320 and Cortet et al, in 1999, reported no cases of leakage as a 8. Cotten A, Dewatre F, Cortet B, et al: Percutaneous vertebro- 6,24,31 complication of injection in 3 different studies. plasty for osteolytic metastases and myeloma: effects of the percentage of lesion filling and the leakage of methyl metha- In our study, asymptomatic lateral or anterior or intradiscal crylate at clinical follow-up. Radiol 1996, 200: 525-30 9. Cotten A, Duquesnoy B: Vertebroplasty: current data and extravasations of the cement occurred in only 5 of 24 future potential. Rev Rhum Engl Ed 1997, 64(11): 645-49 injections (20%) in initial part of our study, with no post 10. Cyteval C, Sarrabere MP, Roux JO, et al: Acute osteoporotic injection radiculopathy or neurological deficit. These were vertebral collapse: open study on percutaneous injection of due to cortical destruction of the anterior vertebral body or acrylic surgical cement in 20 patients. Am J Roentgenol 1999, 173(6): 1685-90 the end plate or the hole produced by the needle after its 11. Cyteval C, Thomas E, Solignac D, et al: Prospective evaluation removal. of fracture risk in osteoporotic patients after low cement volume vertebroplasty. J Radiol 2008, 89(6): 797-801 No pulmonary complication was associated with the leaks 12. Dean JR, Ison KT, Gishen P: The strengthening effect of percutaneous vertebroplasty. Clin Radiol 2000, 55(6): 471-6 of methylmethacrylate into the lumbar venous plexuses as if 13. De Negri P, Tirri T, Paternoster G, Modano P: Treatment of it occurred we stopped the injection immediately until the painful osteoporotic or traumatic vertebral compression fractures PMMA hardened then we continued after 1 minute.32,34 by percutaneous vertebral augmentation procedure: a nonran- domized comparison between vertebroplasty and kyphoplasty. Clin J Pain 2007, 23(5): 425-30 There was no long-term clinical evidence to prove that 14. Deramond H, Depriester C, Galibert P, Le Gars D: Percuta- injection of bone cement into the osteoporotic vertebra will neous vertebroplasty with polymethylmethacrylate. Technique, prevent further compression or fracture. However, Tohmeh indication, and results. Radiol Clin North Am 1998, 36(3): 533- et al, in a postmortem study using 10 spines demonstrated 46 15. Deramond H, Wright NT, Belkoff SM: Temperature elevation that injection of cement into osteoporotic vertebral bodies caused by bone cement polymerization during vertebroplasty. (37) restored strength and stiffness , and the unipedicular Bone 1999, 25(2 Suppl 1): 17S-21S injection was as effective as bipedicular injection.25,37 This 16. Dudeney S, Lieberman I: Percutaneous vertebroplasty in the study suggests that percutaneous vertebroplasty can treatment of osteoporotic vertebral compression fractures: An open prospective study. J Rheumatol 2000, 27(10): 2526 increase the mechanical strength of the osteoporotic 17. Eck JC, Hodges SD, Humphreys SC: Vertebroplasty: A new 3,12 vertebrae. treatment strategy for osteoporotic compression fractures. Am J Orthop 2002, 31(3): 123-7; Discussion 128 18. Evans AJ, Jensen ME, Kip KE, et al: Vertebral compression Conclusions fractures: pain reduction and improvement in functional Percutaneous vertebroplasty is a minimally invasive mobility after percutaneous polymethylmethacrylate vertebro- procedure, which has been recently introduced for the plasty retrospective report of 245 cases. Radiol 2003, 226(2): treatment of osteoporotic VCFs. It is very effective in 366-372 19. Galibert P, Deramond H, Rosat P, Le Gars D: Preliminary note reducing pain and stabilizing the spine with a very low on the treatment of vertebral angioma by percutaneous acrylic complication rate. Vertebroplasty can increase the patient's vertebroplasty. Neurochir 1987, 33(2): 166-8 functional abilities, allowing return to the previous level of 20. Garfin SR, Reilley MA: Minimally invasive treatment of osteo- activity, and preventing further vertebral collapse. porotic vertebral body compression fractures. Spine J 2002, 2 (1): 76-80 21. Grados F, Depriester C, Cayrolle G, et al: Long-term observa- References tions of vertebral osteoporotic fractures treated by percuta- 1. Barbero S, Casorzo I, Durando M, et al: Percutaneous verte- neous vertebroplasty. Rheumatology (Oxford) 2000, 39(12): broplasty: the follow-up. Radiol Med 2008, 113(1): 101-13 1410-14 2. Barr JD, Barr MS, Lemley TJ, McCann RM: Percutaneous 22. Heini PF, Wälchli B, Berlemann U: Percutaneous trans- vertebroplasty for pain relief and spinal stabilization. Spine pedicular vertebroplasty with PMMA: Operative technique and 2000, 25(8): 923-28 early results. A prospective study for the treatment of 3. Bostrom MP, Lane JM: Future directions. Augmentation of osteoporotic compression fractures. Eur Spine J 2000, 9(5): osteoporotic vertebral bodies. Spine 1997, 22(24 Suppl): 38S- 445-50 42S 23. Jensen ME, Dion JE: Percutaneous vertebroplasty in the 4. Braunstein V, Sprecher CM, Gisep A, et al: Long-term reaction treatment of osteoporotic vertebral compression fractures. to bone cement in osteoporotic bone: new bone formation in Clin N Am 2000, 10: 547-68 vertebral bodies after vertebroplasty. J Anat 2008, 212(5): 697- 24. Jensen ME, Evans AJ, Mathis JM, et al: Percutaneous 701 polymethylmethacrylate vertebroplasty in the treatment of 5. Cohen JE, Lylyk P, Ceratto R, et al: Percutaneous vertebro- osteoporotic vertebral body compression fractures: Technical plasty: technique and results in 192 procedures. Neurol Res aspects. Am J Neuroradiol 1997, 18(10): 1897-1904 2004, 26(1): 41-49 25. Kim AK, Jensen ME, Dion JE, et al: Unilateral transpedicular 6. Cortet B, Cotton A, Boutry N, et al: Percutaneous vertebro- percutaneous vertebroplasty: initial experience. Radiol 2002, plasty in the treatment of osteoporotic vertebral compression 222(3): 737-41

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26. Lapras C, Mottolese C, Deruty R, et al: Percutaneous injection embolism caused by acrylic cement: a rare complication of of methyl-metacrylate in osteoporosis and severe vertebral percutaneous vertebroplasty. Am J Neuroradiol 1999, 20(3): osteoloysis (Galibert’s technic). Ann Chir 1989, 43: 371-6 (In 375-7 French) 35. Perrin C, Jullien V, Padovani B, Blaive B: Percutaneous vertebro- 27. Lehman VT, Gray LA, Kallmes DF: Percutaneous vertebro- plasty complicated by pulmonary embolus of acrylic cement. plasty for painful compression fractures in a small cohort of Rev Mal Respir 1999, 16(2): 215-7 (In French) patients with a decreased expectation related placebo effect 36. Ryu KS, Park CK, Kim MC, Kang JK: Dose-dependent due to dementia. Am J Neuroradiol 2008, 29(8): 1461-4 epidural leakage of polymethylmethacrylate after percutaneous 28. Levine SA, Perin LA, Hayes D, Hayes WS: An evidence-based vertebroplasty in patients with osteoporotic vertebral compres- evaluation of percutaneous vertebroplasty. Manag Care 2000, sion fractures. J Neurosurg 2002, 96(1 Suppl): 56-61 9(3): 56-60 37. Tohmeh AG, Mathis JM, Fenton DC, et al: Biomechanical 29. Lim TH, Brebach GT, Renner SM, et al: Biomechanical evalua- efficacy of unipedicular versus bipedicular vertebroplasty for tion of an injectable calcium phosphate cement for vertebro- the management of osteoporotic compression fractures. Spine plasty. Spine 2002, 27(12): 1297-302 1999, 24(17): 1772-6 30. Lin Wc, Cheng TT, Lee YC, et al: New vertebral osteoporotic 38. Tsou IYY, Goh PYT, Peh WCG, et al: Percutaneous vertebro- compression fractures after percutaneous vertebroplasty: retro- plasty in the management of osteoporotic vertebral compres- spective analysis of risk factors. J Vasc Interv Radiol 2008, 19: sion fractures: Initial experience. Ann Acad Med Singapore 225-31 2002, 31(1): 15-20 31. Martin JB, Jean B, Sugiu K, et al: Vertebroplasty: clinical 39. Watts NB, Harris ST, Genant HK: Treatment of painful experience and follow-up results. Bone 1999, 25(2 Suppl): osteoporotic vertebral fractures with percutaneous vertebro- 11S-15S plasty or kyphoplasty. Osteoporos Int 2001, 12(6): 429-437 32. McGraw JK, Heatwole EV, Strnad BT, et al: Predictive value of 40. Weill A, Chiras J, Simon JM, et al: Spinal metastases: indica- intraosseous venography before percutaneous vertebroplasty. tions for and results of percutaneous injection of acrylic surgical J Vasc Interv Radiol 2002, 13(2): 149-53 cement. Radiol 1996, 199(1): 241-47 33. Meunier PJ, Delmas PD, Eastell R, et al: Diagnosis and man- 41. Zoarski GH, Snow P, Olan WJ, et al: Percutaneous vertebro- agement of osteoporosis in postmenopausal women: Clinical plasty for osteoporotic compression fractures: quantitative guidelines. International Committee for Osteoporosis Clinical prospective evaluation of long-term outcomes. J Vasc Interv Guidelines. Clin Ther 1999, 21(6): 1025-44 Radiol 2002, 13: 139-48 34. Padovani B, Kasriel O, Brunner P, Peretti-Viton P: Pulmonary

68 PAN ARAB JOURNAL OF NEUROSURGERY Retrospective Study

Complications and failures of endoscopic third ventriculostomy: Perception of their avoidance

Yasser El Sawaf, Ibrahim Shafik, Reda Baza, Samy Torky

Abstract Objective: Endoscopic third ventriculostomy (ETV) has been shown to be a sufficient alternative in the surgical treatment of occlusive hydrocephalus. The purpose of this study is to evaluate complications and failures of ETV and to define the factors that can minimise their occurrence or effects.

Methods: A retrospective analysis was conducted for 40 patients (16 female and 24 male patients) in whom ETV was performed. Their age ranged from 6 months to 65 years (mean age, 9 years). Hydrocephalus was caused by aqueductal stenosis in 23 patients (10 with previous shunts), and posterior fossa lesions in 17 patients. Endoscopic third ventriculostomy was performed in all patients using a freehand method.

Results: Forty-three ETVs were attempted in 40 cases and were completed in 42. There were no fatal outcomes related to ETV. The overall success rate was 72.5 %. Complications were observed in 15 cases. Complications were CSF leak, fever, meningitis, transient diabetes insipidus, pseudomeningocele, pneumocephalus, haemorrhage and failures.

Conclusion: Many complications can be avoided by determining the correct diagnosis and using suitable technique. Most complications can be managed conservatively and do not produce long-term morbidity. The complication rate decreases markedly with surgical experience, indicating a steep learning curve. (p69-74)

Key words: Hydrocephalus, endoscopic third ventriculostomy, aqueduct stenosis, posterior fossa lesions and neuroendoscopy.

Introduction Endoscopic third ventriculostomy (ETV) has gained much in which it was treated.12,34 As experience with ETV grows, popularity and it is now considered to be a standard method the procedure will be performed by an increasing number of treatment in cases with obstructive hydrocephalus due to of neurosurgeons. It is imperative that surgeons continue to aqueductal stenosis and posterior fossa lesions.3,11,16 It is report their experience with the complications of ETV so characterised by high efficacy and lesser complications, that the procedure can continue to be made as safe as possi- especially when compared with conventional shunting inter- ble.27,28,30 In this study we evaluate the complications and ventions. It obviates the need to place a foreign body such failures of ETV as a treatment for obstructive secondary to as a ventricular shunt, thus avoiding shunt-related complica- aqueductal stenosis and posterior fossa lesions. We also tions such as malfunction, infection and over drainage.18,25,26 seek to identify factors in the pre-, intra- and postoperative Nevertheless, the outcome of ETV was reported to depend periods that can minimise their occurrence or effects. on the origin of each case of hydrocephalus and the institution Material and Methods Patient population At the Department of Neurosurgery, Tanta University, 43 Department of Neurosurgery ETVs were performed in 40 patients (16 female and 24 Faculty of Medicine Tanta University male patients) for the treatment of obstructive hydrocepha- Egypt lus between January 2004 and December 2005. The mean age was 9 years, ranging from 6 months to 65 years. Patient Correspondence: Dr. Yasser El Sawaf selection was based on MRI findings that suggested a non- Department of Neurosurgery communicating hydrocephalus. It was determined that Faculty of Medicine Tanta University hydrocephalus was caused by aqueductal stenosis in 23 Egypt patients (10 patients were previously shunted), and posterior Email: [email protected] fossa lesions in 17 patients.

VOLUME 14, NO. 2, OCTOBER 2010 69 COMPLICATIONS OF ETV - PERCEPTION OF AVOIDANCE • El Sawaf, et al

A retrospective analysis, from clinical notes, operative records intraparenchymal) meningitis and fever. Endoscopic third including videocassettes containing visual information on ventriculostomy was ineffective in 11 cases; early failure procedures and neuroimaging data was performed to eluci- was reported in 3 cases 1 - 2 weeks following surgery and date causes of complications and subsequent failures. ETV was repeated for them. The 3 cases did well after- wards. Late failure was reported in 8 cases and they were Surgical technique and endoscopic equipment treated with VP shunts (Table 3). The procedures were performed by 4 different surgeons. Rigid neuroendoscope with a working length of approxi- Table 1 - Causes of hydrocephalus in 40 cases that underwent mately 26 cm and an outer diameter of 6 mm (18 F), ETV. equipped with three channels for instruments, suction and Lesion location No. No. w/ % No. of No. Tx* irrigation and a 2.3 mm optic was used (Aesculap, Tuttlingen, of pts success Success failures abandoned Germany). Endoscopic third ventriculostomy was performed Aqueductal 13 10 77 3 -- in all patients using a freehand method and the single-arm stenosis fixation device provided by Aesculap was never used. All Aqueductal 10 8 80 2 -- stenosis with cases were performed under general anaesthesia. The malfunctioning patient was in a supine position with the head rested in a VP shunt horseshoe frame. A coronal burr hole was placed approxi- Brain stem 1 1 100 -- -- mately 3 cm lateral from the midline and just anterior to the glioma coronal suture. After incision of the dura, the endoscope 4th ventricular 8 4 50 4 -- tumours was advanced into the lateral ventricle. The foramen of Monro was then identified, and the endoscope was passed Cerebellar glioma 5 3 60 2 1 into the third ventricle. After close inspection of the floor of Pineal tumour 1 1 100 -- -- with infratentorial the third ventricle, the tuber cinereum was identified as the extension target for perforation. In general, the perforation of the floor Cerebellar 1 1 100 -- -- was made halfway between the infundibular recess and haematoma mammillary bodies in the midline. Under continuous visual Cerebellar 1 1 100 -- -- control a bluntly pointed probe was used for perforation abscess then 4 - 6 French (F) Fogarty balloon catheter was used to Abbreviations: Tx* = Treatments widen the stoma. The endoscope was then advanced through the stoma for confirmation of a free communi- Table 2 - Complications of ETV. cation between the third ventricle and the prepontine Complications No. of pts % cistern. Ringer's solution with a temperature of 37°C was Fever (self-limited) 3 7.5 used in most cases for continuous irrigation. CSF leak 4 8.75 Pseudomeningocele 3 7.5 Results Pneumocephalus 2 5 Forty-three ETVs were attempted in 40 cases. There were Subdural haematoma 2 5 no fatal outcomes related to the procedure of ETV in this Intraparenchymal Hge 1 2.5 study. There were no permanent morbidities. Endoscopic Diabetes insipidus 1 2.5 third ventriculostomy was completed in 42 of the 43 ETVs. One procedure was aborted and reported as one case of Meningitis 1 2.5 technical failure due to anatomical distortion of the floor of Failure 12 30 the third ventricle by a posterior fossa tumour which caused invagination of the floor of the third ventricle by the basilar Table 3 - Failures of ETV. artery and its branches. The causes of hydrocephalus in the Type No. Treatment 40 cases are shown in Table 1. Technical 1 Abortion and tumour attack Dynamic early 3 Repeated ETV Overall clinical improvement after ETV was achieved in Dynamic late 8 VP shunt 72.5% of patients. The best results were achieved in patients with aqueductal stenosis followed by patients with benign space-occupying lesions. Complications were observed in Discussion 15 cases (Table 2). Endoscopic third ventriculostomy has generally been accepted as the procedure of choice for treatment of non-commu- There were CSF leak, diabetes insipidus, pseudomenin- nicating hydrocephalus. This procedure is considered to be gocele, pneumocephalus, haemorrhage (subdural haematoma, simple, fast and safe.17,23,34

70 PAN ARAB JOURNAL OF NEUROSURGERY COMPLICATIONS OF ETV - PERCEPTION OF AVOIDANCE • El Sawaf, et al

Data from several series of patients undergoing ETV have Every effort should be made to optimise the selection of been published; however, complications of this procedure surgical candidates on the basis of disease origin. The pre- have not been specifically addressed well enough and most operative radiological examination should include detailed severe complications of ETV have been published as case study of the anatomy of the interpeduncular and prepontine reports.6,22,29 cisterns by inspection sagittal, thin slice, T2-weighted MR images. These images can help to identify the individual The most fatal complication reported was injury to basilar relation of the basilar artery to the floor of the third ventricle artery and in our study we did not face such complication and other anatomical features that could contraindicate the because in all procedures, when videocassettes were procedures like crowding of the neural elements below the reviewed, the rule of correct fenestration site made half way tentorium which decreases the subarachnoid spaces to the between the infundibular recess and the mammillary bodies point that in some patients the interpeduncular and pre- in the midline was strongly adhered to by the surgeons. This pontine cisterns cannot be visualised. Also, patients with way vascular injury is unlikely to occur.14,31,34 Also, prior to unusually small foramen of Monro, a larger than normal surgery sagittal MRI images should be inspected to realise interthalamic adhesion, and an unusually thick third ventri- the individual relation of the basilar artery to the floor of the cle floor. In patients with these conditions, ETV would be third ventricle.15 The floor should be perforated with blunt contraindicated.15 probe to prevent injury to basilar artery.10 Intraoperative bleeding is difficult to quantify as to its In one case the procedure was aborted due to upward significance and there seem to be differing opinions in the herniation of the basilar artery and its perforators in the literature regarding what constitutes "reportable" bleeding. floor of the third ventricle. This was a mass effect from a It is essential for the further refinement of this technique large posterior fossa tumour.32 This case is reported as that haemorrhagic complications be reported.27,28,30 technical failure (Fig. 1). There is still debate over whether technical failures should be considered complications.2 Two subdural haematomas were encountered in our study Problems resulting from an inadequate or insufficient and they were in shunt dependent patients (Fig. 2). One was preoperative evaluation cannot be considered technical symptomatic and we had to close the shunt tube with failures because preoperative MR imaging cannot be used evacuation of the sudural haematoma and the other case to predict the feasibility of an endoscopic procedure in all was small and asymptomatic and did not require further cases. Nevertheless, we believe that technical failure should treatment. Efforts should be made to avoid rapid drainage be considered a complication because, even if the patient of large quantities of CSF, and lost CSF should be replaced suffers no direct consequences from the procedure, he or with lactated Ringer's solution to avoid sudural collection she is still exposed to avoidable anaesthesia and the risks of following ETV. It is advised that shunt should be removed an unnecessary surgical procedure.12 or ligated during ETV or during the days following ETV.13,24

Figures 1a ↑ Preoperative midsagittal and axial T2-weighted Figure 2 - Subdural haematoma complicating ETV. image showing narrowing of pre- pontine cistern due to forward displacement of the brainstem We observed a case of haemorrhagic contusion in the right by posterior fossa mass. b) ← frontal lobe in the trajectory of the telescopic shaft seen on Endoscopic view of the floor of the follow-up MRI scan. That patient awoke with delay but third ventricle of the same patient showing obliterated target area fortunately the case was not associated with any clinical 24 denoting impossible safe crea- sequels. We think this complication could have been tion of the ventriculostoma. avoided by using smaller endoscope than the one which is available in our department (its outer diameter is 6mm). The surgeon, who is one of the authors, mentioned that

VOLUME 14, NO. 2, OCTOBER 2010 71 COMPLICATIONS OF ETV - PERCEPTION OF AVOIDANCE • El Sawaf, et al there were three trials before hitting the ventricle with the or forceps is for perforation.16 Irrigation solutions can also telescopic shaft. He believed it was difficult due to be a cause of injury of hypothalamus. Generous irrigation inaccurate position and incision in that case. So, positioning with normal saline solution has been associated with of the patient is crucial and we advocate keeping the head in disturbance of electrolytes and hypothalamic dysfunction. a midline position with mild neck flexion to simplify the Late arousal and postoperative confusion have also been surgeon's visualisation of the anatomic landmarks. Also, noted. These complications are caused by trauma to sensi- planning the incision and placement of the burr hole play tive hypothalamic structures comprising the walls and floor important roles in avoiding parenchymal injury.19 of the third ventricle.

