Letters to the Editor 869

The MR of the brainstem showed a small A CURT of the herpes group. A strong signal was V DIETZ on ethidium J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.57.7.869 on 1 July 1994. Downloaded from ischaemic lesion in the left dorsolateral Swiss Paraplegic Centre, obtained bromide stained gel. medulla without any further pathology. University Hospital, Balgrist, Characterisation of HSV-2 DNA was Until confirmation of the total occlusion CH-8008 Zurich, achieved by restriction analysis of the of the left vertebral artery the patient was Switzerland amplified product. a-Interferon in CSF was treated with cumarine to prevent arterial Correspondence to: V Dietz, Paraplegic Centre, normal. The patient had no history of embolism. The clinical symptoms progres- Balgrist, Forchstrasse 340, CH-8008 Zurich, recurrent herpes genitalis. There was no sively disappeared over the next 30 days Switzerland. serological evidence for borreliosis, HIV-1 and the patient left hospital with only a 1 Hart RG. Vertebral artery dissection. or HIV-2, HTLV-I, Q-fever, listeriosis, slight ataxic gait and mild dysaesthesia Neurology 1988;38:987-9. cytomegalovirus, measles, varicella zoster, within the left C6 segment. 2 Fakhry SM, Jaques PF, Proctor HJ. Cervical or Epstein-Barr virus infection. CD4 counts vessel injury after blunt trauma. Jf Vasc Surg 1988;8:501-8. were normal and no cause for immuno- Discussion 3 Parent AD, Harkey HL, Touchstone DA, depression could be identified. The patient presented with an anterior dis- Smith EE, Smith RR. Lateral cervical spine Parenteral acyclovir (30 mg/kg daily) was placement and fracture of the left facet joint dislocation and vertebral artery injury. given for 10 days and the patient's neuro- Neurosurgery 1992;31:501-9. between level C5 and C6 and radicular pain 4 Louw JA, Mafoyane NA, Small B, Neser CP. logical state remained unchanged. Six days in the left C6 distribution, and subsequently Occlusion of the vertebral artery in cervical after admission, sparse vesicular lesions developed a left vertebral artery dissection spine dislocations. J Bone Joint Surg 1990; appeared on the patient's buttocks, internal with left brainstem ischaemia. It is 72B:679-81. suspect- 5 Jabre A. Subintimal dissection of the vertebral aspects of the thighs, and lower part of the ed that the dissection occurred during the artery in subluxation of the cervical spine. abdomen. Ten days later, numbness in both 19 day delay with subsequent occlusion Neurosurgery 1991;29:912-5. hands appeared. Examination showed bilat- including the posterior infracerebellar eral arm and shoulder weakness and the artery. By contrast with our case, most disappearance of upper limb reflexes. The reports during the past decade concerning patient then became confused and drowsy, posterior circulation ischaemia connected Herpes simplex virus type 2 ascending developed hyponatraemia and hyperazo- with vertebral artery occlusion occurred myeloradiculitis: 1IU findings and taemia and died on day 21 after admission. spontaneously or were loosely related to rapid diagnosis by the polymerase Necropsy examination was not performed. minor trauma during sport or neck manipu- chain method Subsequently, CSF cultures were reported lation during chiropraxy. These typically as positive for HSV-2. Analysis of serial occurred in young or middle aged adults, Although neurotropic viruses are often sus- serum samples showed a sevenfold rise in equally between the sexes.' Although trau- pected of causing spinal cord injuries, con- anti-HSV antibodies between admission matic fractures of the cervical spine occur firmation by early diagnosis is difficult. and death. IgM antibodies were detected in often, injuries of the vertebral arteries with Ascending myelitis related to herpes sim- one early serum sample. Analysis of the or without clinical symptoms are rarely evi- plex virus type 2 (HSV-2) infection has sel- CSF and serum anti-HSV antibodies ratio dent. The incidence is postulated to be dom been reported and the diagnosis could showed the existence of specific intrathecal between 3% and 10%.2 be established only at postmortem examina- synthesis. In a retrospective study Parent and tion. 1-5 We report the case of an elderly Various clinical syndromes have been coworkers reviewed some 640 patients sus- woman with a subacute ascending myelo- linked to HSV-2 involvement in the nervous taining fractures of the cervical spine. Of radiculitis. MRI showed spinal cord and system. HSV-2 encephalitis typically occurs these, 96 had facet involvement and in only sacral root involvement and the polymerase in the newborn, but accounts for less than five was injury of the vertebral artery diag- chain reaction allowed the rapid identifica- 5% of herpetic encephalitis in children and nosed by initial major neurological deficits tion of HSV-2 DNA in the CSF. adults. Acute, self limited meningitis is such as cerebellar infarction, cortical blind- A 76 year old woman was referred found in young adults with primary genital ness, or pontine infarction, which have been because of urinary retention and parapare- HSV infection. Sacral radiculitis with per- documented by postmortem examination in sis. Three weeks previously, she had noted ineal dysaesthesias, autonomic dysfunction, two cases.3 All these patients had cervical the progressive onset of anorexia, fever and sometimes mild lower limb weakness fractures located at C5-C6 and in one case (38°C), weight loss (4 kg), and low back may also be associated with herpes genitalis in combination with a fracture at C6-C7. pain. Evaluation performed in another hos- in young adults. In most cases, neurological Radiographs showed anterior dislocation at pital showed negative bacterial symptoms occur two to seven after the cultures days http://jnnp.bmj.com/ C5-C6 in four cases and at C6-C7 in one from blood and urine and CT of the thorax, genital eruption and patients recover within case. Bilateral facet fractures were evident abdomen, and lumbosacral spine was nor- three weeks. in four cases. mal. Three days before admission she com- By contrast, HSV-2 involvement in the Only one prospective study exists that plained of right sciatalgia and rapidly spinal cord is rare and severe. Two cases of considers the combination of facet joint dis- developed lower extremity weakness and HSV-2 extensive myeloradiculitis have been location of the cervical spine, the incidence sphincter disturbances. reported in patients with AIDS simultane- of vertebral artery injury, and the neurologi- Neurological examination showed a flac- ously infected with cytomegalovirus,' 2 and cal deficit.4 From 12 consecutively exam- cid paraplegia, a TI0 sensory level, and a two others in diabetic patients.34 HSV-2

ined patients with facet joint dislocation distended bladder. Deep tendon reflexes necrotising myelopathy has also been found on September 30, 2021 by guest. Protected copyright. (C5/C6 in seven, C6/C7 in three, and were absent in the lower limbs and plantar in association with malignancy.5 In all cases C4/C5 in two) nine showed an occlusion of responses were both extensor. In the upper the outcome was fatal within four to seven at least one vertebral artery. Of these nine extremities strength was normal but reflexes weeks, and the diagnosis could only be patients only two with bilateral facet joint were brisk and a bilateral Hoffman sign was made at necropsy, when HSV-2 was recov- dislocations had a transient neurological noted. Mental state and cranial nerves were ered from the spinal cord. There has been a deficit. Further indications of a traumatic or normal. single report of HSV-2 myelitis with a spontaneous dissection of the vertebral Non-enhanced TI weighted images of favourable outcome in which the virus was artery are neck pain and symptoms of a C6 the spine were normal. T2 weighted isolated from the CSF.6 root compression.5 sequences showed a linear hyperintense sig- In our patient, MRI clearly showed Thus a combination of neurological and nal at the T10 level and within the conus myeloradiculitis, on T2 weighted and radiological findings could lead to an early medullaris. TI weighted images with con- gadolinium enhanced TI weighted sagittal diagnosis and may indicate development of trast injection showed an enhancement of sequences. Although well correlated with a dissection of the vertebral artery. We sug- both the posterior meninges and the roots the clinical features, however, MRI findings gest that patients with the clinical symptoms of the cauda equina. lacked specificity. and