Small haemorrhages occurred during the procedures which According to many authors, it is mentioned in the medical were venous in origin and occurred at the margins of the literature that patients who have been previously shunted ventirculostomy and ceased spontaneously with or even are technically more difficult to perform ETV upon, as they without irrigation. We think that the management of most have less marked ventricular dilatation, a thicker ventricular intraoperative haemorrhage consists of irrigation and floor and often abnormal anatomy.9,21 It is also mentioned patience as previously seen during review of videocassettes. that in some patients, an ETV procedure may have to be Electrocautery was never used in this series and it is abandoned if the floor of the third ventricle is too thick.21 In certainly dangerous in cramped ventricular spaces.33 When our study we observed no problems with those patients. the source of bleeding is in the floor of third ventricle at the Thus, previous shunting did not increase the risk of ETV. site of fenestration, a balloon catheter can be used to apply Endoscopic third ventriculostomy was successful, resulting haemostatic pressure to the bleeding tissue. It should be in shunt independence. noted that if significant intraventricular bleeding occurs, an external ventricular drain should be left in place at the close Two cases of mild pneumocephalus were reported in our of the procedure. It is also important that the surgeons put in study. Although pneumocephalus is usually considered a their minds that ETV is a minimally invasive procedure and minor or insignificant complication, it can delay postopera- should be abandoned if conditions like significant bleeding tive recovery and can be associated with headache, nausea or unfavourable anatomy make fenestrating the floor too and vomiting. Entrapment of air at the time of surgery can dangerous.1,33 interfere with direct visualisation of the anatomic landmarks that are essential to performing a third ventriculostomy Four cases of CSF leak were encountered in this study. Two safely. This can be minimised by keeping the patient's head cases were transient leaks and represented the transient in a midline anatomic position with the burr hole at or near period of persistent intracranial hypertension frequently the most superior point, by carefully flushing all irrigation observed in the immediate postoperative period and there lines of air bubbles, and by irrigating gently while introduc- was spontaneous stoppage of CSF leak. The other 2 cases ing the endoscope. Attention should be paid to minimising continued to have persistent leak and was a sign of CSF loss, especially in the early stages of the procedure. treatment failure. They both had repeated ETV. One finally Nitrous oxide should not be used for anaesthesia during required shunt placement. The possibility of a CSF leak ETV because of the potential for formation of tension may be minimised by using the smallest appropriate endo- pneumocephalus.31,34 scope (especially in patients with large ventricles) and by minimising the dural opening. Perneczky et al, recommend Four cases were reported to have fever in the postoperative that the outer diameter of a cranial endoscope used in period. Fever was self limited in 3 cases and lasted 24 - 48 newborn and young paediatric patients must not exceed 3 - hours. In this setting, fever may be due to residual blood in 4 mm (9 - 12 F). They stated that 6 mm (18 F) is well toler- the ventricular system or may be caused by generous ated in adults. A layered closure of the scalp is necessary.16 irrigation with normal saline solutions which act as a trauma to sensitive hypothalamic structures comprising the The floor of the third ventricle is not a membrane but a part walls and floor of the third ventricle. The 4th case of fever of the hypothalamus. We found in our study one case which had meningitis proved by analysis and culture of the CSF. had postoperative transient diabetes insipidus and re- Pre-existing ventricular shunt hardware was found in that sponded to mere fluid replacement. In this case the floor case and it was removed. We believe that it is important to was relatively thick. However the surgeon kept probing the remove all shunt hardware after ETV whenever possible. It floor gently without the aid of electric coagulation. It is must be emphasised that careful and thorough sterilisation apparently safe to puncture the floor of the third ventricle of the endoscopic equipment and the use of perioperative but only when it is thinned as a result of pressure of antibiotics can minimise the occurrence of infection.10,13,25,26 hydrocephalus.30 According to Perneczky et al, in the case of an extremely tough or floating floor, bipolar coagulation The success of third ventriculostomy lies in large part in

72 PAN ARAB JOURNAL OF NEUROSURGERY COMPLICATIONS OF ETV - PERCEPTION OF AVOIDANCE • El Sawaf, et al selecting patients whose CSF physiology can respond months of well-being.2,8,12,34 favourably to the procedure. Early failure is the result of factors including bleeding around the fenestration site, Pseudomeningocele was encountered in 3 patients and they unnoticed additional arachnoid membranes occluding the were all infants. Pseudomeningocele are subgaleal CSF flow of CSF, and an inadequate size of the fenestration.5,8 collections with an overall incidence of 2%. Their favouring Late failure is the result of subsequent closure of the factors are large dural openings, subcutaneous over dissec- fenestration by gliotic tissue or arachnoid membrane. tion and a loose skin; for these reasons, they have been Closure of ventriculostomy whether early or late can be more frequently described in infants. The immature managed by repeating the procedure.4,17,20 In our series, 3 subarachnoid spaces of these patients might contribute to patients with recurring signs and symptoms of intracranial this process; due to the elevated pressure gradient at the hypertension underwent repeated surgery within 2 weeks level of the arachnoid villi, CSF may tend to flow along the from the first procedure. Early failure was due to missed lower resistance pathway offered by the ventriculostomy second membrane in one case and small stoma in the other tract. Large burr holes and long incisions of the dura mater 2 cases. In these cases third ventriculostomy would allow a may also lead to progressively enlarging skull defects, with transient improvement as a result of the ventricular tapping the same mechanisms of enlarging post-traumatic growing performed during the procedure, followed by recurrence of fractures. Single lumbar tap for each case with tight head symptoms in the days following the procedure and sagittal bandage resolved the problem. T2-weighted MRI performed after the procedure did not demonstrate any flow artifact at the level of the third In order to reduce the incidence of these complications, ventriculostomy. It must be mentioned that intermittent Cinalli has proposed a modification of the traditional burr symptoms of intracranial hypertension are consistently seen hole technique in infants. The skin incision is done on the within the first day after ETV and referred to as the right anterior angle of the anterior fontanelle. A small area "adaptation period". The adaptation period may falsely lead of the frontal and parietal bones is dissected from the dura to the assumption that ETV has failed. Therefore, close and removed in order to allow a rectilinear parasagittal clinical follow-up is mandatory and sometimes even dural incision; after removal of the endoscope, the dura is transient ICP monitoring is helpful.2,24 sutured with a 5/0 resorbable suture in a watertight fashion.7

It must be stated that the optimum size of the perforation is Conclusion unknown but most authors think that a size of at least 5 mm In conclusion, many of the complications of ETV are is sufficient. Balloon catheters are available in different transient in nature. Skilled management of intraoperative diameters but catheters smaller than 1 mm (3 F), enabling problems can keep these complications limited to the intra- perforation size of a maximum of 5 mm, were found not to operative or perioperative period. be useful. The preferred diameters in our series are 1.3 mm and 2 mm (4 and 6 F) enabling a perforation size of up to The complication rate of endoscopic ETV is low in 10 mm. A disadvantage of catheters that are too large is the experienced hands. Although the technique has been greatly danger of injury to small vessels.8,24,27,28,30 In one case, a refined since its advent almost a century ago, today's second membrane was present beneath the floor of the third neurosurgeon must never forget that this seemingly simple ventricle. Our findings support the recommendation of procedure holds the potential for a number of devastating others to advance the endoscope through the perforation complications. Most of the severe complications occur at and to visualise a free communication along the basilar the beginning of the endoscopic career. That is why inten- artery (a merit of the freehand method).16 It is important to sive training in the technique is advisable and experience is mention that those early failures were registered at earlier important to the success of ETV and for avoiding compli- dates in the study and reduction of failures afterwards is cations. The surgeon should be aware of these potential indicative of a steep learning curve with accumulation of complications and should take all precautions to avoid the authors' personal experience and better preoperative them. It is imperative that surgeons continue to report their judgment.28,31 experience with the complications of ETV so that the procedure can continue to be made as safe as possible. There are several reports in the literature of death following late failure of ETV and this remains a management problem References because the failure can occur in a short period of time and 1. Abtin K, Thompson BG, Walker ML: Basilar artery perforation may be unpredictable. Tumour progression and inadequate as a complication of endoscopic third ventriculostomy. Pediatr CSF absorption at the level of the arachnoid villi may result Neurosurg 1998, 28(1): 35-41 2. Beems T, Grotenhuis JA: Long-term complications and in early or late failure. It is not understood why a cohort of definition of failure of neuroendoscopic procedures. Childs patients with open fenestrations exhibits deterioration after Nerv Syst 2004, 20: 868-877

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3. Brockmeyer D, Abtin K, Carey L, Walker ML: Endoscopic third Neurochir Suppl (Wien) 1994, 61: 79-83 ventriculostomy: An outcome analysis. Pediatr Neurosurg 19. Jones RFC, Stening WA, Brydon M: Endoscopic third ventricu- 1998, 28(5): 236-40 lostomy. Neurosurg 1990, 26(1): 86-92 4. Buxton N, Macarthur D, Mallucci C, et al: Neuroendoscopy in 20. Kadrian D, van Gelder J, Florida D, Jones R, Vonau M, Teo C, the premature population. Childs Nerv Syst 1998, 14(11): 649- et al: Long-term reliability of endoscopic third ventriculostomy. 652 Neurosurg 2005, 56(6): 1271-1278 5. Buxton N, Macarthur D, Mallucci C, et al: Neuroendoscopic 21. Kehler U, Gliemroth J, Knopp U, Arnold H: How to perforate third ventriculostomy in patients less than 1 year old. Pediatr safely a resistant floor of the 3rd ventricle? Technical note. Neurosurg 1998, 29(2): 73-76 Minim Invasive Neurosurg 1998, 41(4): 198-199 6. Buxton N, Punt J: Cerebral infarction after neuroendoscopic 22. McLaughlin MR, Wahlig JB, Kaufmann AM, Albright AL: third ventriculostomy: case report. Neurosurg 2000, 46(4): 999- Traumatic basilar aneurysm after endoscopic third ventricu- 1001; Discussion 1001-2 lostomy: case report. Neurosurg 1997, 41(6): 1400-3; Discussion 7. Cinalli G: Endoscopic third ventirculostomy. In: Cinalli G, Maixner 1403-4 WJ, Sainte-Rose C (eds), Pediatric Hydrocephalus. Milan, 23. Oka K, Yamamoto M, Ikeda K, Tomonaga M: Flexible Italy, Springer-Verlag 1998, pp 361-388 endoneurosurgical therapy for aqueductal stenosis. Neurosurg 8. Cinalli G, Sainte-Rose C, Chumas P, Zerah M, Brunelle F, Lot 1993, 33(2): 236-42; Discussion 242-3 G, et al: Failure of third ventriculostomy in the treatment of 24. Peretta P, Ragazzi P, Galarza M, Genitori L, Giordano F, aqueductal stenosis in children. J Neurosurg 1999, 90(3): 448- Mussa F, Cinalli G: Complications and pitfalls of neuroendo- 454 scopic surgery in children. J Neurosurg 2006, 105(3 Suppl): 9. Cinalli G, Salazar C, Mallucci C, Yada JZ, Zerah M, Sainte- 187-193 Rose C: The role of endoscopic third ventriculostomy in the 25. Pudenz RH, Foltz EL: Hydrocephalus: overdrainage by management of shunt malfunction. Neurosurg 1998, 43(6): ventricular shunts. A review and recommendations. Surg 1323-7; Discussion 1327-9 Neurol 1991, 35(3): 200-12 10. Cohen AR: Endoscopic ventricular surgery. Pediatr Neurosurg 26. Rekate HL: The role of endoscopic third venrticulostomy in the 1993, 19(3): 127-134 management of shunt malfunction. Neurosurg 1998, 43(6): 11. Drake JM: Ventriculostomy for treatment of hydrocephalus. 1323-1329 Neurosurg Clin N Am 1993, 4(4): 657-66 27. Schroeder HW, Niendorf WR, Gaab MR: Complications of 12. Fukuhara T, Vorster SJ, Luciano MG: Risk factors for failure of endoscopic third ventriculostomy. J Neurosurg 2002, 96(6): endoscopic third ventriculostomy for obstructive hydro- 1032-40 cephalus. Neurosurg 2000, 46(5): 1100-9; Discussion 1109-11 28. Schroeder HW, Oertel J, Gaab MR: Incidence of complications 13. Grant JA, McLone DG: Third ventriculostomy: a review. Surg in neuroendoscopic surgery. Childs Nerv Syst 2004, 20: 878- Neurol 1997, 47(3): 210-212 83 14. Handler MH, Abbott R, Lee M: A near-fatal complication of 29. Schroeder HW, Warzok RW, Assaf JA, Gaab MR: Fatal endoscopic third ventriculostomy: case report. Neurosurg subarachnoid hemorrhage after endoscopic third ventricu- 1994, 35(3): 525-7; Discussion 527-8 lostomy. Case report. J Neurosurg 1999, 90(1): 153-5 15. Hayashi N, Endo S, Hamada H, et al: Role of preoperative 30. Teo C, Rahman S, Boop FA, Cherny B: Complications of endo- midsagittal magnetic resonance imaging in endoscopic third scopic neurosurgery. Childs Nerv Syst 1996, 12(5): 248-53 ventriculostomy. Minim Invasive Neurosurg 1999, 42(2): 79-82 31. Walker M: Complications of third ventriculostomy. Neurosurg 16. Hopf NJ, Grunert P, Fries G, Resch KD, Perneczky A: Clin N Am 2004, 15: 61-66 Endoscopic third ventriculostomy: outcome analysis of 100 32. van Aalst J, Beuls EA, van Nie FA, Vles JS, Cornips EM: Acute consecutive procedures. Neurosurg 1999, 44(4): 795-804; distortion of the anatomy of the third ventricle during third Discussion 804-6 ventriculostomy. Report of four cases. J Neurosurg 2002, 96 17. Jones RFC, Brazier DH, Kwok BCT, et al: Neuroendoscopic (3): 597-9 third ventriculostomy. In: Cohen AR, Haines SJ (eds), Minimally 33. Vloeberghs M, Cartmill M: Improved safety of neuroendoscopic Invasive Techniques in Neurosurgery. Baltimore, Williams & third ventriculostomy by using an operative Doppler ultrasound Wilkins 1995, pp 33-48 probe. Technical note. Neurosurg Focus 1999, 6(4): E13 18. Jones RFC, Kwok BCT, Stening WA, Vonau M: Neuro- 34. Wellons JC 3rd, Bagley CA, George TM: A simple and safe endoscopic third ventriculostomy. A practical alternative to technique for endoscopic third ventriculocisternostomy. Pediatr extracranial shunts in non-communicating hydrocephalus. Acta Neurosurg 1999, 30(4): 219-223

74 PAN ARAB JOURNAL OF NEUROSURGERY Anatomical Study

Papez circuit: An anatomical study by cadaveric dissection

Forhad Hossain Chowdhury1, Akhlaque Hossain Khan2

Abstract Objective: This study was done to completely study the Papez circuit by cadaveric dissection and it’s relation to ventricles and other related anatomical structures.

Methods: Eight formalin fixed cerebral hemispheres were microscopically dissected for Papez circuit. Klingler’s technique of fibre dissection was adopted. The circuit was dissected from medial and superior-lateral cerebral surfaces. Bilateral hemispheric dissection was done simultaneously in intact brain from superior-lateral surface in two brains (4 hemispheres). During and after dissection it’s relation with lateral ventricles and other related structures were studied.

Results: Papez circuit was demonstrated by total dissection. Fibres leave the hippocampal formation and proceed through the fornix; most of these fibres have been shown to terminate in the mammillary nuclei of the hypothalamus. From here, the mammillothalamic tract ascends to the anterior group of thalamic nuclei. This group of nuclei projects to the cingulate gyrus through the anterior limb of internal capsule to anterior cingulate gyrus. From the cingulate gyrus there is an association bundle; the cingulum, which connects the cingulate gyrus with the parahippocampal gyrus part of the limbic lobe. The parahippocampal gyrus projects to the hippocampal formation and circuit is completed. Relation of different parts of the circuit with surrounding structures were also clearly seen.

Conclusion: Knowledge of the microsurgical anatomy of the Papez circuit is not only important for understanding memory mechanism and other limbic functions but also very important in management of lateral and third ventricular lesions, in transcallosal, transventricular, supraseller and temporal lobe surgery, and for psycho-neurosurgery. (p75-80)

Key words: Papez circuit, fornix, mammillothalamic tract, , parahippocampal gyrus, cingulate gyrus and microsurgical anatomy.

Introduction Neoplastic, vascular and other pathological lesions in ven- ing with the above mentioned pathologies he must be aware tricular system (lateral and third ventricle) can produce of the microsurgical anatomy of the Papez circuit, ventricu- clinical neuropsychiatric problems by involving the facets lar system and other related neural structures to maintain of Papez circuit or by pressure over it. Different lesions in integrity of the circuit so that the patient does not suffer hippocampus can produce intractable epilepsy where surgi- postoperative neurological deficit/s. cal treatment is needed. One must not forget the role of in intractable psychiatric illness involving In this study, we attempted to dissect the Papez circuit com- the Papez circuit. When a neurosurgeon finds himself deal- pletely and to evaluate the anatomical relation of the circuit to surrounding neuro-structures and ventricular system. This type of cadaveric study appears rarely in the literature. 1 Department of Neurosurgery Dhaka Medical College Hospital 2Department of Neurosurgery Materials and methods Banghabondhu Sheikh Muzib Medical University (BSMMU) Dhaka Eight cerebral hemispheres that were formalinated for 3 - 6 Bangladesh months were taken for dissection of Papez circuit in the Department of Neurosurgery, King Edward Memorial Hospi- Correspondence: Dr. Forhad Hossain Chowdhury tal, Mumbai, India during the author’s fellowship in that Department of Neurosurgery Department. The circuit was dissected using Klingler’s fibre Dhaka Medical College Hospital 32 Bokshibazar, Dhaka-1200 dissection method under operating microscope. Dissection Bangladesh was done by bamboo made small spatula surgical dissector Email: [email protected] and sharp cutting instruments.

VOLUME 14, NO. 2, OCTOBER 2010 75 PAPEZ CIRCUIT: AN ANATOMICAL STUDY BY CADAVERIC DISSECTION • Chowdhury & Khan

Dissection was started from superior and lateral surfaces of by superior-lateral and medial surface dissection of cerebral cerebral hemispheres in an intact brain simultaneously on hemisphere. In our dissection, parts of the circuit are both hemispheres. The part of the brain above the corpus (sequentially) hippocampus - fornix - mammillary body - callosum was transected and removed. Body, trigone, fron- mammillothalamic tract - anterior group of thalamic nucleus - tal and occipital horn of lateral ventricle was opened anterior thalamic projection to cingulate gyrus - cingulum- carefully by dissecting and removing the corpus callosal parahippocampus - dentate gyrus - hippocampus. Parts are fibres under microscope. Whole corpus callosum was re- shown in Figure 1. moved dissecting and separating underlying fornix, septum pellucidum and hippocampal commissure, except the most anterior part of genu and most posterior part of splenium. Extreme care was taken not to damage the caudate nucleus. Then temporal horn was exposed by removing its roof (tapetum and optic radiation). Choroid plexus was removed from foramen of Monro to choroidal point in temporal horn. The following structures were dissected out and identified sequentially, septum pellucidum, fornix with its commissure and hippocampus, stria medullaris, stria termi- nalis, caudate nucleus and thalamus. An oblique incision was made from a point 3.5 cm lateral to frontal pole to head of caudate nucleus just lateral to the anterior part of stria terminalis, then the incision extended posteriorly up to the fimbria of fornix just lateral to stria terminalis. The incision was deepened inferiorly and part of the caudate nucleus, Figure 1 - Papez circuit and amygdala: (1) amgdala, (2) hippo- thalamus, corona radiata, internal capsule, lantiform nucleus campus, (3) fornix, (4) mammillary body, (5) mammillothalamic tract, and part of upper midbrain were removed to expose the (6) anterior thalamic nuclei, (7) anterior thalamic radiation, (8) fimbria, sabiculm, dentate gyrus and parahippocampal gyrus. cingulate gyrus, (9) parahippocampal gyrus and dentate gyrus. Uncus was identified and amygdala was dissected out anterior-medio-superior to head of hippocampus and chor- Hippocampus (Figs. 2-5): This is thought to be the centre oidal point. (nucleus) of Papez circuit. The circuit starts and ends here. It is situated in the floor of temporal horn lateral to it is Before commencing medial dissection, brain was bisected collateral eminence. Anterior to head of hippocampus is into two symmetrical halves by cutting strictly in the midline amagdala. It is separated medially from thalamus by choroids with sharp instrument. Dissection started from foramen of fissure and choroids plexus of temporal horn. It has head, Monro. Column of fornix was dissected down to mammillary body and tail (from anterior to posterior). Tail is continued body lateral to ependymal lining through hypothalamus. as fimbria of fornix posteriorly. The hippocampus is con- Precommissural fibres of fornix to septal area were dis- nected with septal area, hypothalamus and thalamus of the sected and preserved. Anterior commissure was identified same side by foniceal fibres and by commissural fibres to between pre- and postcommissural fibres of fornix. Mam- opposite side. It is also connected with parahippocampal millothalamic tract was traced from mammillary body that gyrus and cingulate gyrus through dentate gyrus. Hippo- passes superio-laterally and posteriorly through the thalamus campus, sabiculum and dentate gyrus are together called to anterior group of nucleus. Anterior thalamic radiation, hippocampal formation. projected anteriorly and anterio-superiorly, that passed lateral to head of caudate nucleus to join the anterior limb of Fornix (Figs. 2-9): The fornix consists mainly of hippo- internal capsule were dissected out. These fibres curved campomammillary and hippocamposeptal fibres. Forniceal superio-medially and pass through the fibres of corpus white fibres arise from the hippocampus, subiculum and callosum to end in anterior part of cingulate gyrus. Gray dentate gyrus. It wraps the thalamus in the wall of lateral mater and short association fibres of cingulate gyrus were ventricle. The fornix is C-shaped. It has four parts: fimbria, removed and cingulum fascicle was dissected out through- crus, body, and columns of fornix. out the whole cingulate gyrus to parahippocampal gyrus. Fibres passing to hippocampus through dentate gyrus from Fimbria starts from tail of hippocampus and forms the pos- parahippcampus were then dissected out. terior medial part of floor of temporal horn. Fimbriodentate sulcus separates it from dentate gyrus and it is separated Results from the geniculate body, optic and auditory radiations by In our study we dissected out all parts of the Papez circuit the choroidal fissure.

76 PAN ARAB JOURNAL OF NEUROSURGERY PAPEZ CIRCUIT: AN ANATOMICAL STUDY BY CADAVERIC DISSECTION • Chowdhury & Khan

Figure 2 - Parts of Papez circuit, right lateral ventricular parts and other related structures are shown after exposure of whole ventricle from above, part of caudate nucleus and thalamus removed: (1) foramen of Monro, (2) choroid plexus of body of lateral ventricle, (3) fornix, (4) ante- rior horn of lateral ventricle, (5) septum pellucidum, (6) rostrum of corpus callosum, (7) caudate nucleus, (8) thalamus, (9) pineal region, (10) sple- nium, (11) cerebellum, (12) occipital horn of lateral ventricle, (13) trigone of lateral ventricle, (14) temporal horn of lateral ventricle, (15) hippocam- pus, (16) uncinate fascicle and (17) temporal pole.

Figure 3 - Parts of Papez circuit, right lateral ventricular parts and other related structures are shown after exposure of whole ventricle from above bilaterally by removing necessary parts of hemisphere and corpus callo- sum, thalamus, caudate nucleus, lantiform nucleus, internal capsule and crus cerebri: (1) amygdala, (2) head of hippocampus, (3) fimbria fornix, (4) dentate gyrus, (5) collateral eminence in temporal horn, (6 and 7) occipital horn of lateral ventricle, (8) occipital lobe, (9) cerebellum, (10) splenium, (11) choroid plexus, (12) trigone of lateral ventricle, (13) body of fornix, (14) septum pellucidum, (15) foramen of Monro, (16) thalamus, (17) head of caudate nucleus, (18) frontal horn of lateral ventricle, (19) rostrum of corpus callosum, (20) frontal lobe, (21) parahippocampal gyrus and (22) temporal pole.

Figure 4 - Parts of Papez circuit, right lateral ventricular parts and other related structures are shown after exposure of whole ventricle from above by removing necessary parts of hemisphere and corpus callosum, thalamus, caudate nucleus, lantiform nucleus, internal capsule and crus cerebri with special focus on temporal horn structures: (1) frontal area, (2) temporal pole, (3) uncinate fascicle, (4) anterior perforated substance, (5) internal capsule, (6) amygdala, (7) hippocampus, (8) fimbria, (9) dentate gyrus, (10) parahippocampal gyrus, (11) uncus, (12) choroid fissure, (13) crus fornix, (14) optic radiation, (15) foramen of Monro, (16) thalamus, (17) septum pellucidum and septal vein, (18) rostrum of corpus callosum and (19) body of fornix.

Figure 5 - Parts of Papez circuit, right lateral ventricular parts and other related structures are shown after exposure of whole ventricle from above bilaterally by removing necessary parts of hemisphere and corpus callo- sum, thalamus, caudate nucleus, lantiform nucleus, internal capsule and crus cerebri: (1) frontal pole, (2) rostrum of corpus callosum, (3) head of corpus callosum, (4) septum pellucidum, (5) foramen of Monro, (6) body of fornix, (7) crus fornix, (8) hippocampal commissure (9) striaterminalis, (10) splenium, (11) fimbria, (12) hippocampus, (13) amagdala, (14) thalamo- caudate sulcus, (15) dentate gyrus, (16) collateral eminence in temporal horn, (17) occipital horn of lateral ventricle, (18) optic radiation, (19) cerebellum, (20) occipital lobe, (21) insula and external capsule

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The crus of the fornix is the posterior continuation of fimbria in the floor of the third ventricle situated on the both sides that covers the posterior surface of the pulvinar in the of midline behind the pituitary stalk and can easily be seen medial part of the atrium and arches superio-medial toward both from ventricular and inferior surface of the brain. the lower surface of the splenium. It forms the medial part Column of fornix ends and mammillothalamic tract originates of anterior wall of atrium. Thin sheet of white fibre that from here. Mammillothalamic tract ascends superio-laterally interconnects medial edge of both crus below the splenium and posteriorly through the thalamus to anterior group of is known as hippocampal commissure. nucleus. Some fibres of the tract also go to dorso-medial nucleus of thalamus. Body of the fornix is formed by the joining of both crus in midline at the junction of body and atrium of lateral ventri- Anterior group of thalamic nuclei (Figs. 7-9): Anterior cle. Passing forward above the thalami superiorly it blends thalamic group of nuclei lies anteriorly near the foramen of with lower edge of the septum pellucidum and forms the Monro. They receive mammillothalamic tract and project medial wall of the body of the lateral ventricle. anterior thalamic radiation.

Column of fornix begins when body of fornix separates into Anterior thalamic projection (Figs. 7-9): Fibres from a pair of columns at the anterior pole of the thalamus; the anterior group of thalamic nucleus project anteriorly and columns that arch anterior-inferiorly to form the superior anterior-superiorly passing lateral to head of caudate nu- and anterior margins of the foramen of Monro. Then they cleus and links to anterior limb of internal capsule. These blend into the lateral walls of the third ventricle and pass fibres curve superio-medially and pass through the fibres of down behind the anterior commissure through the hypo- corpus callosum to end in anterior part of cingulate gyrus. thalamus to mammillary body. Some fibres of the anterior thalamic projection are seen to

The part of the thalamus lateral to the body of the fornix end in head of caudate nucleus and some fibres are seen to forms the floor of the body of the lateral ventricle and the be passed through the lateral part of caudate nucleus. Small part medial to the fornix forms part of the lateral wall of the portion of this radiation is also passed to prefrontal cortex. velum interpositum and third ventricle. The crus of the fornix crosses the pulvinar approximately midway between Cingulate gyrus (Figs. 6-9): This is on the medial cerebral the medial and lateral edge of the pulvinar. The part of the surface, just above the corpus callosum. It begins anterior to pulvinar lateral to the crus of the fornix forms part of the rostrum of corpus callosum and septal area. It follows the anterior wall of the atrium. The lower border of the fornix curvature of callosum, below and behind the splenium and forms the upper border of the choroidal fissure and the it continues with parahippocampal gyrus through isthmus. lower border of choroid fissure is formed by stira medullaris; Anteriorly, it receives anterior thalamic radiation. The choroids plexus is attached here. The septum pellucidum cingulum within the cingulate gyrus contains long and short stretches across the interval between the anterior parts of the association fibres that follow the curve of the cingulate corpus callosum and the body of the fornix. It is composed gyrus and corpus callosum. Cingulum ends in parahippo- of paired laminae and separates the frontal horns and bodies campal gyrus. of lateral ventricles. Parahippocampal gyrus (Figs. 3-5,8,9): This is the Mammillary body and mammillothalamic tract (Figs. most medial gyrus on the inferior surface of the temporal 6-9): Mammillary bodies are egg-shaped paired structures lobe. It receives cingulum and projects to hippocampus through the dentate gyrus.

Dentate gyrus (Figs. 3-5): A short narrow gyrus can only be seen to adequate lateral retraction of parahippocampal gyrus by its characteristic appearance. It connects para- hippocampal gyrus to hippocampus.

Figure 6 ← Medial surface of cerebral hemisphere showing: (1) mammillary body, (2).optic nerve, (3) anterior commissure, (4) column of fornix, (5) foramen of Monro, (6) head of caudate nucleus, (7) septal area, (8) precomisural fornix, (9) rostrum of corpus callosum, (10) cingulate gyrus, (11) splenium, (12) posterior commissure, (13) habinular commissure, (14) thala- mus, (15) thalamohypothalamic sulcus, (16) tectum and (17) midbrain.

78 PAN ARAB JOURNAL OF NEUROSURGERY PAPEZ CIRCUIT: AN ANATOMICAL STUDY BY CADAVERIC DISSECTION • Chowdhury & Khan

Figure 7 - Medial surface of cerebral hemisphere showing white fibre dissection for parts of Papez circuit: (1) mammillary body, (2) post commis- sural fornix, (3) mammillothalamic tract, (4) column of fornix, (5) rostrum of corpus callosum, (6) splenium (7) stria terminalis, (8) precommissural fornix, (9) anterior commissure, (10) septal area, (11) anterior thalamic radiation, (12) head of caudate nucleus and (13) cingulate gyrus.

Figure 8 - Medial surface of cerebral hemisphere showing white fibre dissection for parts of Papez circuit after removal of brain stem, cerebellum and part of thalamus and caudate nucleus: (1) mammillary body, (2) post commissural fornix, (3) mammillothalamic tract, (4) anteror thalamic nuclei, (5) body of fornix, (6) stria terminalis, (7) caudate nucleus, (8) ante- rior thalamic radiation, (9) crus fornix, (10) fimbria fornix, (11) uncus, (12) parahippocampal gyrus, (13) cingulum, (14) rostrum of corpus callosum and (15) temporal pole.

Figure 9 - Medial surface of cerebral hemisphere showing white fibre dissection for parts of Papez circuit after removal of brain stem, cerebellum and part of thalamus and caudate nucleus: (1) mammillary body, (2) mam- millothalamic tract, (3) fornix, (4) anteror thalamic nuclei, (5) anterior thalamic radiation, (6) rostrum of corpus callosum, (7) cingulum, (8) crus fornix, (9) parahippocampal gyrus, (10) trigone of lateral ventricle, (11) thalamus, (12) caudate nucleus, (13) uncus, (14) splenium, (15) body of lateral ventricle and (16) temporal pole.

Discussion parahippocampal gyrus, and the dentate gyrus.1,2,7,9 In our In 1937 James Papez described a pathway involving some study we dissected the above mentioned parts to identify the limbic and cortical structures and associated pathways.10 These, circuit anatomically and in our dissection we also found that he postulated, formed the anatomical substrate for emotional anterior thalamic projection communicates not only cingu- experiences.10 The pathway forms a series of connections, late gyrus but also to prefrontal cortex and caudate nucleus. which has since been called the Papez circuit or medial limbic Through precomissural fibres of fornix the circuit also has circuit.2,3,7 Defense reaction circuit and baso-lateral circuit connection with septal area (Fig. 7). There is also commu- are the other two circuits of limbic lobe.2,3 The parts of Papez nication between hippocampus and amygdala. circuit includes, hippocampus, fornix, mammillary body, mammillothalamic tract, anterior group of thalamic nucleus, The circuit may be affected by various pathologies, such as anterior thalamic projection to cingulate gyrus, cingulum, degenerative (Alzheimer’s disease), deficiency condition (Vita-

VOLUME 14, NO. 2, OCTOBER 2010 79 PAPEZ CIRCUIT: AN ANATOMICAL STUDY BY CADAVERIC DISSECTION • Chowdhury & Khan min B1 deficiency, Korsakoff psychosis4), vascular (infarction, the hippocampal formation, leads to a unique condition in AVM, angioma), hypoxia (temporal mesial sclarosis6,8) which the person can no longer form new declarative or neoplastic (intrinsic to Papez circuit-glioma, extrinsic to the episodic memories, although older memories are intact. The circuit-glioma, meningioma, colloid cyst, craniopharyngioma, individual cannot remember what occurred moments before. choroids plexus papilloma, and other tumours), trauma, Therefore, the individual is unable to learn (i.e., to acquire new iatrogenic (postsurgical), psychiatric illness.2 These patients information) and is not able to function independently.7 may present with defective functions of Papez circuit such as memory disturbance (especially recent), personality Conclusion changes, changes in emotional behaviour, loss of sponta- Knowledge of the microsurgical anatomy of the Papez neity and initiative, affective disorders, hallucinations, circuit is not only important for understanding memory intractable epilepsy with or without features of intracranial mechanism and other limbic functions but also very impor- space occupying lesion.5 Various investigations including tant in management of lateral and third ventricular lesions, neuro-imaging can identify these conditions with ease. in transcallosal, transventricular, supraseller and temporal lobe surgery, and psycho-neurosurgery. Surgical intervention is needed in neoplastic, vascular, intractable epilepsy and intractable psychiatric conditions. Acknowledgment: The authors’ would like to express Appropriate surgical approach and peroperative identifi- the deep appreciation to Prof. Dr. Atul H Goel, who placed cation and preservation of Papez circuit along with other his laboratory facilities at our disposal. He stimulated the related vital structures are essential for a good outcome in technique and introduced the fibre dissection technique in these surgeries. Lesion making surgeries in psychiatric his Microneurosurgical fellowship Course at King Edward illness should be done only after definite indications with Memorial Hospital, Parel, Mumbai, India. extreme caution.2 In case of intractable epilepsy where mesial temporal excision is planned, the opposite mesial References temporal lobe’s function should be kept intact, otherwise 1. Carpenter MB (ed): Core Text of Neuroanatomy, 4 Ed. Baltimore, postoperatively patient may become disastrously disabled.7 Williams & Wilkins 1991, pp 375-383 The hippocampal formation is one of the critical structures 2. Feldman RP, Alterman RL, Goodrich JT: Contemporary psycho- surgery and a look to the future. J Neurosurg 2001, 95(6): 944-956 for memory. This function of the hippocampal formation 3. Goldenberg PL: Functional neurosurgery. In: Schmidek HH, became understood because of an individual known in the Sweet WH (eds), Operative Neurosurgical Techniques: Indica- literature as H.M., who has been extensively studied by tions, Methods and Results, 2 Ed. Orlando, Grune & Stratton neuropsychologists. H.M. had surgery several decades ago 1988, Vol. 2, pp 1035-1068 4. Haberland C (ed): Acquired neurometabolic disease. In: Clinical for a valid therapeutic reason - the removal of an epileptic Neuropathology: Text and Color Atlas. New York, Demos Medical area in the temporal lobe of one side, which was the source Publishing 2007, pp 199-211 of intractable seizures. Most importantly, the surgeons did 5. Haberland C (ed): Basics of Neuropathology. In: Clinical Neuro- not know, and could not know according to the methods pathology: Text and Color Atlas. New York, Demos Medical Publishing 2007, pp 7-30 available at that time, that the contralateral hippocampal area 6. Haberland C (ed): Cerebral hypoxia. In: Clinical Neuropatho- was also severely damaged. This surgery occurred, unfortu- logy: Text and Color Atlas. New York, Demos Medical Publishing nately, before the functional contribution of this area to 2007, pp 33-41 memory formation was not known. Since the surgery, H.M. 7. Hendelman WJ (ed): The limbic system. In: Atlas of Functional Neuroanatomy, 2nd Ed. New York, CRC Press 2006, pp 202-238 has not been able to form any new memory for events or 8. Hogan RE: Mesial temporal sclerosis: Clinicopathological cor- facts, although he has been taught new motor skills (called relations. Arch Neurol 2001, 58(9): 1484-1486 procedural memory).7 9. MacLean PD: The limbic system (‘visceral brain’) and emo- tional behavior. Arch Neurol Psychiatry 1955, 73(2): 130-134 10. Papez JW: A proposed mechanism of emotion. Arch Neurol We now know that bilateral damage or removal of the ante- Psychiatry 1937, 38(4): 725-743 rior temporal lobe structures, including the amygdala and

80 PAN ARAB JOURNAL OF NEUROSURGERY Technical Note

Intraoperative localization of intracranial cavernomas by real time 3D ultrasonography: First experiences

Islam Aboulfetouh, NH Ulrich, Oliver Bozinov, Helmut Bertalanffy

Abstract Objective: The aim of this preliminary study was to test the feasibility of intraoperative real time 3D ultrasonography (US) in the prediction of brain cavernomas localization.

Patients and methods: During a time period of May to August 2009, 7 consecutive patients with intracranial cavernomas (3 supratentorial, 4 brainstem), were operated with the support of intraoperative true real time 3D US (iU 22, Philips, USA).

Results: The ultrasound images were achieved in 2 simultaneous orthogonal planes to find the exact site of corticotomy. Intraoperative true real time 3D US anatomically provided detailed information and good delineation of landmarks in 5 patients (all patients with supratentorial cavernomas and 2 patients with brainstem cavernomas) with a fair delineation in 2 patients with deep-seated brainstem cavernomas.

Conclusion: Our primary experience regarding this new technology suggests that intraoperative true real time 3D US is useful to localize cerebral cavernomas and less useful in localization of deep-seated brainstem cavernomas.

Key words: Brain cavernoma, image-guided neurosurgery and intraoperative 3D ultrasonography. (p81-85)

Introduction When surgery in eloquent area is intended, image guidance technology of conventional US, providing visualisation of allows the planning of the surgical approach and provides physiological and pathologic morphologic volumes of the anatomic orientation during dissection(4,5), particularly in brain.14 deep-seated cavernomas.1 Several studies have shown that intraoperative US is a reliable real time tool for assessing It is difficult for the neurosurgeon to understand and use tumour volume and defining tumour margins.8,10,16 US, most neurosurgeons are more familiar with MRI than US. Nonetheless, neurosurgeons who have started to use Intraoperative ultrasonography (iOUS) reduces operation intraoperative 3D US and want to explore its possibilities time by making it easier to locate the tumour; thus injury to can learn very quickly.27 normal cerebral tissue decreases with shorter operation 28 time. As 3D US continues to develop, the presence of real-time 3D US imaging equipment in the clinical setting will For a few years, 3D US has been available as an advanced expand and stimulate new areas of investigation and identify new areas where 3D US can further enhance clinical care.13 Demonstration of anatomic relationship is Neurosurgery Department improved by the multi-planar display. The 3D US is more Zurich University Hospital Zurich accurate in volume measurement, allowing an improved Switzerland assessment of size and shape of focal lesions, thus also improving follow-up.3,19,18 Correspondence: Dr. Islam Aboulfetouh Abdelaziz Neurosurgery Department During a time period from May to August 2009 a total of 7 Zurich University Hospital Frauenklinikstrasse 10 consecutive patients with intracranial cavernomas were CH-8091 Zurich examined intraoperatively by real time 3D US. It was the Switzerland Tel: (41 44) 255 2659 aim of this preliminary study to test the feasibility of real Fax: (41 44) 255 4505 time 3D US in intraoperative localization and detection of Email: [email protected] brain cavernomas.