type of injury described here are prone Her CSF contained 73 leucocytes/mm3 The direct diagnosis of nervous system to development of a dissection or occlusion (97% lymphocytes), 132 mg/dl protein and infection by HSV is difficult as isolation of of the vertebral artery. Early diagnostic pro- 68 mg/dl glucose. Electrophoresis of CSF the virus from CSF is most often unsuccess- cedures by non-invasive diagnostic tech- protein showed 26% y-globulins with an ful and serological confirmation is too late. niques such as MR angiography and oligoclonal distribution and a raised Recently, the polymerase chain reaction has ultrasound techniques coupled with treat- IgG/albumin ratio (0 56; normal <0 25). A proved to be a powerful tool in the rapid ment at the onset of a possible dissection polymerase chain reaction was performed diagnosis of meningoencephalitis due to may help to prevent the formation of a on CSF with a pair of primers that allowed herpes viruses. In this case, it allowed us to microembolism or arterial occlusion. the simultaneous detection of four viruses identify HSV-2 in the CSF immediately 870 Letters to the Editor after admission, a week before the appear- ism. An MRI showed a unilateral infarction nosis due to unilateral lesion. Although the ance of the cutaneous eruption. involving the left cerebral peduncle. precise anatomical basis for peduncular hal- J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.57.7.869 on 1 July 1994. Downloaded from The pathogenesis of HSV-2 myelitis in A previously healthy 70 year old right lucinosis remains unclear, it seems that the this case remains ill defined. Although IgM handed woman presented with visual hallu- substantia nigra pars reticulata (SNpr) is antibodies were detected in the first serum cinations. Two months previously, she directly implicated.3 Little is known about sample we are not sure that the patient's began, one night, to see objects (motor- the pathogenesis of peduncular hallucinosis. myeloradiculitis reflected true HSV-2 pri- bikes), animals (dogs, horses), and people A relation with the rapid eye movement mary infection because reappearance of (Japanese) entering and driving silently (REM) phase of sleep has been proposed. IgM antibodies can be detected in herpes round her room, across the entire visual In this sense, it is known that the SNpr may virus reactivation. All but one preriously field. Although the images were of normal play an important part in the regulation of reported case of HSV-2 myelitis were fatal colours and sizes, she was aware that they the different phases of sleep through its and occurred in immunocompromised were not real, and never described "deja connections with centromedian/parafascicu- hosts. This patient was neither diabetic nor vu" or "jamais vu" phenomena, tactile, or lar nuclei of the thalamus, superior collicu- HIV-infected, but despite normal CT auditory hallucinosis. The hallucinatory lus, and reticular formation.4 examinations of the thorax and abdomen, a events became progressively longer and Finally, although we cannot rule out the hidden malignancy cannot be excluded. more frequent, lasting from minutes to presence of brainstem Lewy bodies, it is Reactivation of a latent infection within hours, during both day and night. Her probable that the right hemiparkinsonism dorsal root ganglia neurons with a contigu- medical history included mild hypertension, found in our patient was related to ous spread via sacral ganglia to the spinal normofunctional multinodular goitre, and ischaemic damage of the left substantia cord has already been proposed in HSV-2 surgery for left breast carcinoma six years nigra pars compacta.5 ascending myelitis.' It is of note that despite earlier, without subsequent evidence of RAUL DE LA FUENTE FERNANDEZ antiviral treatment, our patient died within recurrence. There were no other remarkable JOSE MAREY LOPEZ six weeks. Thus an immunologically personal or familial antecedents. On exami- PABLO REY DEL CORRAL mediated injury triggered by the herpes nation, she was alert, oriented, and cooper- Service ofNeurology infection could also be involved here, as in ative. She remembered four of five words FERNANDO DE LA IGLESIA MARTINEZ Department ofInternal Medicine, another patient with HSV-1 myelitis whose