VOLUME 14, NO. 2, OCTOBER 2010 81 3D ULTRASONOGRAPHY LOCALIZATION OF INTRACRANIAL CAVERNOMAS • Aboulfetouh, et al

Patients and methods interest by free-hand movement for 15 seconds. Clinical data Between May to August 2009 a preliminary study of 7 Orthogonal sets of images can be generated in any desired consecutive patients with brainstem cavernomas underwent plane through the 3D volume. In addition to the standard surgical resection at the Department of Neurosurgery at the sagittal and coronal planes when 2D US is performed, University Hospital of Zurich and were examined for intra- during 3D US, the brain can also be viewed in the axial operative localization with real time 3D iOUS (Philips iU plane. The 3D probe swings mechanically in a fan-like 22-). The main clinical features, the size and depth of caver- manner (Fig. 3) during volume acquisition, recording sets of noma to the surface of the patients are summarised in Table 1. tomograms at fixed angular increments, which are digital- ised and saved to the computer memory.

Table 1 - Patients summary. Pt Age Sex Localization Bleeding Quality of 3D Depth of the Size of the Treatment No. before lesion (mm) lesion (mm) 1 47 Female Brainstem + Good 37.4 13.5 × 9.4 Rt suboccipital pontine 2 25 Female with multiple Brainstem mid + Fair delineation 46.5 13.3 × 16.4 Median supracerebellar brain 3 64 Female associated with left cere- Brainstem + Good 17.8 6.9 × 11.6 Combined suboccipital bellopontine angle meningioma medullary 4 38 Male Left frontal - Good 7.7 11.5 × 12.3 Frontal craniotomy 5 35 Male with multiple cavernoma Left distal + Good 14.3 34.4 × 29.5 Temporal craniotomy with another cerebellar cavernoma Sylvian fissure 6 19 Male Brainstem + Fair delineation 41.1 12.3 × 8.4 Median suboccipital dorsal pontine 7 25 Male with multiple cavernoma Right frontal + Good 31.1 14.8 × 15 Frontal craniotomy operated before for brainstem cavernoma 2 years ago

There were 4 cases with brainstem cavernomas and 3 cases with supratentorial cavernomas.

Preoperative magnetic resonance imaging (MRI) was per- formed in all patients with T1-, T2-weighted and contrast- enhanced images. The maximum diameter of the cavernoma Figure 1 - 3D × 7-2 MHz probe. was determined on T1-weighted contrast-enhanced MRI and compared with sonographical measurements.

Neuronavigation was applied for preoperative localization and planning of the craniotomy in all patients. Surgery was performed by standard microsurgical technique, all patients were verified histopathologically.

Technique We used 3 types of probes for this ultrasound machine. Probe frequency for 2D C 8 - 5 MHz. Probe frequency 2D 15 - 7 io MHz for detection of lesions up to 3 cm depth. Probe frequency for 3D × 7 - 2 MHz, (Fig. 1) the ultrasound probe covered with aseptic sheath was placed directly on the surface of the dura mater exposed by craniotomy (Fig. 2). The distance to the cavernoma from different cortical Figure 2 - Intraoperative localization of brainstem medullary cavernoma by 3D probe. entry points based on cortical vascular and functional anat- omy were measured. No patient registration was needed for the 3D US volume. First the 2D probe was used to detect the lesion then the live During the pyramid-shaped 3D data when necessary, the 3D was utilised and when the first 3D US volume was maximum depth of the US image and the focus positions of acquired the probe was tilted over the anatomic area of the US beams were adjusted to obtain optimal image quality at

82 PAN ARAB JOURNAL OF NEUROSURGERY 3D ULTRASONOGRAPHY LOCALIZATION OF INTRACRANIAL CAVERNOMAS • Aboulfetouh, et al the cavernoma location. The measurement of cavernoma size and its distance from the dura alongwith the appearance of the cavernoma and surrounding tissues were noted. In addition, colour Doppler flow imaging was used to detect blood flow signals. We did not have any technical diffi- culties with the use of the probe.

Figure 4 - T2 MRI shows pontine cavernoma. Figure 5 - Shows 3D localization with good delineation and clear borders of pontine cavernoma (good image quality).

Figure 3 - Shows the 3 orthogonal planes of US probe.

Results Figure 6 - Shows intraoperative excision of pontine cavernoma. Most neurosurgeons are more familiar with MRI and computerised tomography than with US images. In the present study we found that it was important to be familiar with US imaging of the lesion.

There were 4 males and 3 females between the ages of 19 - 64; 4 cases with brainstem cavernomas and 3 with supraten- torial cavernoma, all 7 patients underwent surgery. Complete resection of all treated cavernoma was achieved. The depth of the pathological lesions varied between (7.7 - 46.5 mm). Figure 7 - MRI sagittal shows brainstem medullary cavernoma. The size of the vascular malformations ranged from (6.9 × Figure 8 - Shows 3D localization and good delineation and 11.6 mm to 34.4 × 29.5 mm). In all cases we used the real clear border of the medullary cavernoma. time 3D US for localization. Image quality was good in 5 cases; 2 with brainstem figure and all supratentorial caver- nomas, (Figs. 4 - 10). Good imaging quality was defined as exact cavernoma delineation with high degree of accuracy; US could also display the distribution of the vasculature of the lesion. Interpretation of US imaging modality of real time 3D US represents a good solution to the problem of brain shift in neuronavigation because it easily provides an updated, and hence more accurate, map of the patient’s true anatomy. The system enabled acquisition of 3D US data in diagnostic quality. Three-dimensional image-processing Figure 9 - MRI axial shows left distal Sylvian cavernoma. permits the analysis of US data interactively in 3 orthogonal Figure 10 - 3D localization and good delineation of the left distal Sylvian cavernoma. planes. Compared to conventional US 3D image analysis improved assessment of details, provided better spatial orientation and facilitated image interpretation. In the colour Doppler mode, the intraoperative US could

VOLUME 14, NO. 2, OCTOBER 2010 83 3D ULTRASONOGRAPHY LOCALIZATION OF INTRACRANIAL CAVERNOMAS • Aboulfetouh, et al also display the distribution of the vasculature of the lesion. the neuronavigation platform is performed by the calibra- Fair imaging in 2 patients of deep-seated brainstem caver- tion of an US probe. This must be completed either during nomas; patient number 2 (Figs. 11 and 12) and patient or before the surgery.23 number 6, (their depth were 46.5 mm and 41.1 mm), respectively. Fair imaging quality was considered to have Advantages been obtained in cases in which the cavernoma margins Our primary experiences with the use of real time 3D US in could not be delineated from the surrounding brain. We intraoperative localization of brainstem cavernomas is very found that image quality in these locations was not optimal. promising. There will be no need to correct the preoperative MR image mentally by the surgeons and there is no need for registration of the preoperative patient data and also no need for calibration of the US probe.

The integration of neuronavigation and intraoperative 3D US either with two hardware components reported by Unsguaard et al(26), or one platform navigation system reported by Tirakoti et al(24), have the drawback of the presence of the brain shift intraoperatively. Brain shift occurs not only in a linear fashion in one dimension but may also be curvilin- ear in 3D.24 Figure 11 - MRI T2 shows mid brain cavernoma. Figure 12 - 3D with fair delineation of mid brain cavernoma. The available real time 3D US probes make the possibility to work in real time imaging of good quality exciting and Discussion offers some new possibilities for the advance of neuro- surgery. Also real time 3D US eliminates the brain shift Cavernoma surgery is a special challenge since patients with 27 intracerebral cavernoma frequently show only minor or no problem completely. neurological symptoms at all. In addition, cavernoma are In our study, the use of the 3D real time US can eliminate often localized in the depth of the brain or in functionally the brain shift and also display the lesion clearly, and it can critical areas.1,2,21 be repeated from different angles to provide an updated and

more accurate map of the patient's anatomy and good The pathologic basis of cavernoma is a malformation of anatomic information. cerebral vessels, which can develop in any cerebral tissue at any time. Exploration to locate a small, deep cavernoma 28 As shown by Regelsberger et al, developments in US that can be very time-consuming. focus on 3D imaging and morphological aspects of the tumour associated blood supply thereby further optimising In the literature, it is meanwhile generally established that the operative strategies.17 The 3D allows a multiplanar some form of intraoperative localization technique should review thus showing pathology in critical sections not be available in the surgery of small, subcortical cavernoma achievable by 2D US and volume measurements are more to minimise surgical morbidity especially in deep and 12,15 accurate. This allows the detection of even subtle changes critical locations. during evolving disease.

Neuronavigation based on preoperative imaging can be of The 3D US also provides an ideal teaching tool, as one can limited use as there may be extensive brain shift during 20 4-6,22,25 virtually rescan the area and study any anatomic relation. surgery. These systems do not yet have the real-time 26 capability of US. Significantly shorter acquisition time of the 3D images and the availability of axial planes for diagnostic precision were Lunardi et al, reported that the main advantage of intra- considered to be major advantages of 3D US.7 operative sonographic guidance is the possibility to choose the dissection plane in relation to microanatomy of the The real benefit of 3D US is that it provides rapidly cortical surface and its vasculature and to measure depth of acquired three orthogonal plane information. The technique subcortical cavernoma.11 Two-dimensional US imaging is ideal for early identification of cavernoma and the without navigation guidance may be considered as a limited surgeon to formulate and modify an operative strategy for tool for image-guided neurosurgery because of its insuffi- attacking these lesions. cient data for anatomic orientation, especially when deep lesions are investigated.9 The integration of ultrasound into We believe that intraoperative 3D US might impact the

84 PAN ARAB JOURNAL OF NEUROSURGERY 3D ULTRASONOGRAPHY LOCALIZATION OF INTRACRANIAL CAVERNOMAS • Aboulfetouh, et al intraoperative management of brain cavernoma by providing tumors and determining the extent of resection: a comparative a real time image which enables immediate localization of study with magnetic resonance imaging. J Neurosurg 1996, 84 (5): 737-741 the cavernoma and avoid injury to normal cerebral tissues, 9. Jödicke A, Deinsberger W, Erbe H, Kriete A, Böker DK: Intra- which decreases with reduction of operation time. This will operative three-dimensional ultrasonography: An approach to eventually be useful to increase the safety of neurosurgical register brain shift using multidimensional image processing. procedures. All supratentorial cavernoma were well local- Minim Invasive Neurosurg 1998, 41(1): 13-19 10. LeRoux PD, Berger MS, Ojemann GA, Wang K, Mack LA: ized and the images with real time add more anatomical Correlation of intraoperative ultrasound tumour volumes and accuracy. margins with preoperative computerized tomography scans. An intraoperative method to enhance tumour resection. J Limitations Neurosurg 1989, 71: 691-698 11. Lunardi P, Acqui M: The echo-guided removal of cerebral cav- The fair quality in deep-seated brainstem cavernoma may ernous angiomas. Acta Neurochir (Wien) 1993, 123: 113-117 be because of improper positioning of the patient, the probe 12. McCormick PC, Michelsen WJ: Management of intracranial and depth of the lesion. It was difficult or impossible to cavernous and venous malformations. In: Barrow DL (ed), obtain good image with the real time 3D US. Ultrasono- Intracranial Vascular Malformations. AANS Publications Committee, Park Ridge 1990, pp 197-217 graphy did not provide sufficient information to visualize 13. Nelson TR: Three-dimensional ultrasound imaging. 3D/4D and assess deep-seated brainstem cavernoma in 2 cases. Ultrasound Imaging Meeting, UIA Annual Meeting 3/2006 14. Nelson TR, Pretorius DH: Three-dimensional ultrasound Nevertheless, this is a newly developed technique, with imaging. Ultrasound Med Biol 1998, 24: 1243-1270 15. Ojemann RG, Crowell RM, Ogilvy CS: Management of cranial relatively few cases and the technique needs further and spinal cavernous angioma. Clin Neurosurg 1993, 40: 98-123 evaluation concerning its limitation in detecting deep-seated 16. Quencer RM, Montalvo BM: Intraoperative cranial sonography. brainstem cavernomas and its role in detection of residual Neuroradiol 1986, 28: 528-550 cavernoma intraoperatively. 17. Regelsberger J, Lohmann F, Helmke K, Westphal M: Ultra- sound-guided surgery of deep seated brain lesions. Eu J Ultra 2000, 12(2): 115-121 Conclusion 18. Riccabona M: Use of three-dimensional ultrasound, harmonic In conclusion, intraoperative 3D US is a feasible new imaging, and echo-enhancing agents in neurosonography. J Ultrasound Med 2000, 19(3): S43 technology for the localization of brainstem cavernomas. 19. Riccabona M: Advanced techniques in paediatric cranial Our primary experience has demonstrated that it is less ultrasound. J Ultrasound Med 2002, 21: S90 useful with a fair image quality in localization of deep- 20. Riccabona M, Nelson TR, Weitzer C, Resch B, Pretorius DP: seated brainstem cavernomas. Potential of three-dimensional ultrasound in neonatal and paediatric neurosonography. Eur Radiol 2003, 13(9): 2082-2093 21. Robinson JR, Awad IA, Little JR: Natural history of the References cavernous angioma. J Neurosurg 1991, 75: 709-714 1. Bertalanffy H, Benes L, Miyazawa T, Alberti O, Seigel AM, Sure 22. Solheim O, Selbekk T, Lindseth F, Unsgard G: Navigated U: Cerebral cavernomas in the adult. Review of the literature resection of giant intracranial meningioma based on intra- and analysis of 72 surgically treated patients. Neurosurg Rev operative 3D ultrasound. Acta Neurochir (Wien) 2009, 151(9): 2002, 25: 1-53; Discussion 54-5 1143-1151 2. Bertalanffy H, Kühn G, Scheremet R, Seeger W: Indications for 23. Sure H, Benes L, Bozinov O, Woydt M, Tirakotai W, Bertalanffy surgery and prognosis in patients with cerebral cavernous H: Intraoperative landmarking of vascular anatomy by angiomas. Neurol Med Chir (Tokyo) 1992, 32(9): 659-666 integration of duplex and Doppler ultrasonography in image- 3. Fordham LA: 3D sonography in pediatrics. J Ultrasound Med guided surgery. Technical note. Surg Neurol 2005, 63(2): 133- 2002, 21: S38 41; Discussion 141-2 4. Germano IM, Villalobos H, Silvers A, Post KD: Clinical use of 24. Tirakotai W, Miller D, Heinze S, Benes L, Bertalanffy H, Sure the optical digitizer for intracranial neuronavigation. Neurosurg U: A novel platform for image-guided ultrasound. Neurosurg 1999, 45: 261-69; Discussion 269-70 2006, 58: 710-718 5. Golfinos JG, Fitzpatrick BC, Smith LR, Spetzler RF: Clinical 25. Tirakotai W, Sure U, Benes L, Krischek B, Bien S, Bertalanffy use of a frameless stereotactic arm: Results of 325 cases. J H: Image-guided transsylvian, transinsular approach for insular Neurosurg 1995, 83(2): 197-205 cavernous angiomas. Neurosurg 2003, 53(6): 1299-304; 6. Grumprecht HK, Widenka DC, Lumenta CB: BrainLab Discussion 1304-5 VectorVision Neuronavigation System: technology and clinical 26. Unsgaard G, Gronningsaeter A, Ommedal S, Nagelhus experience in 131 cases. Neurosurg 1999, 44(1): 97-104; Hernes TA: Brain operations guided by real-time two-dimen- Discussion 104-5 sional ultrasound: new possibilities as a result of improved 7. Haiden N, Klebermass K, Rücklinder E, Berger A, Prusa AR, image quality. Neurosurg 2002, 51(2): 402-11; Discussion 411-2 Rohrmeister K, Wandl-Vergesslich K, Kohlhauser-Vollmuth C: 27. Unsgaard G, Rygh OM, Selbekk T, Muller TB, Kolstad F, 3D ultrasonographic imaging of the cerebral ventricular system Lindseth F, Nagelhus Hernes TA: Intra-operative 3D ultrasound in very low birth weight infants. Ultrasound Med Biol 2005, 31 in neurosurgery. Acta Neurochir (Wien) 2006, 148(3): 235-253 (1): 7-14 28. Wang W, Wang Y, Dong Y, Wang Y: The value of intraopera- 8. Hammoud MA, Ligon BL, ElSouki R, Shi WM, Schomer DF, tive ultrasonography in neurosurgery. J Med Ultrasonics 2006, Sawaya R: Use of intraoperative ultrasound for localizing 33(1): 61-64

VOLUME 14, NO. 2, OCTOBER 2010 85 Case Review

Cavernous haemangioma of the skull

Mohammed Benzagmout1, Taoufiq Harmouch2, Afaf Amarti2, Khalid Chakour1, Mohammed El Faïz Chaoui1

Abstract: Calvarial cavernous haemangioma is a rare tumour, comprising about 0.2% of all benign neoplasms of the skull. The authors report the case of a 42-year-old woman admitted for a slow growing right parietal mass, hard to pressure, with freely mobile skin above the lesion. Cranial CT scan showed osteolytic lesion with erosion of the tabula externa. Surgery was performed with en bloc resection of the tumour completed by reconstruction of the osseous defect with methylmethacrylate. (p86-88)

Key words: Cavernous haemangioma, bone neoplasms, tumour and skull.

Introduction Skeletal cavernous haemangioma are uncommon tumours, The skull x-ray revealed a radiolucent osteolytic lesion in accounting for 0.7 - 1% of all bone neoplasms. The verte- the right parietal bone (Fig. 1). Computerised tomography bral column is most often affected, followed by the skull. (CT) scan in bony windows demonstrated a 3 cm osteolytic Calvarial cavernous haemangioma is rare, comprising about lesion with a totally intradiploic “honeycomb” pattern (Fig. 10% of all benign neoplasms of the skull.6,8,11 It arises from 2). The inner table of the bone was intact. Neither exo- nor the intrinsic vasculature of the bone and usually manifests endocranial evolvement was noticed. as a hard bony slow growing mass.10 In this work, we emphasize the rarity of this neoplasm and discuss epide- miological, clinical, radiological and therapeutical features of this tumour.

Case Report A 42-year-old woman, with unremarkable medical history, was hospitalized at our department for a small painless swelling of the right parietal bone. The mass appeared four years ago and increased progressively without any signs of intracranial hypertension or other associated symptoms. Physical examination revealed a right parietal painless firm mass of 4 cm × 3 cm dimensions. It had a regular edge and a smooth surface, and was fixed to the underlying bone. Figure 1 - Conventional radiograph of the skull in right lateral The overlying scalp was normal. The neurologic and view showing a radiolucent osteolytic lesion in the parietal bone. general examinations did not reveal any abnormalities.

1Department of Neurosurgery 2Department of Anatomopathology University Hospital Hassan II Fez Morocco

Correspondence: Dr. Mohammed Benzagmout Department of Neurosurgery University Hospital Hassan II BP 8589 Atlas 30003 Fez Morocco Tel: (212 61) 297 297 Figure 2 - Cranial CT scan in axial view, bony window, showing Fax: (212 35) 944 789 a lytic, expansile lesion with a honeycomb appearance in the Email: [email protected] right parietal bone.

86 PAN ARAB JOURNAL OF NEUROSURGERY CAVERNOUS HAEMANGIOMA OF THE SKULL • Benzagmout, et al

Surgery was performed with en bloc resection of the Discussion parietal tumour (Fig. 3). Then, the osseous defect was Intraosseous cavernous haemangioma are rare benign tu- reconstructed with methylmethacrylate. The postoperative mours of blood vessels, most commonly located in the course was uneventful and the histological examination spinal column followed by the skull. Firstly described by revealed thin-walled vascular channels lined by a single Toynbee in 1845, calvarial cavernous haemangioma layer of flattened endothelial cells interspersed among bony represent slow-growing lesions which typically occur in trabeculae, consistent with an intraosseous cavernous haem- women during the second through fourth decades, as in our angioma (Fig. 4). case. They are usually solitary.7,12,14,15 The parietal bone is thought to be most commonly affected, followed by the frontal bone and less frequently by the occipital and temporal bones.2

Clinical symptoms typically include pain and anaesthetic bulge. Indeed, the lesion usually manifests as a palpable hard bony slow growing mass, covered by normal skin.1 Neurologic deficits are unusual because the tumour tends to expand externally.11 However, depending on the site of the lesion, it can cause symptoms such as facial nerve paralysis and hearing loss in the temporal region(5,10), or visual loss and proptosis when involving the orbit.9

Plain x-rays identify the lesion as an extensive well- circumscribed area of rarefaction with a sunburst pattern of trabeculations radiating from a common centre. These characteristics are better defined on CT scan, especially for smaller lesions that are not discernible on conventional radiographs.11 Indeed, the CT scan demonstrates the lesion as an intradiploic lytic mass with a honeycomb or soap- bubble pattern. Moreover, the CT scan helps in planning surgery because it shows the site and extent of tumour better in bone windows.14 On MRI, the tumour usually appears mottled and heterogeneous containing increased and decreased signal intensities on both T1- and T2-weighted images, with typical enhancement after administration of gadolinium.3,11 Histologically, most of the calvarial haem- angioma are of the cavernous type.10 They are composed of Figure 3 - Intraoperative views showing the external (a) and groups of large dilated blood vessels separated by fibrous internal (b) aspects of the tumour after total removal. septa, as seen in our case.

The differential diagnosis of this tumour includes osteoma, aneurysmal bone cyst, giant cell tumour, fibrous dysplasia, meningioma, metastatic disease, Paget disease, dermoid and epidermoid cyst. All these tumours and others can mimic calvarial cavernous haemangioma, especially in smaller lesions and the diagnosis is usually made by histological study.

The treatment of choice for cranial haemangioma is surgical en bloc resection followed by .1,11 To prevent recurrence, removal of the lesion plus a 1 cm wide margin of uninvolved bone is always recommended. This method removes the tumour intact and remains the standard Figure 4 - Histological study showing the tumour after colora- approach. It also obviates the risk of significant bleeding tion by H&E (G x 10). during surgery because the sinusoids remain undisturbed.

VOLUME 14, NO. 2, OCTOBER 2010 87 CAVERNOUS HAEMANGIOMA OF THE SKULL • Benzagmout, et al

The other therapeutic options for these tumours are curet- gioma of the temporal bone. Arch Otolaryngol Head Neck tage and radiation therapy. Nevertheless, curettage has the Surg 1990, 116(8): 965-7 6. Gazzaz M, Maftah M, Akhaddar A, et al: Cavernous haeman- disadvantage of involving excessive blood loss and carries a gioma of the frontal bone. Pan Arab J Neurosurg 2002, 6(1): 3,13 higher risk of recurrence than total excision. Radiation 93-5 therapy may stop the tumour growth but does not reduce 7. Heckl S, Aschoff A, Kunze S: Cavernomas of the skull: review the size of the tumour and has a significant risk of of the literature 1975-2000. Neurosurg Rev 2002, 25: 56-62; 3,4,10,11 Discussion 66-7 malignant transformation. 8. Honda M, Toda K, Baba H, Yonekura M: Congenital cavernous angioma of the temporal bone: case report. Surg Neurol 2003, Conclusion 59(2): 120-3; Discussion 123 9. Hook SR, Font RL, McCrary JA, Harper RL: Intraosseous Skull cavernous haemangiomas are rare slow growing be- capillary hemangioma of the frontal bone. Am J Ophthalmol nign tumours. Gross total surgical excision is the optimal 1987, 103(6): 824-7 surgical treatment for this tumour to obtain a definitive di- 10. Khanam H, Lipper MH, Wolff CL, Lopes MB: Calvarial heman- agnosis and to avoid recurrence. giomas: report of two cases and review of the literature. Surg Neurol 2001, 55(1): 63-7; Discussion 67 11. Liu JK, Burger PC, Harnsberger HR, Couldwell WT: Primary References intraosseous skull base cavernous hemangioma: Case report. 1. Ajja A, Oukacha N, Gazzaz M et al: Cavernous hemangioma Skull Base 2003, 13(4): 219-28 of the parietal bone. A case report. J Neurosurg Sci 2005, 49 12. Peter C: Multiple calvarial hemangioma. Australian Radiol (4): 159-62; Discussion 162 2000, 44: 118-20 2. Banerji D, Inao S, Sugita K, Kaur A, Chhabra DK: Primary 13. Peterson DL, Murk SE, Story JL: Multifocal cavernous intraosseous orbital hemangioma: A case report and review of hemangioma of the skull: report of a case and review of the the literature. Neurosurg 1994, 35(6): 1131-4 literature. Neurosurg 1992, 30(5): 778-81; Discussion 782 3. Dogan S, Kocaeli H, Sahin S, Korfali E, Saraydaroglu O: Large 14. Suzuki Y, Ikeda H, Matsumoto K: Neuroradiological features of cavernous hemangioma of the frontal bone: Case report. intraosseous cavernous hemangioma: case report. Neurol Neurol Med Chir (Tokyo) 2005, 45(5): 264-7 Med Chir (Tokyo) 2001, 41(5): 279-82 4. Fredrickson JM, Haight JS, Noyek AM: Radiation-induced 15. Yoshida D, Sugisaki Y, Shimura T, Teramoto A: Cavernous carcinoma in a hemangioma. Otolaryngol Head Neck Surg hemangioma of the skull in a neonate. Child Nerv Syst 1999, 1979, 87(5): 584-6 15: 351-3 5. Gavilan J, Nistal M, Gavilan C, Calvo M: Ossifying heman-

88 PAN ARAB JOURNAL OF NEUROSURGERY Case Review

Haemorrhagic cerebral metastasis of alveolar soft part sarcoma

Mohamed Lmejjati1, Rhita Harifi1, Chakir Loqa1, Badia Belaabidia2, Said Ait Ben Ali1

Abstract Background: Christopherson et al, first described alveolar soft part sarcoma (ASPS) as a tumour of uncertain histogenesis.2 It is primarily a tumour in young adults with a female preponderance. This lesion generally affects the extremities or the head and neck region. It has a high propensity for distant metastasis that localises in the lung, bone and brain.

Methods: We report an uncommon case of ASPS in a young man which was revealed by haemorrhagic cerebral metastasis. The histologic characteristics and therapeutic difficulties of this lesion are described.

Results: A 22-year-old young patient with unremarkable past medical history was admitted in our institution for cerebral vascular accident with Glasgow Coma Scale (GCS) of 13/15. The computerised tomography (CT) scan revealed a right frontal haematoma. The diagnosis of arteriovenous malformation was discussed, however was normal. On general examination a paravertebral lumbar mass was diagnosed and operated. The histologic exam confirmed diagnosis of ASPS. Ten days later the patient presented with the same symptomatology with a GCS of 11/15. The CT scan showed multiple intracerebral lesions with one haemorrhagic in the right parietal lobe for which the patient was operated in emergency. Histologic exam revealed a cerebral metastasis of ASPS. The patient was referred for radiotherapy and chemotherapy. The outcome was characterised by the death of the patient 2 months later. (p89-91)

Conclusion: As in this case we should remember that full clinical examination is primary in this diagnosis.

Key words: Alveolar sarcoma, cerebral metastasis, tumour and metastasis.

Introduction Alveolar soft part sarcoma (ASPS) is a rare tumour ac- in the literature.7,12 counting for less than 1% of the sarcoma subtypes. It usually arises in the soft tissues of the extremities in young Case Report adults.15,16 Its histopathogenesis is unclear.5 Brain metas- A 22-year-old boy with unremarkable past medical history tases are unusual as the initial presentation. Reports of was admitted in our emergency for cerebral vascular accident these lesions presenting with haemorrhagic cerebral metas- with Glascow Coma Scale (GCS) of 13/15. Computerised tasis are infrequent and only two cases have been reported tomography (CT) scan taken revealed a right frontal haem- atoma (Fig. 1). Arteriovenous malformation was suspected but cerebral angiography taken appeared normal. Three days later, the patient was conscious without deficit and the 1Department of Neurosurgery general exam revealed a left paravertebral mass measuring 2Department of Anatomic Pathology 10 cm/5 cm, indolent without associated symptoms for University Hospital CHU Mohammed VI which the patient was operated confirming the diagnosis of Marrakesh ASPS. The tumour had an arachnoid pattern with variable Morocco alveolar structures circumscribed by thin vasculoconjunc- Correspondence: tival channels. Cytoplasm of the tumour cells contained Dr. Mohamed Lmejjati periodic acid schiff positive and granular material. Cyto- University Hospital, CHU Mohammed VI Faculty of Medicine keratin, vimentin, S-100 protein and glial fibrillary protein BP 7010, Sidi Abbad 40000 were negative (Fig. 2). Ten days after the patient presented Marrakesh Morocco again but with GCS of 11/15. The second CT scan showed Tel: (212) 61 30 67 61 / (212) 24 34 11 28 multiple intracerebral lesions and one that was haem- Email: [email protected] orrhagic in the right parietal lobe (Fig. 3a and b). The

VOLUME 14, NO. 2, OCTOBER 2010 89 HAEMORRHAGIC CEREBRAL METASTASIS OF ALVEOLAR SOFT PART SARCOMA • Lmejjati, et al

Figure 1 - Axial CT scan without contrast showed a right frontal Figure 2 - Photomicrograph of the lesion showing the lobe haematoma. variable alveolar structures circumscribed by thin vasculoconjunctival channels.

A B

Figure 3a - Axial CT scan without contrast showing a distinction of the right frontal haematoma lyzing to let a small nodule spontane- ously hyperdense surrounded by brain oedema and lesion in both parietal lobes (b). Right side with haematoma with midline shift to the left. patient was operated immediately. Histology confirmed the a recent discovery of cytogenitic abnormalities at 17q25.4,5,8 cerebral metastasis for ASPS. The patient was referred for radiotherapy and chemotherapy but the outcome was the The natural history of the tumour is deceptively indolent. death of the patient 2 months later. There is a propensity to develop metastases and these might even occur very late in the course of the disease. Discussion Alveolar soft part sarcoma was first described by Although it is a slow growing tumour alveolar soft part Christopherson et al, as a rare and distinct tumour of sarcoma has a high propensity for distant metastases, most uncertain histogenesis that usually affects the extremities in authors agree that distant metastases are common and young adults.2 According to Batsakis, 25% of all ASPS are associated with a high fatality rate.5 The most common found in the head and neck region and occur more sites of the metastases are lung (42%), bone (19%), brain frequently in young females (female to male ratio 2:1).5 (15%) and lymph nodes (7%), however, metastasis can Most cases have a primary site in the lower limbs and show occur anywhere.5,8 In another series, the incidence of brain right sided laterality, as described by Fassbender.3 The metastases was reported as 19% and always noted in histopathogenesis is uncertain but Ronald et al, hypothe- association with metastases to other sites.13 Frontal lobe sized that ASPS arise from displaced paraganglionic involvement is seen in more than 50% of cases reported.1,7,9 mesoderm and have a close homology with paragangliomas This was the case in our patient; the brain metastases was of the carotid body type.14 Most of the other studies revealed by VCA as primary manifestation of ASPS. Only indicate a muscle origin.10,11 The cytogenetics of this tumour two cases have been reported so far.15 is gradually being uncovered by some authors reporting trisomy for chromosome 7 and monosomy for chromo- There is no consensus on the optimum treatment. Most somes 8 and 18 as a constant feature.4,5 There has also been authors agree that adequate surgical resection usually guar-

90 PAN ARAB JOURNAL OF NEUROSURGERY HAEMORRHAGIC CEREBRAL METASTASIS OF ALVEOLAR SOFT PART SARCOMA • Lmejjati, et al antees good local control and remains the cornerstone for to the brain: Results of surgical treatment. Neurosurg 1994, 35 therapy.5,8,15 The addition of radiotherapy may further (1): 185-190; Discussion 190-191 5 2. Christopherson WM, Foote FW Jr, Stewart FW: Alveolar soft- enhance this, as was illustrated by Sherman et al. All six part sarcomas; structurally characteristic tumors of uncertain patients in Sherman’s study were reported to have excellent histogensis. Cancer 1952, 5(1): 100-111 local control with either adjuvant or preoperative radiation. 3. Fassbender HG: Das alveolär myoblastensarkom der skelett- However, the numbers are small and further studies are muskulatur. Oncologia 1960, 13(2): 184-191 4. Joyama S, Ueda T, Shimizu K, et al: Chromosome rearrange- warranted. In children radiation is indicated only if surgical ment at 17q25 and xp11.2 in alveolar soft-part sarcoma: A margins are positive or inadequate, and for unresectable case report and review of literature. Cancer 1999, 86(7): 1246- tumours.6 1250 5. Kanhere HA, Pai PS, Neeli SI, Kantharia R, Saoji RR, D’Cruz AK: Alveolar soft part sarcoma of the head and neck. Int J Oral Various chemotherapeutic agents including vincristine, Maxillofac Surg 2005, 34(3): 268-272 cyclophosphamide, actinomycin D and cytoxan have been 6. Kebudi R, Ayan I, Görgün O, Ağaoğlu FY, Vural S, Darendellier tried.5 Thiotepa is the drug known to cause regression.5 It E: Brain metastasis in pediatric extracranial solid tumors: is reported that metastatic ASPS is resistant to conventional Survey and literature review. J Neurooncol 2005, 71(1): 43-48 13 7. Lewis AJ: Sarcoma metastatic to the brain. Cancer 1988, 61 doxorubicin-based chemotherapy. As our patient’s condi- (3): 593-601 tion was rapidly deteriorating he was returned home to his 8. Lieberman PH, Foote FW Jr, Stewart FW, Berg JW: Alveolar family and died 2 months later. soft-part sarcoma. JAMA 1966, 198(10): 1047-1051 9. Lokich JJ: The management of cerebral metastasis. JAMA 1975, 234(7): 748-751 Conclusion 10. Miettinen M, Ekfors T: Alveolar soft part sarcoma. Immuno- Alveolar soft part sarcoma is a rare neoplasm which histochemical evidence for muscle cell differentiation. Am J Clin Pathol 1990, 93(1): 32-38 accounts for a significant proportion of sarcomas that 11. Mukai M, Torikata C, Iri H, et al: Histogenesis of alveolar soft metastasize to the brain and need to kept in mind when one part sarcoma. An Immunohistochemical and biomechanical encounters a case with typical histology, as in our case. study. Am J Surg Path 1986, 10(3): 212-218 Due to the rarity of the tumour a common consensus on the 12. Perry JR, Bilbao JM: Metastatic alveolar soft part sarcoma presenting as a dural-based cerebral mass. Neurosurg 1994, mode of treatment is difficult to achieve. Surgery remains 34(1): 168-170 the primary modality and local recurrence rates are low 13. Portera PA, Preyaskumar RP, Sorry WF, et al: Alveolar soft part following adequate excision. Adjuvant radiation therapy sarcoma. Cancer 2001, 91: 585-591 can be offered in cases with inadequate margins. Role of 14. Ronald AW, David MB, Frederick H, et al: Histopathogenesis of alveolar soft part sarcoma. Cancer 1972, 29: 191-204 chemotherapy is not yet defined and further studies 15. Sujit Kumar GS, Chacko G, Chacko AG, Rajshekhar V: addressing its use and its role in reducing distant metastasis Alveolar soft-part sarcoma presenting with multiple intracranial are needed. metastases. Neurol India 2004, 52(2): 257-258 16. Weiss SW, Goldblum JR (eds): Malignant soft tissue tumors of uncertain type. In: Enzinger and Weiss’s Soft Tissue Tumors, References 4th Ed. London, St Louis, Mosby 2001, Chapter 37, pp 1483- 1. Bindal RK, Sawaya RE, Leavens ME, et al: Sarcoma metastatic 1572

VOLUME 14, NO. 2, OCTOBER 2010 91 Case Review

Giant cerebral cavernous malformation

Abrar R Waliuddin, Bakur A Jamjoom, Ahmed Waliuddin, Abdulhakim B Jamjoom

Abstract: Cerebral cavernous malformations (CCMs) that are 6 cm or more in size have been loosely labeled as “giant” in the literature. Lesions of such size are rare with only 21 cases reported in the literature to date. We report a 27-year-old female patient who presented with raised intracranial pressure and focal neurological deficits which were related to a giant CCM. The pathology was documented by a preoperative MRI and confirmed by histopathological examination after the lesion was excised completely. A review of all the reported giant CCMs is carried out and it is hoped that the presentation will draw neurosurgeons attention to the clinical, radiological and histological features of giant CCMs and encourage a wider acceptance of giant CCMs as the subgroup of CCMs that are 6 cm or more in size.

Key words: Cerebral cavernous malformation, giant cavernous malformation cavernoma and cavernous angioma. (p92-95)

Introduction Cavernous malformations also known as cavernous angiomas, ness and forgetfulness. On examination she was drowsy and cavernous haemangiomas and cavernomas are congenital confused with mild left sided weakness including the face vascular malformations of unknown origin. They can be and bilateral mild papilloedema. Computed tomography found in the brain in 0.5% of population and are usually 1 - (CT) brain (Fig. 1) revealed a 7 × 5 cm non-contrast en- 75 mm in size.4,10,14,18 Unlike aneurysms and arteriovenous hancing mass in the right temporo-parietal region causing malformations (AVMs) which are defined as ‘giant’ by midline shift and obstructive hydrocephalus. The lesion had having diameters of 2.5 cm and 6 cm respectively, no such ill-defined borders but within it there were areas of hyper- threshold dimension has been approved for giant cerebral density representing bleeding and/or calcification with no cavernous malformations (CCMs). However, the term giant evidence of necrosis. In view of the patient’s depressed CCM has been used by some authors for lesions that were 6 level of consciousness and the presence of obstructive cm or more in size.3,12 Large CCM that would qualify as hydrocephalus we elected to treat the patient in the first giant, by being 6 cm or more in size, are rare with only 21 instance with a left ventriculoperitoneal shunt which was cases reported in the literature up to date.7,21 We report a done as an emergency. The patient improved following the female patient with a giant CCM who presented with raised shunt and she became alert and oriented. Magnetic intracranial pressure (ICP) and focal deficits. The aim of the resonance imaging (MRI) brain (Figs. 2a and b) demon- presentation is to draw the neurosurgeons attention to the strated that the non-enhancing lesion had a mixed signal clinical, radiological and histological features that are core of solid and multicystic components with areas of T2 characteristic of giant CCMs and to encourage a wider hypointensities suggestive of haemosidren and/or calcifi- recognition of giant CCMs as the subgroup of CCMs that cation. are 6 cm or more in size. The patient underwent a right temporo-parietal craniotomy Case Report and total excision of the lesion which was well-capsulated, A 27-year-old female patient presented to our neurosurgical multiseptated, avascular and containing dark brown fluid unit with a 2 months history of headache, vomiting, dizzi- (Fig. 3). Histopathological examination confirmed the mass to be a giant CCM (Fig. 4). The lesion appeared as a honey- comb of dilated vascular spaces containing organizing Section of Neurosurgery thrombi separated by fibrotic septae showing excess iron King Khalid National Guards Hospital deposits and a granulomatous reaction, with foreign body Jeddah Saudi Arabia type giant cells noted, without evidence of intervening brain tissue. Some of the vascular spaces have endothelium and Correspondence: Prof. Abdulhakim Jamjoom contain thin layers of smooth muscle fibres in their walls. Section of Neurosurgery King Khalid National Guards Hospital The patient made a good postoperative recovery with PO Box 9515 Jeddah 21423 improvement in her neurological status. A follow-up CT Saudi Arabia scan (Fig. 5) showed complete excision of the lesion. She Email: [email protected] remained well at 2 years follow-up.

92 PAN ARAB JOURNAL OF NEUROSURGERY GIANT CEREBRAL CAVERNOUS MALFORMATION • Jamjoom, et al

A B

Figure 1 ↑← CT brain (with contrast) showing the 7 × 5 cm non-enhancing giant CCM in the right temporo-parietal region causing midline shift and obstructive hydrocephalus. Figures 2 ↑→ MRI brain, axial T1 with contrast (a) and axial T2 with contrast (b), in which the non-enhancing giant CCM showed a mixed signal core of solid and multicystic components with areas of T2 hypointen- sities suggestive of haemosidren and/or calcification.

Figure 3 - Operative specimen of the giant CCM showing a well-capsulated, multiseptated lesion containing dark brown fluid. Figure 4 - Histopathological examination of the giant CCM (H&E stain) showing dilated vascular spaces containing organizing thrombi separated by fibrotic septae with excess iron deposits. Figure 5 - Follow-up plain CT scan showing complete excision of the giant CCM and collapsed ventricles.

Discussion Giant extra-axial intracranial cavernous angiomas are reported In this review we will adhere to the 6 cm or more size for in the scalp, pericranium, pituitary gland, middle cranial the definition of giant CCM while malformations less than fossa and cavernous sinus.23 In the brain, giant lesions are 6 cm will be referred to as usual size lesions. Giant CCM extremely rare with only 21 confirmed cases reported in the could have increased in size as a result of repetitive literature.7,21 We have reviewed all the reported giant CCMs intralesional haemorrhages in a usual size CCM, however that had histopathological and size verification in an effort the exact pathophysiology is still a matter of speculation. to highlight the clinical, diagnostic and management fea- Naturally it would be interesting to study such lesions for tures that are peculiar to the giant CCMs. their proliferative and neoangiogenic ability.1,21 Out of the 22 giant CCMs, including ours, one case was a 1- Cerebral cavernous malformation can be solitary or multi- day-old neonate, 5 were below the age of 1 year, 6 were ple and may appear as sporadic or in familial cases. aged between 1 year and 10 years and 3 cases in the second Pathologically they are defined as mulberry like assembly decade of life. The remaining cases were distributed in the of thin walled vascular sinusoids (caverns) lined by thin later decades of life thereby indicating that giant CCM is endothelium lacking smooth muscle and elastin without more common in the infants and paediatric age group, intervening brain parenchyma, surrounded by haemosiderin which is in striking contrast to the usual size CCM which is deposits and gliosis, which may or may not be throm- more common in the 3rd and 4th decade of life.19,21 Usual bosed.16 Although by definition the CCMs do not contain size CCMs do not show any predilection to either sex unlike intervening brain parenchyma as in our case, a number of the female sex preponderance we have observed among reported cases were found to contain intervening brain giant CCM.21 Nineteen out of the 22 cases were located in parenchyma, a finding that questions maintaining this fea- the supratentorial compartment and only 3 were seen in the ture as the essential histopathological criterion in the diag- cerebellum which is the generally accepted norm in the nosis of CCMs.5,17,20 Giant CCMs appear to have similar usual size CCMs.2,7,19,21 histological features compared to usual size CCMs except that the cystic characteristics are much more pronounced in Unlike giant CCMs, usual size lesions are frequently dis- the giant lesions.3,12 covered as incidental findings during investigations for

VOLUME 14, NO. 2, OCTOBER 2010 93 GIANT CEREBRAL CAVERNOUS MALFORMATION • Jamjoom, et al unrelated symptoms. About 11 - 44% of usual size CCMs influence the management. The association of venous mal- are reported to be asymptomatic and although symptoms formations have been observed in 10 - 30% of the CCMs depend partly on location, asymptomatic lesions have been (15,17) but was not recorded in any of the 22 gaint CCMs.7,21 found to be in all locations.21 Seizures are the most common manifestation found in about 60 - 70% as more CCMs are Whether the CCM is giant or usual size, the gold standard located in the supratentorial region.1,16 The other mode of of treatment when considered is complete surgical excision presentation is focal neurological deficits, which are the of the lesion.1,16 The indications for surgical treatment for result of mass effect exerted by the lesion itself due to intra- CCM in general can be classified according to the present- lesional or perilesional haemorrhage which is much more ing symptoms: seizures, haemorrhage or focal neurological pronounced in the brainstem. Of the 22 giant CCM cases, deficits. Surgical treatment is best reserved for supraten- the presenting complaint was seizures in 9 (41%), raised torial and some cerebellar CCMs. The brainstem CMs are ICP and neurological deterioration in 6 (27%) and focal best managed conservatively and followed-up with MRI deficits in 5 (23%). In addition, 2 lesions located in the scanning. In patients with seizures one accepted indication cerebellum presented with obstructive hydrocephalus.7,21 is intractable epilepsy. Some studies suggest that resection This is not surprising as the large size of giant CCMs make of the area of haemosiderin ring and the gliotic brain helps them more likely to present with raised ICP and focal control seizures while some have shown that by doing so deficits than usual size CCMs. there was no demonstrable change in epilepsy control.16

Although quite a few patients with CCMs complain of In patients who are symptomatic after haemorrhage with headache as a presenting symptom, the presentation with MRI evidence, surgery should be considered. When surgery overt haemorrhage is less common. The annual prospective is contemplated it is advisable to excise the venous angioma haemorrhage risk in patients with usual size CCMs has which coexists along with the CCM. The intralesional been estimated to be 0.25 - 6%.16 Risk factors for haem- haemorrhage is usually contained and even when extra- orrhage have been attributed to female sex, pregnancy, and lesional haemorrhage does occur it is not as significant as deep locations of CCMs such as, basal ganglia, thalamus, that occurring after rupture of an aneurysm or AVM.11 It is cerebellum and brainstem. Rarely patients present with interesting to note that some workers have recommended cranial nerve paralysis and hydrocephalus.2 Intracerebral prophylactic surgery for asymptomatic CCMs in women bleeding has not been reported in any of the giant CCMs. contemplating pregnancy as long as the lesion is accessible This could be related to the capsular thickening that is seen and in a non-eloquent area.16 In patients having progres- in giant CCMs or possibly the presumed reduction of intra- sively increasing neurological deficits surgery is indi- lesional pressure with the increase in size of the malformation. cated.16,21 Out of all the 22 reported giant CCMs, 18 had gross total excision and 2 had subtotal resection, one Although CCMs are strictly angiographically occult, an neonate was not operated and one had only biopsy. The area of avascular mass or evidence of neo-vascularisation outcome in operated cases was good and there were no can be seen occasionally on digital subtraction angiography. operative mortalities. Magnetic resonance imaging is more sensitive and specific than CT in detecting CCMs. Typical MRI findings of Stereotactic radiosurgery has been used in the treatment of CCMs are a well-defined lobulated lesion with a central selected usual size CCMs for a variety of indications with core of mixed signal intensities surrounded by a rim of conflicting reports regarding its efficacy and safety. The hypointensity signal, low T2-signal intensity in the rim is radiation induced neurological deficits still a remaining due to perilesional haemorrhages and the intralesional low challenge. Gamma knife surgery has shown some promise T2-intensity reflects calcification.15,22 Intra-axial CCMs do by reducing the size of the lesion particularly in locations not enhance significantly and it is this property of CCMs like brainstem and basal ganglia.8,13 Linac radiosurgery has which is used to differentiate them from AVMs. In a study also been claimed to be safe and showed lesion regression Pinker et al, demonstrated varying degrees of enhancement and obliteration of the tubules.6,9 Not surprisingly, radio- to gadolinium.15 Based on their findings they questioned the surgery has not been tried for giant CCMs due to their size significance of diagnosis of CCMs purely on the pattern of at the presentation and the danger to life posed by the mass enhancement and suggested that the criterion should be effect. Therefore surgical intervention with an aim at total based on morphological appearance and signal intensity extirpation remains the best modality. abnormalities. Out of 22 giant CCM cases, CT scan report records were available in 18 cases of which 9 (50%) showed Conclusion minimal to moderate enhancement. Magnetic resonance Neurosurgeons should be aware that CCMs of 6 cm or imaging contrast study is also useful to rule out the presence more are considered giant. They are rare with characteristic of an accompanying venous malformation which may imaging and pathological features. They are likely to pre-

94 PAN ARAB JOURNAL OF NEUROSURGERY GIANT CEREBRAL CAVERNOUS MALFORMATION • Jamjoom, et al sent with mass effect rather than bleeding and should be cavernous malformations. Neurosurg 2001, 48(1): 47-52 included in the differential diagnosis of intracranial space 12. Lawton MT, Vates GE, Quinones-Hinojosa A, McDonald WC, Marchuk DA, Young WL: Giant infiltrative cavernous malfor- occupying lesions. mation: clinical presentation, intervention and genetic analysis: case report. Neurosurg 2004, 55: 979-980 References 13. Liscak R, Vladyka V, Simonova G, Vymazal J, Norotny J Jr: Gamma knife surgery of brain cavernous hemangioma. J 1. Attar A, Ugur HC, Savas A, Yuceer N, Egemen N: Surgical Neurosurg 2005, 102: 207-13 treatment of intracranial cavernous angiomas. J Clin Neurosci 14. McCormick WF: Pathology of vascular malformations of the 2001, 8(3): 235-9 brain. In: Wilson CB, Stein BM (eds), Intracranial Arteriovenous 2. Avci E, Ozturk A, Baba F, Karabag H, Cakir A: Huge caver- Malformations. Baltimore, Williams & Wilkins 1984, pp 44-63 noma with massive intracerebral hemorrhage in a child. 15. Pinker K, Stavrou I, Knosp E, Trattnig S: Are cerebral caverno- Turkish Neurosurg 2007, 17(1): 23-26 mas truly nonenhancing lesions and thereby distinguishable 3. Clatterbuck RE, Moriarity JL, Elmaci I, Lee RR, Breiter SN, from arteriovenous malformations? MRI findings and histopa- Rigamonti D: Dynamic nature of cavernous malformations: a thological correlation. Magn Reson Imaging 2006, 24: 631-637 prospective magnetic resonance imaging study with volumetric 16. Raychaudhuri R, Batjer HH, Awad IA: Intracranial cavernous analysis. J Neurosurg 2000, 75: 782-785 angioma: a practical revive of clinical and biological aspects. 4. Delcurling Jr O, Kelly Jr DL, Elster AD, Craven TE: An analysis Surg Neurol 2005, 63: 319-328 of the natural history of cavernous angiomas. J Neurosurg 17. Rigamonti D, Johnson PC, Spetzler RF, et al: Cavernous 1991, 75: 702-8 malformations and capillary telangectasia: a spectrum within a 5. Frischer JM, Pipp I, Stavrou I, Trattnig S, Hainfellner JA, Knosp single pathological entity. Neurosurg 1991, 28: 60-4 E: Cerebral cavernous malformations: congruency of histo- 18. Robinson JR, Awad IA, Little JR: Natural history of cavernous pathological features with the current clinical definition. J angioma. J Neurosurg 1991, 75: 709-14 Neurol Neurosurg Psychiatry 2008, 79: 783 -788 19. Thiex R, Kruger R, Friese S, Gronewaller E, Kuker W: Giant 6. Huang YC, Yseng CK, Chang CN, Wei KC, Liao CC, Hsu PW: cavernoma of the brain stem: value of delayed MR imaging Linac radiosurgery for intracranial cavernous malformation: 10 after contrast injection. Eur Radiol 2003, 13(Suppl 4): L219-L 225 year experience. Clin Neurol Neurosurg 2006, 108(8): 750-6 20. Tomlinson FH, Houser DW, Scheithauer BW, et al: Angio- 7. Khan P, Tubay M, Osborn A, Blaser S, Couldwell WT: Radio- graphically occult vascular malformations: a correlative study graphic features of tumefactive giant cavernous angiomas. of features on magnetic resonance imaging and histological Acta Neurochir (Wien) 2008, 150: 49-55 examination. Neurosurg 1994, 34: 792-9 8. Ki MS, Pyo SY, Jeong YJ, Lee SI, Jung YT, Sim JH: Gamma 21. van Lindert EJ, Tan TC, Grotenhuis JA, Wesseling P: Giant knife surgery for intracranial cavernous hemangioma. J cavernous hemangiomas: report of three cases. Neurosurg Neurosurg 2005, 102: 102-6 Rev 2007, 30: 83-92 9. Kim DG, Choe WJ, Pack SH, Chung HT, Kim IJ, Han DH: 22. Zhao Y, Du GH, Wang YF, Wu JS, Xie LQ, Mao Y, Zhou LF: Radiosurgery for intracranial cavernous malformation. Acta Multiple intracranial cavernous malformations: clinical features Neurochir (Wien) 2002, 144(9): 869-78 and treatment. Surg Neurol 2007, 68: 493-499 10. Kim DS, Park YG, Choi JU, Chung SS, Lee KC: An analysis of 23. Zhou LF, Mao Y, Chen L: Diagnosis and surgical treatment of the natural history of cavernous malformations. Surg Neurol cavernous sinus hemangiomas: An experience of 20 cases. 1997, 48: 143-146 Surg Neurol 2003, 60: 31-8 11. Kuppersmith MJ, Kalish H, Epstein F, Yu G, Berenstein A, Woo H, Jafar J, Mandel G, De Lara F: Natural history of brainstem

VOLUME 14, NO. 2, OCTOBER 2010 95 Case Review

Spinal subdural haematoma following traumatic lumbar puncture in a patient with normal coagulation profile

Manish K Kasliwal, Deepak Agrawal, Bhawani Shanker Sharma

Abstract: Spinal subdural haematoma following lumbar puncture is a rare cause of spinal cord compression with very few cases reported in the literature. It is usually associated with some predisposing factors like haemorrhagic diathesis, anticoagulant therapy, vascular malformation, tumour or trauma. The authors report a case of spinal subdural haematoma in a 30-year-old male following a traumatic lumbar puncture with no obvious coagulation disorder. The patient had a poor outcome with minimal improvement in paraparesis at last follow-up, even after timely decompression. Although coagulation abnormalities are commonly found in most of these cases, our case illustrates the fact that spinal subdural haematoma can occur even in the presence of normal coagulation profile and a high index of suspicion with early diagnosis and intervention is warranted to prevent irreversible neurological deterioration, especially following a traumatic lumbar puncture. (p96-98)

Key words: Lumbar puncture, spinal subdural haematoma, anticoagulant therapy and paraplegia.

Introduction Spinal subdural haematoma following lumbar puncture is a bony injury. A diagnosis of traumatic cerebrospinal fluid rare cause of spinal cord compression with a very few cases (CSF) rhinorrhoea was made and a lumbar CSF drain was reported as per an extensive review of the literature by the inserted. Number 18 Tuhoy needle was used to perform the authors.1,3,6-9,11-20,22,2431,33 Most of the cases are usually asso- lumbar puncture at L3 - L4 level with slight difficulty in ciated with some predisposing factors like haemorrhagic performance of the procedure requiring four attempts to diathesis, anticoagulant therapy, vascular malformation, localise the space with the final tap being traumatic; the tumour or trauma.4,15 Regardless of the aetiology, early diag- CSF remained blood tinged for one hour. Cerebrospinal nosis with high index of suspicion is very important, as fluid examination revealed RBC 3000/microlitre, WBC 4/ delay in surgical intervention can have dire consequences. microlitre, predominantly lymphocytes, sugar 46 mg/dl We report a case of spinal subdural haematoma following a against a blood sugar of 99 mg/dl and proteins 35/dl. The traumatic lumbar puncture in a patient who did not have patient was kept under close neurological monitoring after any coagulation abnormality. the procedure. Six hours after the procedure, the patient developed paraparesis with asymmetrical weakness and Case Report sensory loss in both lower limbs and diminution of bilateral A 30-year-old male presented with headache and watery ankle and knee jerks. Emergent surgical decompression was discharge from left nostril 10 days after the occurrence of a performed after magnetic resonance imaging (MRI) re- road traffic accident with minor head injury with non- vealed a lumbar spinal subdural haematoma (Figs. 1a and b). contrast CT not showing any evidence of parenchymal or

Department of Neurosurgery Neurosciences Centre All India Institute of Medical Sciences New Delhi India

Correspondence: Dr. Deepak Agrawal Department of Neurosurgery Neurosciences Centre All India Institute of Medical Sciences New Delhi - 110029 Figures 1a - T2-weighted sagittal and India (b) axial MRI of the lumbar spine show- Tel: (91 986) 839 8242 ing the extensive subarachnoid and Fax: (91 112) 658 9650 subdural haematoma present anterior Email: [email protected] to the cauda equina.

96 PAN ARAB JOURNAL OF NEUROSURGERY TRAUMATIC LUMBAR PUNCTURE • Agrawal, et al

Magnetic resonance imaging did not reveal any other attributes the formation of spinal subdural haematoma to the associated lesion as a predisposing factor for the same with tracking of blood from the subarachnoid space to the the coagulation parameters being normal. (Platelets count subdural space. Since subarachnoid space consists of blood 2.6 lacs/microlitre, PT 12/14, INR 1.15, APTT 30/32, vessels which are subjected to fluctuations of pressure thrombin time 14/16). Puncture of the radiculomedullary transmitted from abdomen and thorax, a momentarily large vessels was thought to be the plausible aetiology for the fluctuation in pressure or a minor trauma can sometimes development of the haematoma. Though surgical evacua- cause these vessels to bleed resulting in subarachnoid blood tion was performed as soon as the diagnosis was confirmed which can then track into the subdural space forming a with postoperative imaging showing no residual haematoma subdural haematoma.5,21,23 (Fig. 2), the patient had only minimal improvement in power even at 3 months follow-up. Magnetic resonance imaging has now become the inves- tigation of choice and should be done in any patient developing new focal sensory or motor deficits following a lumbar puncture. A coagulation screen is also mandatory to rule out any underlying bleeding diathesis.

Although recovery is not guaranteed, emergent surgical decompression remains the cornerstone in the management Figure 2 - Postoperative CT lumber of this condition. There is only one case in the literature spine (sagittal reconstruction) showing where any neurological recovery occurred after 24 hours of complete evacuation of the clot. paraplegia, and even then, the extent of recovery of power was insufficient to overcome gravity.26 In fact when a subarachnoid clot accompanies the subdural haematoma, as was in our case, there have been no complete recoveries reported.3,13,25,29,32 The grim outcome occurs in spite of the fact that nerve roots are known to withstand compression for prolonged periods of time and points towards other mechanisms being involved. We hypothesise that the nerve Discussion roots could become ischaemic following thrombosis and/or Lumbar puncture has been used since antiquity for the vasospasm of the supplying radicular vessels in spinal diagnosis of meningitis by analysis of the CSF. It is subdural haematoma. Support for this hypothesis comes generally an innocuous procedure with post lumbar punc- from a report in which cauda equina infarct developed ture headache being the most common complication. following deep venous thrombosis.2 Cerebral and spinal herniation, cranial neuropathies, nerve root irritation, low back pain, stylet associated problems, Conclusions infections and bleeding complications are the other uncom- Spinal subdural haematoma is a rare complication of lumbar mon complications reported following a lumbar puncture.10 puncture and delay in diagnosis and management can be Spinal subdural haematoma complicating lumbar puncture catastrophic. Although coagulation abnormalities are is rare and most of these cases had been associated with commonly found in most of the cases, our case again some bleeding diathesis or anticoagulant therapy predispos- illustrates the fact that spinal subdural haematoma can occur ing the patient to risk of haematoma.21,33 Only 72 such in presence of normal coagulation profile. A traumatic tap cases have been reported, most being associated with some can be a harbinger of an underlying haematoma and such predisposing factors; the most common of which is an patients should be kept under close neurological monitoring associated coagulation abnormality.3 Domenicucci et al, to diagnose this rare complication. reviewed cases of spinal subdural haematomas and found haemostatic abnormalities in about 54% of patients.5 The References aetiopathogenesis of traumatic spinal subdural haematoma, 1. Anton E, Otegui A, Alonso A: Spinal subdural haematoma however, is uncertain and obscure. The lack of bridging complicating lumbar puncture. Rev Neurol 2000, 31(6): 597 2. Biesek D, Ksiazkiewicz B, Wanat-Slupska E: Conus medullaris veins in the spinal subdural space is thought to be the reason and cauda equina infarct in the course of thrombosis of deep 4 behind the rarity of spinal subdural haematomas. Never- veins of lower extremities. Pol Merkur Lekarski 2004, 17(99): theless, puncture of the radiculomedullary artery or vein of 273-4 Adamkiewicz, the only significant vessels in the subdural 3. Bills DC, Blumbergs P, North JB: Iatrogenic spinal subdural haematoma. Aust N Z J Surg 1991, 61(9): 703-6 space, remains the most plausible explanation for subdural 4. Bortolotti C, Wang H, Fraser K, Lanzino G: Subacute spinal 8 bleeding after lumbar puncture. Another hypothesis subdural hematoma after spontaneous resolution of cranial

VOLUME 14, NO. 2, OCTOBER 2010 97 TRAUMATIC LUMBAR PUNCTURE • Agrawal, et al

subdural hematoma: causal relationship or coincidence? repeated lumbar spinal puncture and drainage. J Trauma Case report. J Neurosurg 2004, 100(4 Suppl Spine): 372-4 1996, 40(4): 654-5 5. Domenicucci M, Ramieri A, Ciappetta P, Delfini R: Non- 20. Metzger G, Singbartl G: Spinal epidural hematoma following traumatic acute spinal subdural hematoma: report of five cases epidural anesthesia versus spontaneous spinal subdural and review of the literature. J Neurosurg 1999, 91(1 Suppl): hematoma. Two case reports. Acta Anaesthesiol Scand 1991, 65-73 35(2): 105-7 6. Dunn D, Dhopesh V, Mobini J: Spinal subdural hematoma: a 21. Miller DR, Ray A, Hourihan MD: Spinal subdural haematoma: possible hazard of lumbar puncture in an alcoholic. JAMA how relevant is the INR? Spinal Cord 2004, 42(8): 477-80 1979, 241(16): 1712-3 22. Pryle BJ, Carter JA, Cadoux-Hudson T: Delayed paraplegia 7. Durupt S, Durieu I, Nove-Josserand R, Raynal C, Levrat R, following spinal anaesthesia. Spinal subdural haematoma Vital Durand D: A rare complication of lumbar puncture: the following dural puncture with a 25 G pencil point needle at T12- spinal subdural hematoma. Rev Med Intern 2000, 21(2): 199- L1 in a patient taking aspirin. Anaesthesia 1996, 51(3): 263-5 200 23. Rader JP: Chronic subdural hematoma of the spinal cord: 8. Edelson RN, Chernik NL, Posner JB: Spinal subdural hem- report of a case. N Eng J Med 1955, 253(9): 374-6 atomas complicating lumbar puncture. Arch Neurol 1974, 31 24. Reina MA, López A, Benito-León J, Pulido P, María F: Intra- (2): 134-7 cranial and spinal subdural hematoma: a rare complication of 9. Egede LE, Moses H, Wang H: Spinal subdural hematoma: a epidural and subarachnoid anesthesia. Rev Esp Anestesiol rare complication of lumbar puncture. Case report and review Reanim 2004, 51(1): 28-39 of the literature. Md Med J 1999, 48(1): 15-7 25. Roscoe MW, Barrington TW: Acute spinal subdural hematoma. 10. Evans RW: Complications of lumbar puncture. Neurol Clin A case report and review of literature. Spine Spine. 1984, 9(7): 1998, 16(1): 83-105 672-5 11. Flores Torre M, Merino Angulo J, Gómez Jiménez F, Aguirre 26. Russell NA, Mangan MA: Acute spinal cord compression by Errasti C: Spinal subdural hematoma following lumbar puncture subarachnoid and subdural hematoma occurring in associa- in a patient with hemorrhagic diathesis. Med Clin (Barc) 1982, tion with brachial plexus avulsion. Case report. J Neurosurg 79(4): 199 1980, 52(3): 410-3 12. Gaspar P, Roux FX, Davous P, Rondot P: Spinal subdural 27. Spanu G, Berlanda P, Rodriguez y Baena R: Spinal subdural hematoma. An unusual complication of lumbar puncture. Sem haematoma: a rare complication of lumbar puncture. Case Hop 1982, 58(14): 873-5 report and review of the literature. Neurochirurgia (Stuttg) 13. Greensite FS, Katz J: Spinal subdural hematoma associated 1988, 31(5): 157-9 with attempted epidural anesthesia and subsequent continu- 28. Tekkok IH, Carter DA, Brinker R: Spinal subdural haematoma ous spinal anesthesia. Anesth Analg 1980, 59(1): 72-3 as a complication of immediate epidural blood patch. Can J 14. Guthikonda M, Schmidek HH, Wallman LJ, Snyder TM: Spinal Anaesth 1996, 43(3): 306-9 subdural hematoma: Case report and review of the literature. 29. Tomarken JL: Spinal subdural hematoma. Ann Emerg Med Neurosurg 1979, 5(5): 614-6 1985, 14(3): 261-3 15. Gutterman P: Acute spinal subdural hematoma following lumbar 30. Tomarken JL: Spinal subdural hematoma: a case report and puncture. Surg Neurol 1977, 7(6): 355-6 literature review. Am J Emerg Med 1987, 5(2): 123-5 16. Hasegawa H, Bitoh S, Obashi J, Ohtsuki H, Yamamoto T: 31. Vallee B, Besson G, Garre H, Monnerie JL, Le Guyader J: Spinal subdural hematoma following lumbar puncture. Case Acute spinal subdural hematoma at the resumption of report. Neurol Med Chir (Tokyo) 1985, 25(8): 687-90 anticoagulant treatment, interrupted for lumbar puncture. Nouv 17. Hurt RW, Shaw MD, Russell JA: Spinal subdural haematoma: Presse Med 1980, 9(45): 3457-8 an unusual complication of lumbar puncture. Surg Neurol 32. Vinters HV, Barnett HJ, Kaufmann JC: Subdural hematoma of 1977, 8(4): 296-7 the spinal cord and widespread subarachnoid hemorrhage 18. Kulkarni AV, Willinsky RA, Gray T, Cusimano MD: Serial complicating anticoagulant therapy. Stroke 1980, 11(5): 459- magnetic resonance imaging findings for a spontaneously 64 resolving spinal subdural hematoma: case report. Neurosurg 33. Wirtz PW, Bloem BR, van der Meer FJ, Brouwer OF: Para- 1998, 42(2): 398-400; Discussion 400-1 paresis after lumbar puncture in a male with leukemia. Pediatr 19. Lee JI, Hong SC, Shin HJ, Eoh W, Byun HS, Kim JH: Neurol 2000, 23(1): 67-8 Traumatic spinal subdural hematoma: rapid resolution after

98 PAN ARAB JOURNAL OF NEUROSURGERY Case Review

Complications in craniocerebral aspergillosis of sino-nasal origin in immunocompetent patients

Arshad A Siddiqui1, Saad H Bashir2, Ahmed Ali Shah3, Syed Ather Enam2

Abstract: Four immunocompetent patients of craniocerebral aspergillosis of sino-nasal origin who developed compli- cations have been described. They developed intracerebral haemorrhages, multifocal infarctions and intraventricular dissemination of aspergillus infection and 2 patients developed acute hydrocephalus. All 4 patients have been managed by standard therapeutic regimens including both surgical resection followed by antifungal therapy while CSF shunting was done as required. Only one patient survived till the last clinical follow-up. Magnetic resonance angiography along with routine magnetic resonance imaging at the time of initial work-up may be helpful to pick up mycotic aneurysms and vascular occlusions. (p99-103)

Key words: Aspergillosis, cerebrovascular complications, aspergillus hydrocephalus and immunocompetent hosts.

Introduction Craniocerebral invasive aspergillosis of sino-nasal origin is mode of spread, which occurs in patients with immunosup- a challenging clinical entity in the clinical practice.11 It has pression.2,6,7 Only a few anecdotal reports of complications been mostly described in immunocompromised patients with a of craniocerebral aspergillosis of sino-nasal origin in immuno- very high mortality of 85 - 100%.5 Invasive aspergillosis competent hosts have been described.5,17 We report 4 cases with their origin from nose and/or paranasal sinuses mainly that developed different complications in invasive asper- occurs in immunocompetent individuals with comparatively gillosis of sino-nasal origin in immunocompetent patients. lower mortality rate of 13 - 50%.3 In our recent clinical experience of craniocerebral aspergillosis of sino-nasal origin Case Report (14,16) in immunocompetent patients , we described anatomical Case 1: Thirty-five-year-old male presented with 6 months categorisation in correlation with clinical outcome and pre- history of right-sided facial numbness and headaches. On operative antifungal therapy with itraconazole, based on clinical examination, right-sided muscles of mastication 15 peculiar magnetic resonance imaging (MRI) features. showed atrophy. Pinprick sensation was absent in all the 3 divisions of trigeminal nerves, jaw jerk was also impaired. The cerebral complications due to angio-invasive predilection Rest of the clinical examination was unremarkable. On of aspergillosis have been described with haematogenous MRI (Fig. 1a), there was a mass lesion in the right middle cranial fossa extending posteriorly to the ipsilateral cerebel- lopontine angle. The lesion was isointense on T1-weighted

1Department of Neurosurgery images and hypointense on T2-weighted images with bright Hospital Sultanah Aminah Johor Bahru enhancement on contrast study. Mucosal enhancement was Johor also seen in the adjacent ethmoid and sphenoid sinuses Malaysia indicating the epicentre of lesion in the paranasal sinuses. 2Division of Neurosurgery Histopathological examination of the tissue from sphenoid The Aga Khan University Karachi sinus showed non-specific inflammation. To obtain a defi- Pakistan nite diagnosis, biopsy of the right middle cranial fossa mass

3Department of Neurosurgery was done by right temporo-parietal craniotomy. Histo- Civil Hospital pathology of this tissue revealed septate fungal hyphae, Karachi suggestive of diagnosis of aspergillosis, along with non- Pakistan caseating granulomatous inflammation with epithelioid and Correspondence: multinucleate giant cells. Patient was started on intravenous Dr. Arshad A Siddiqui th Department of Neurosurgery amphotericine-β deoxycholate (Fungizone). On 5 postop- Hospital Sultanah Aminah erative day, patient developed hemiparesis on right side Johor Bahru – 80100, Johor Malaysia (ipsilateral to surgical side) with significant drop in conscious Tel: (60 12) 739 1375 level. Repeat MRI (Fig. 1b) revealed multiple infarcts on Email: [email protected] both the side of surgery as well as contralateral to it,

VOLUME 14, NO. 2, OCTOBER 2010 99 COMPLICATIONS OF CRANIOCEREBRAL ASPERGILLOSIS OF SINO-NASAL ORIGIN • Siddiqui, et al

Figure 1a - Preoperative T1-weighted post-contrast axial image showing a contrast enhancing mass lesion involving the right paracav- ernous region with extension into the posterior cranial fossa. The lesion is also encasing the ICA and basilar arteries. Figure 1b - Postoperative coronal FLAIR image of the same patient after 5 days showing multiple areas of infarction involving the ipsilateral (to lesion) temporal lobe and contralateral basal ganglia and bilateral cerebellar hemispheres. Figure 2a - Preoperative T1-weighted post-gadolinium coronal image that shows a contrast enhancing mass lesion in the left paracavernous region with extension in the floor of middle cranial fossa. There is also involvement of adjacent sphenoid sinus. Figure 2b - Postoperative T1-weighted gadolinium- enhanced sagittal image showing dissemination of the aspergillosis into the posterior fossa, occipital lobes and into the lateral ventri- cles with enhancement of ventricular wall (indicative of ventriculitis). Figure 3a - Preoperative T1-weighted coronal post contrast image showing a mass lesion in left paracavernous region with extension in to the middle cranial fossa along evidence of infective process in the adjacent sphenoid sinus. Figure 3b - Postoperative CT scan brain axial section showing left frontal lobe haematoma with spillage of blood into the ventricles. There is postoperative temporal lobe changes related to the temporoparietal craniotomy. including the regions of basal ganglia and internal capsule decreased sensation on the left side of his face (trigeminal and in the cerebellum. The patient deteriorated despite distribution) with left-sided lateral rectus paresis (6th nerve administration of amphotericine-β and died of extensive palsy). Magnetic resonance imaging brain (Fig. 3a) re- cerebral damage on 18th postoperative day. vealed a mass lesion in the left paracavernous region, which was iso-intense on T1-weighted and hypo-intense on T2- Case 2: Forty-two-year-old male with no clinical co- weighted images and showed bright homogenous enhance- morbid conditions admitted with complaints of headaches ment on post-gadolinium study. Evidence of a similar and blurring of vision with diplopia to the left for the last 8 disease process was present in adjacent sphenoid sinus. months. On clinical examination, the patient had complete The patient underwent left pterional craniotomy and biopsy left ophthalmoplegia but visual acuity and fundoscopic of the tissue taken from the paracavernous mass revealed examination were normal. Rest of the neurological and aspergillosis on histopathology. This patient was treated systemic examination was also normal. Magnetic resonance with intravenous amphotericine-β (cumulative dosage of 3 imaging brain (Fig. 2a) revealed a contrast enhancing mass gms for 5 weeks), titrated according to clinical tolerance lesion in the left paracavernous region with extension in the and renal functions. Subsequently, patient was started on floor of middle cranial fossa. There was also mucosal oral itraconazole 400 mg per day. After 6 months, the thickening and enhancement in the sphenoid sinus. Histo- patient presented with sudden deterioration in conscious pathology of the biopsy (obtained from left paracavernous level. Magnetic resonance imaging brain (Fig. 3b) revealed mass by left pterional craniotomy) was reported to be an increase in the size of the previously operated left para- fungal infection suggestive of aspergillosis. The patient cavernous mass and dilatation of ventricles with periven- was started on intravenous amphotericine-β but his con- tricular enhancement. Contrast enhancing lesions were also scious level suddenly dropped on 3rd postoperative day. seen in the posterior fossa and left parietal lobe. The patient Computerised tomography (CT) scan of brain was done was started on intravenous amphotericine-β combined with which revealed left frontal haematoma remote from the intraventricular amphotericine-β after placement of an exter- surgical site (Fig. 2b). The patient was subsequently nal ventricular drain. Cerebrospinal fluid culture revealed a intubated and ventilated but he died of severe brain damage growth of aspergillus flavus. The patient showed no on 10th postoperative day. improvement in his clinical condition and in view of poor prognosis; patient had disseminated intracerebral and Case 3: Thirty-eight-year-old male with otherwise good intraventricular aspergillosis, the patient’s family decided health presented with left-sided facial pain and numbness against ventilatory support. The patient died of fulminant for the last 4 months. On clinical examination, he had intracranial aspergillosis on 11th day of admission.

100 PAN ARAB JOURNAL OF NEUROSURGERY COMPLICATIONS OF CRANIOCEREBRAL ASPERGILLOSIS OF SINO-NASAL ORIGIN • Siddiqui, et al

Case 4: Forty-year-old male was admitted with complaints ability to digest elastic tissue by release of the enzyme of headache and left ophthalmoplegia for the last 4 months. elastase that facilitates destruction of potential barriers to Computerised tomography scan of brain showed a contrast fungal infection.13 Many of the most effective barriers, enhancing mass lesion in the left paracavernous region. He including arterial walls contain significant elastin compo- was operated by temporal craniotomy for biopsy of the nents. Strains of aspergillus that produce elastase exhibit mass, which showed a fungal infection with septate hyphae much higher mortality rates in animal infection studies than suggestive of aspergillosis. The patient was treated with elastase negative strains.18 The increased invasiveness and intravenous amphotericine-β for 3 months with total dose of mortality associated with elastase producing aspergillus 2.5 gms given over a period of 4 weeks according to suggests that ability to cross elastic barrier contributes clinical tolerance and renal functions. Afterwards, the greatly to the organism virulence, including its ability to patient presented with progressively worsening headaches invade and infect the arterial wall. The tendency of associated with nausea and vomiting. Fundoscopy revealed aspergillus hyphae to intramural growth results in the bilateral papilloedema. Repeat CT scan showed gross dila- formation of mycotic aneurysm that, if not diagnosed, may tation of the ventricles. A ventriculoperitoneal shunt was become a source of intracerebral haemorrhage (ICH) or placed. Cerebrospinal fluid culture revealed aspergillus subarachnoid haemorrhage (SAH). Early diagnosis and flavus. The patient was started on oral itraconazole 400 management is the only way to avoid the devastating mgs per day and continued for 8 months. In his last follow- manifestations of ICH or SAH. We think that non-invasive up at one year, patient is alive and clinical examination imaging like MR angiography (done along with initial showed that patient still has visual disturbances but his preoperative imaging) might be helpful to identify these headaches and vomiting had improved. mycotic aneurysms which become a source of intracranial haemorrhages. Discussion Matsumura et al, described central nervous system (CNS) Cerebral infarctions also represents a characteristic aspergillosis as follows:9 complication of CNS vascular involvement of aspergillosis 1. Brain abscesses and granulomas vessel by invasion and extension of hyphae into the lumen 2. Meningitis and encephalitis which may cause insitu thrombosis or embolization of 3. Intracerebral haemorrhage hyphal masses.5,15,17 It is notable that 3 of the 4 cases who 4. Subarachnoid haemorrhage developed ischemic infarcts (Case 1), intracranial haemor- 5. Cerebral infarction foci rhage (Case 2) multifocal intracerebral dissemination with 6. Occlusion of a major artery. intraventricular extension (Case 3), located in the paracav- ernous region with involvement of cavernous sinus and in All these manifestations are described in intracranial close proximity to major vessels of circle of Willis. aspergillosis in immunocompromised patients which reach Interestingly, the ischemic infarcts were not seen in the the brain by haematogenous spread. All these compli- distribution of major parent vessels, rather these were deep- cations especially those involving angio-invasiveness are seated ischemic areas in the territory of small perforating extremely rare in immunocompetent hosts who have vessels. It is possible that diffuse intramural hyphal masses intracranial aspergillosis extending from nose and paranasal (producing thromboembolism) cause the perforating arteries sinuses.16 The criteria used for immunocompetence of to lose their patency early, due to their narrow diameters. patients are both clinical and radiological as described by Embolism of aspergillus hyphae in the cerebral vessels the same authors in their earlier review.16 All our 4 patients results in multiple infarctions and multifocal intracerebral had radiological evidence of disease process present in the dissemination. paranasal sinuses which had extended intracranially either by contiguous or by non-contiguous spread. These catastrophic manifestations of aspergillosis are usually described in immunocompromised patients, usually Two of our cases (Cases 1 and 2) developed cerebrovascular correlated with deranged immunological responses, but complications. Case 1 developed multiple intracerebral none of our patients was immunocompromised. The infarctions while Case 2 had intracerebral haemorrhage. studies of aspergillosis of sino-nasal origin in immuno- Haemorrhagic and ischemic cerebrovascular complications competent hosts are mainly from Sudan, Saudi Arabia, are directly related to angio-invasive nature of asper- India and Pakistan.11,12,14-16 This apparent prevalence in gillosis.4,18 immunocompetent host is thought to be due to tropical environment, bad hygiene and poor socioeconomic condi- The vascular invasiveness of aspergillosis is frequently seen tions in these regions.11,16 It is quite possible that some yet in the lungs, where pulmonary vascular invasion is well- undiscerned mechanisms of decreased resistance may be documented.8 This infectious vasculopathy is related to its operating.

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Two of our patients (Cases 3 and 4) developed hydro- tuberculous meningitic hydrocephalus with no peritoneal cephalus and both showed growth of aspergillus flavus on spread of infection (Table 1). culture. One patient was treated by temporary placement of external ventricular drain (for intraventricular amphotericine- Conclusion β) but others were treated with placement of permanent Haemorrhagic and ischemic cerebrovascular complications ventriculoperitoneal (VP) shunt. To our knowledge, only 2 may occur in immunocompetent hosts who have asper- cases of hydrocephalus have been reported till date. gillosis of sino-nasal origin. Disseminated intracerebral Mechanism of developing hydrocephalus is due to aspergillosis with ventriculitis is possibly due to angio- aspergillus meningitis resulting in communicating hydro- invasive spread. Non-communicating hydrocephalus is due cephalus.1,10 In Case 3, external ventricular drain was to meningitic complication of aspergillosis. Early diagnosis placed as there was MRI evidence of ventriculitis and and management may prevent these catastrophic manifesta- amphotericine-β was given this way. As there was no tions. Magnetic resonance angiography along with routine ventriculitis in Case 4, a VP shunt was placed despite MRI at the time of initial investigations may be helpful to growth of aspergillus flavus on culture. The rationale of pick up mycotic aneurysms and vascular occlusions. placing a VP shunt in this case is same as placing VP shunt Conventional angiography may be added accordingly, to in other chronic granulomatous infections like post- confirm and manage cerebrovascular complications.

Table 1 - Clinical summaries and complications in the four cases of craniocerebral aspergillosis of sino-nasal origin in immunocom- petent hosts. Sex/ age Clinical manifestations Location Surgery Complications Antifungal therapy (years) M/35 Headaches, right-sided facial Right middle cranial Craniotomy Multiple infarctions Intravenous amphotericin-β numbness, complete 5th nerve fossa with extension in palsy cerebellopontine angle M/42 Headaches, visual disturbances, Left paracavernous Craniotomy Intracerebral haemorrhage Intravenous amphotericin-β ophthalmoplegia M/38 Headaches, left-sided facial Left paracavernous Craniotomy - plus Multifocal intracranial Intravenous amphotericin-β numbness and trigeminal external ventricular dissemination with (plus intraventricular) plus neuralgia drain ventriculitis oral itraconazole M/40 Headaches, visual disturbances, Left paracavernous Ventriculoperitoneal Hydrocephalus Intravenous amphotericin-β ophthalmoplegia shunt plus itraconazole

References to cause pulmonary invasive aspergillosis in mice. Infect 1. Bryan CS, DiSalvo AF, Huffman LJ, Kaplan W, Kaufman L: Immun 1984, 43(1): 320-325 Communicating hydrocephalus caused by Aspergillus flavus. 9. Matsumura S, Sato S, Fujiwara H, Takamatsu H, Kajiwara T, South Med J 1980, 73(12): 1641-1644 Yamashiro K, Miyagawa A: Cerebral aspergillosis as a cerebral 2. DeLone DR, Goldstein RA, Petermann G, Salamat MS, Miles vascular accident. No To Shinkei 1988, 40(3): 225-232. JM, Knechtle SJ, Brown WB: Disseminated aspergillosis 10. Morrow R, Wong B, Finkelstein WE, Sternberg SS, Armstrong involving the brain: distribution and imaging characteristics. D: Aspergillosis of the cerebral ventricles in a heroin abuser. Am J Neuroradiol 1999, 20(9): 1597-1604 Case report and review of the literature. Arch Intern Med 1983, 3. Denning DW, Stevens DA: Antifungal and surgical treatment of 143(1): 161-184 invasive aspergillosis: review of 2,121 published cases. Rev 11. Murthy JM, Sundaram C, Prasad VS, Purohit AK, Rammurti S, Infect Dis 1990, 12(6): 1147-1201 Laxmi V: Aspergillosis of central nervous system: a study of 21 4. Endo T, Tominaga T, Konno H, Yoshimoto T: Fatal subarach- patients seen in a university hospital in south India. J Assoc noid hemorrhage, with brainstem and cerebellar infarction, Physicians India 2000, 48(7): 677-681 caused by Aspergillosis infection after cerebral aneurysm 12. Naim-Ur-Rahman, Jamjoom A, Al-Hedaithy SS, Jamjoom ZA, surgery: case report. Neurosurg 2002, 50(5): 1147-50; Discus- Al-Sohaibani MO, Aziz SA: Cranial and intracranial asper- sion 1150-1 gillosis of sino-nasal origin. Report of nine cases. Acta 5. Hurst RW, Judkins A, Bolger W, Chu A, Loevner LA: Mycotic Neurochir (Wien) 1996, 138(8): 944-950. aneurysm and cerebral infarction resulting from fungal sinusitis: 13. Rhodes JC, Bode RB, MacCaun-Kirsch CM: Elastase produc- imaging and pathological correlation. Am J Neuroradiol 2001, tion in clinical isolates of aspergillus. Diagn Micobiol Infect Dis 22(5): 858-863 1988, 10(3): 165-70 6. Jinkins JR, Siqueira E, Al-Kawi MZ: Cranial manifestations of 14. Shamim MS, Siddiqui AA, Enam SA, Shah AA, Jooma R, aspergillosis. Neuroradiol 1987, 29(2): 181-185 Anwar S: Craniocerebral aspergillosis in immunocompetent 7. Kitakami A, Nishizawa Y, Yamamoto S, Chiba M, Tuiki K, hosts: Surgical perspective. Neurol India 2007, 55(3): 274-281. Hasegawa H, et al: Two cases of cerebral aspergillosis with 15. Siddiqui AA, Bashir SH, Shah AA, Sajjad Z, Ahmed N, Jooma intracerebral hemorrhage. No Shinkei Geka 1988, 16(7): 863- R, Enam SA: Diagnostic MR imaging features of cranio-cere- 868 bral aspergillosis of sino-nasal origin in immunocompetent 8. Kothary M, Chase T Jr, Macmillan JD: Correlation of elastase patients. Acta Neurochir (Wien) 2006, 148(2): 155-166; Dis- production by some strains of Aspergillus fumigatus with ability cussion 166

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16. Siddiqui AA, Shah AA, Bashir SH: Craniocerebral aspergillosis artery causing a fatal subarachnoid hemorrhage. Intern Med of sinonasal origin in immunocompetent patients: clinical 1995, 34(6): 550-553 spectrum and outcome in 25 cases. Neurosurg 2004, 55(3): 18. Takahashi Y, Sugita Y, Maruiwa H, Hirohata M, Tokutomi T, 602-611; Discussion 611-3 Shigemori M: Fatal hemorrhage from rupture of the intracranial 17. Suzuki K, Iwabuchi N, Kuramochi S, Nakanoma J, Suzuki Y, internal carotid artery caused by aspergillus arteritis. Serizawa H, et al: Aspergillus aneurysm of middle cerebral Neurosurg Rev 1998, 21: 198-201

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Editorial 63-68 Percutaneous vertebroplasty for osteoporotic fracture of dorsolumbar and ix Message from PANS President Elect & 8th lumbar vertebra: Surgical technique and PANS Congress President early outcome Benaissa Abdennebi Ahmed Yehya, Abdelaziz El-Nekady x-xii Inside this Issue Retrospective Study

Review Articles 69-74 Complications and failures of endoscopic third ventriculostomy: Perception of their 1-11 Minimally invasive neurosurgery avoidance Allen L Ho, Peter M Black Yasser El Sawaf, Ibrahim Shafik, Reda Baza, Samy Torky 12-20 Microsurgical management of craniopharyngiomas Mohmammad-Yashar S Kalani, Kaith K Anatomical Study Almefty, Alim Mitha, Peter Nakaji, Robert F Spetzler 75-80 Papez circuit: An anatomical study by cadaveric dissection 21-28 Current management of traumatic Forhad Hossain Chowdhury, Akhlaque intracranial hypertension: A systematic Hossain Khan approach Mohammed H Bangash, Saleh S Baeesa Technical Note 29-37 Outcome predicting factors in intracranial aneurysms: Defining the complex aneurysms 81-85 Intraoperative localization of intracranial Raj Kumar, Vivek Kumar Vaid, Samir Kumar cavernomas by real time 3D Kalra, Sanjay Behari, Ashok Kumar Mahapatra ultrasonography: First experiences Islam Aboulfetouh, NH Ulrich, Oliver Bozinov, Helmut Bertalanffy Education & Training

38-45 Distal median- and ulnar nerve Case Reviews compression syndromes Jens Haase 86-88 Cavernous haemangioma of the skull Mohammed Benzagmout, Taoufiq Harmouch, Afaf Amarti, Khalid Chakour, Mohammed El Original Articles Faïz Chaoui

89-91 Haemorrhagic cerebral metastasis of 46-50 Experience with peripheral nerve injury in alveolar soft part sarcoma lower extremities Mohamed Lmejjati, Rhita Harifi, Chakir Loqa, Hatem Badr, Mohammed Kassem, Ahmed Badia Belaabidia, Said Ait Ben Ali Zaher, Mohammed Mansour, Ashraf Shaker

51-55 Percutaneous image guided lumbar disc 92-95 Giant cerebral cavernous malformation nucleoplasty: A minimal invasive Abrar R Waliuddin, Bakur A Jamjoom, technique for lumbar disc decompression Ahmed Waliuddin, Abdulhakim B Jamjoom

Khaled Saeed Ebrahim, Amr AlShehaby, Mohamed 96-98 Spinal subdural haematoma following A AlWardany, Ahmed Darwish, Mohamed traumatic lumbar puncture in a patient Awad with normal coagulation profile Manish K Kasliwal, Deepak Agrawal, Bhawani Shanker Sharma Clinical Studies 99-103 Complications in craniocerebral 56-62 Microsurgical excision of anterior and aspergillosis of sino-nasal origin in anterolateral intradural lesions of the immunocompetent patients foramen magnum Arshad A Siddiqui, Saad H Bashir, Ahmed Ali Hisham Aboul-Enein Shah, Syed Ather Enam

VOLUME 14, NO. 2, OCTOBER 2010 vii Contents

104-107 Intracranial hypotension 122-123 Neuropathological Feature Makarand Kulkarni, Sanjay Mongia, K Ibrahim S Tillawi Ravishankar, Vinay Chauhan, Manoj Deshmukh 124-126 Book Reviews 108-111 Extradural with intradural-extramedullary and intramedullary tuberculoma of the 127-128 Forthcoming Events spine without bony involvement Nigel Peter Symss, Goutham Cugati, Anil 129-130 Instructions to Authors Pande, Ravi Ramamurthi, MC Vasudevan 131 Subscription Form

Case Reports 132-1 — Abstracts in Arabic

112-114 Post-traumatic dorsal spinal extradural haematoma without osseous lesion Lamia Bencherif, Mohammed Benzagmout, Zidane Ihabe, Leila Mahfouf, Abdennebi Benaissa

115-116 Lumbar radicular pain caused by epidural varices Majid Reza Farrokhi, Hadi Niknam

117-118 Idiopathic lumbar epidural lipomatosis: A rare cause of lumbar canal stenosis Govindan Thiagarajan, Sivashanmugam Dhandapani

119-121 Pituitary apoplexy following open cholecystectomy Pralaya K Nayak, K Mohini Rao

viii PAN ARAB JOURNAL OF NEUROSURGERY

Inside This Issue

Minimally invasive neurosurgery: The concept of increasing the Neurosurgeon’s ability to the goal of tumour resection for better survival but simultaneously preserving as much normal tissue as possible. Utilising the tools of neuronavigation and functional imaging, e.g., fMRI, electrocortical stimulation, MRS, FDG and SPECT can also aid neurosurgeon discriminate between compromised and normal tissue. Endoscopic skull base surgery leads to reduced post- op discomfort and complications. It has increased the access of removal and controls the procedure, obviates brain retraction and minimise manipulation allowing gross total resection. Endovascular neurosurgery has achieved less invasive interventions and favourable outcomes. Enormous examples are given, e.g. stents, coiling, embolisation, angioplasty balloons which have allowed significant control of vascular diseases. Device developments have solved many complicated lesions and reduced their recurrence.

Microsurgical treatment of craniopharyngiomas: Several skull base approaches performed. We should feel comfortable to use the suitable technique according to the novel imaging modalities. Multidisciplinary team work assigned to provide patients with optimum treatment for their particular lesion and its anatomy. Approaches are described in concise detail such as transsphenoidal, endoscopic endonasal, interhemispheric, precallosal, transcallosal and orbitozygomatic.

Current management of traumatic intracranial hypertension: A systematic approach: Guidelines and recommendations are detailed for advocated treatments for TBI, including different monitoring; ABC’s of trauma victims to not only stabilise cardiopulmonary function but also preserve CBF to a compromised brain. Hypoxia, hypotension and disturbances of electrolytes and glucose can have detrimental effect on cerebral function. Intracranial pressure, A-line, CVP line, A-VO2 monitors are very helpful. Increase of TCD pulsatility index can identify states of decreased CPP. Electroencephalography and evoked potentials can provide early warning signs of neurologic changes. Brain stem indices are good prognosticators. Pre-hospital care and transportation to special trauma centres needs great emphasis.

Outcome predicting factor in intracranial aneurysms: Defining the complex aneurysm: Sixteen independent factors broadly categorised into 3 categories; patient related, radiology surgical factors had proved a significant correlation with the outcome, e.g. WFNS grade, clinical vasospasm, smoking, Fischer grade (CT scan distribution) and posterior circulation. Higher score would label the aneurysm as a complex. This is a very logic prognostic conclusion to guide how aggressive the treatment should be.

Distal median- and ulnar nerve compression syndromes: Meticulous and expert technique should be learnt and performed for these rewarding surgeries. Details are thoroughly described covering the subject. Clinical anatomy is a must for junior surgeons to achieve safe surgery.

Experience with peripheral nerve injury in lower extremities: Peripheral nerve lesions should be managed closely. History and clinical examination together with NCS are essential. Serial follow-up and special plan is drawn for each case on its own merits to avoid future disability. Grading of the lesion, timing of the surgery are important factors for better outcome. Type of surgery depends on whether the lesion is partial or complete. Earlier repair of transected nerves results in satisfactory outcome. Length of the graft is essential for functional recovery.

Percutaneous image guided lumbar disc nucleoplasty: A minimal invasive technique for lumbar disc decompression: Radiofrequency energy can ablate nuclear material and create small channels within the disc. Patients undergoing this procedure can experience relief of sciatica if well selected but long efficacy is yet to be seen. It is safe and simple and has short hospital stay.

Microsurgical excision of anterior and anterolateral intradural lesion of the foramen magnum: Far lateral approach to the skull base mandates 3D understanding of the specific anatomy of the region, tailoring the exposure in bloodless field intradurally is a good working angle with the aim of brain stem decompression strategy. Vertebral artery deserves special mobilization extradurally to avoid its injury as an important anatomic landmark. Drilling of the occipital condyle and ± jugular tubercle only needed when the tumour is entirely anterior to the medulla. Fortunately it will not affect the craniocervical instability.

x PAN ARAB JOURNAL OF NEUROSURGERY Inside this Issue

Percutaneous vertebroplasty for osteoporotic fracture of dorsolumbar and lumbar vertebra: Surgical technique and early outcome: It is an image-guided minimally invasive non-surgical procedure used to strengthen the fractured spinal vertebrae affected by osteoporosis. Fluoroscopy imaging screen is to make sure that cement mixture does not enter the spinal canal. It also relieves pain and improves mobility within 24 hours. Generally, it prevents further vertebral collapse by metastasis, myeloma and haemangioma.

Complications and failures of endoscopic third ventriculostomy: Perception of their avoidance: The steep learning curve is the key to reduce complications, namely; CSF leak, meningitis, transient DI, pseudomeningocele, pneumocephalus, haemorrhage and failures. Surgical technique is described and it is a simple procedure that could potentially have devastating complications and all precautions should be taken to avoid them.

Papez circuit: An anatomical study by cadaveric dissection: Periventricular lesions tackled in different surgical approaches necessitates knowledge of the microsurgical anatomy of the Papez circuit in order to avoid injuring the memory and emotional mechanisms of the limbic function and its psychological problems. This is an interesting dissection visualising the 3D imagination of the circuit. Follow the tracts and the connections (association fibres) to complete the circuit and study the surrounding structures through the bilateral hemispheric dissection.

Intraoperative localization of intracranial cavernomas by real time 3D ultrasonography: First experiences: Cerebral cavernomas can easily be delineated by 3D ultrasound intraoperatively. Unfortunately it is less useful in brain stem cavernomas. It shortens the OR time by providing anatomic orientation, tumour volume and defining tumour margins. It shows 3 orthogonal plane information. It is very helpful to avoid injury to normal brain. Neuro- navigation used to localise and plan the craniotomy. Limitations can be due to improper positioning of the probe and depth of the lesion.

Cavernous haemangioma of the skull: Case report of a rare benign tumour in the calvarium with erosion of the tabular externa. Craniectomy defect Is reconstructed with methlymethacrylate. Histology would be the key for the diagnosis to distinguish it from other hideous lesions.

Haemorrhagic cerebral metastasis of alveolar soft part sarcoma: Full clinical examination is the basic for teaching of medicine. Paravertebral lumbar mass revealed as alveolar soft part sarcoma of uncertain histogenesis. Brain metastasis is unusual as first presentation. Craniotomy confirmed the original lesion. Patient submitted to radio and chemotherapy but has fatal outcome. Alveolar soft part sarcoma came from displaced paraganglionic mesoderm and have close homology with paragangliomas of the carotid body. Other studies indicate muscle origin. Recent discovery of cytogenetic abnormalities at 17q25. They spread to lungs, bones, brain and lymph nodes.

Giant cerebral cavernous malformation: A case report of raised ICP and neurological deficit related to a cavernoma (confirmed histologically). Giant cavernoma should be accepted as a subgroup. Complete excision is a must for reversing deficit and improved headache. It’s size is attributed to proliferation and neoangiogenic ability. Can be simple or multiple, sporadic or familial. There is no intervening brain parenchyma with cystic characteristics. They can present as a mass effect rather than bleeding.

Spinal subdural haematoma following traumatic lumbar puncture in a patient with normal coagulation profile: Traumatic LP rarely causing SDH in absence of any coagulopathy. It may have significant morbidity despite urgent surgical evacuation. Puncture of the radiculopathy artery or vein of Adamkiewicz may explain the tragedy. Coagulation screen and MRI are important frontline investigations. Subarachnoid clot and SDH were a sign of poor prognosis for motor recovery.

Complications in craniocerebral aspergillosis of sino-nasal origin in immunocompetent patients: Immunocompromised patients are prone to this kind of fungal infection. They carry fatal prognosis because of dissemination and poor resistance. Their origin could be from nose or paranasal sinuses. Magnetic resonance imaging can give high index of suspicion to start antifungal therapy preoperatively. They spread through the circulation and they can cause brain abscess, meningitis, encephalitis, ICH, SAH, cerebral infarctions and occlusion of a major vessel (angioinvasive spread). Tropical environment, bad hygiene and poor socioeconomic conditions are indicative factors. Magnetic resonance angiography or conventional angiogram can pick up mycotic aneurysm and vascular occlusions.

VOLUME 14, NO. 2, OCTOBER 2010 xi Inside this Issue

Intracranial hypotension: Different aetiology and clinical presentations have been seen in this rare condition. It can be post traumatic, post spinal surgical, ruptured perineural cyst in the pleura or just spontaneous. Bed rest, hydration and oral theophylline are among the conservative treatments. Transsphenoidal surgical repair was indicated for fracture middle fossa base. Classical MRI appearance of this subdural collection. Autologous blood patch in the spinal epidural space is very effective in sealing the leak.

Extradural with intradural-extramedullary and intramedullary tuberculoma of the spine without bony involvement: A case report of cerebral and spinal cord TB that needed twice to excise the lesion from intramedullary, extramedullary and intradural sites. The peculiar finding was the absence of any bony involvement. Mycobacterium TB can involve neural and perineural tissues. Magnetic resonance imaging with contrast plays an important role in diagnosis. Surgery confirms the diagnosis with the continuation of anti-TB therapy and steroids.

Post-traumatic dorsal spine extradural haematoma without osseous lesion: Athletes are prone to extradural venous bleeding without any coagulopathy. Late after trauma the haematoma enlarged and caused pain and paraparesis. Increased venous pressure led to accumulation of blood that caused spinal compression. Absence of bony injury is attributed to the laxity of the ligaments. Magnetic resonance imaging was the key investigation. Post-op recovery was excellent. The main prognostic factor is the duration between trauma and cord compression, the degree of medullary compression and the timing of the surgery.

Lumbar radicular pain caused by epidural varices: Acute sciatica can be caused by some vascular malformations in the absence of lumbar disc or canal stenosis. Varicose vein was seen at surgery compressing the nerve root. Other causes could be due to vertebral haemangioma with epidural extension, epidural cavernous haemangioma and epidural AVM. Increased epidural venous pressure can cause acute enlargement of the varices. Magnetic resonance imaging, abdominal sonography is recommended to rule out vascular malformations or venous thrombosis.

Idiopathic lumbar epidural lipomatosis: A rare cause of lumbar canal stenosis: Long term use of steroids and obesity are associated with this rare cause of lumbar canal stenosis. Some are truly idiopathic. There is definite male preponderance. Magnetic resonance imaging is the investigation of choice. The compression may be from the fat itself or due to engorged veins.

Pituitary apoplexy following open cholecystectomy: Severe headache, deterioration of conscious level and visual disturbances after GA for general surgery is quite a strange presentation. Urgent CT or MRI brain is mandatory. There were no previous symptoms related to the pituitary gland. Intratumoural haemorrhage with mass effect is the cornerstone for diagnosis. Third nerve palsy, seizures and hemiplegia can be associated signs and hypertension may be a predisposing factor. Emergency transsphenoidal surgery is performed to save vision within 1 week of the visual symptoms.

Dr. Mohamed Abdulrahim, FRCS Consultant Neurosurgeon Division of Neurosurgery Department of Clinical Neurosciences Riyadh Military Hospital Saudi Arabia

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