after five minutes, and the mini mental state HospitalYuan Canalejo, necropsy examination showed patchy test was 32/35. She remained in a left tilted La Coruna, Spain demyelination of the spinal cord.7 posture, and showed severe impairment of EMMANUEL ELLIE postural reflexes, mild bradykinesia, cog- Correspondence to: Dr Rail de la Fuente Service de Neurologie, Fernandez, Servicio de Neurologia, Hospital Juan Hopital du Haut-Livique, wheel rigidity, and intermittent resting Canalejo, Xubias de Arriba, 84, 15006 La CHU Bordeaux, France tremor in the right extremities (mainly in Coruina, Spain. FLORE ROZENBERG the lower limb). Tendon reflexes were brisk Service de Bactiriologie-Virologie, 1 Lhermitte J. Syndrome de la calotte du pedon- H6pital Saint-Vincent de Paul, and increased on the right side, but there cule cerebral. Les troubles psycho-sensoriels Paris, France was no clonus and the plantar responses dans les lesions du mesocephale. Rev Neurol VINCENT DOUSSET were flexor. There were no other remark- (Paris) 1922;38: 1359-65. Service de Neuroradiologie, able findings on general or neurological 2 Geller TI, Bellur SN. Peduncular hallucinosis: CHU Bordeaux, France magnetic resonance imaging confirmation of MARIE BEYLOT-BARRY examination. Laboratory investigations, mesencephalic infarction during life. Ann Service de Dermatologie, including ESR, routine haematological, bio- Neurol 1987;21:602-4. CHU Bordeaux, France chemical, and immunological studies, thy- 3 McKee AC, Levine DN, Kowall NW, Richardson EP Jr. Peduncular hallucinosis roid function tests, serological tests for associated with isolated infarction of the Correspondence to: Dr Emmanuel Ellie, fluid INSERM U394, rue Camille Saint-Saens, 33077 syphilis, cerebrospinal examination, substantia nigra pars reticulata. Ann Neurol EEG, and cranial CT were normal or nega- 1990;27:500-4. Bordeaux Cedex, France. 4 Beckstead RM, Frankfurter A. The distribu- tive. An MRI showed an abnormal high tion and some morphological features of 1 Britton CB, Mesa-Tejada R, Fenoglio CM, intensity signal in the left cerebral peduncle substantia nigra that project to the thala- Hays AP, Garvey GJ, Miller JR. A new on T2-weighted images (figure). Multiple mus, superior colliculus and pedunculopon- complication of AIDS: thoracic myelitis foci of T2-weighted high signal intensity tine nucleus in the monkey. Neuroscience caused by herpes simplex virus. Neurology 1982;7:2377-88. 1985;35:1071-4. were also seen throughout the periventricu- 5 Jellinger K. The pathology of parkinsonism. 2 Tucker T, Dix RD, Katzen C, Davis RI, lar white matter. Such findings were consis- In: Marsden CD, Fahn S, eds. Movement Schmidley JW. Cytomegalovirus and herpes tent with ischaemic damage. Despite disorders. Vol 2. London: Butterworths, simplex virus ascending myelitis in a patient 1987:124-65. http://jnnp.bmj.com/ with acquired immune deficiency syndrome. treatment with haloperidol and phenytoin Ann Neurol 1985;18:74-9. the patient became more impaired within 3 Wiley CA, VanPatten PD, Carpenter PM, the next several weeks, showing continuous Powell HC, Thal U. Acute ascending visual hallucinations, with frequent episodes necrotizing myelopathy caused by herpes Intraneural ganglion of the sciatic simplex virus type 2. Neurolog 1987;37: of agitation and disorientation. 1791-4. Vascular lesion of the upper brainstem is nerve: detection by ultrasound 4 Iwamasa T, Yoshitake H, Sakuda H, et al. the most often cause of peduncular halluci- Acute ascending myelitis in Okinawa caused Intraneural ganglia are a rare cause of by herpes simplex virus type 2. Virchows nosis. A case of peduncular hallucinosis due bilateral infarction peripheral nerve lesions most often affecting Arch A Pathol Anat Histopathol 1991;418: to mesencephalic diag- on September 30, 2021 by guest. Protected copyright. 71-5. nosed by MRI has been recently reported.2 the peroneal nerve. Their origin is 5 Iwamasa T, Utsumi Y, Sakuda H, et al. Two The present one is the first report, however, unknown. Some 50 cases were reported up cases of necrotizing myelopathy associated to 1979.' Sciatic nerve ganglia are very rare. with malignancy caused by herpes simplex to our knowledge, of peduncular halluci- virus type 2. Acta Neuropathol (Bert) 1989; We report a sciatic nerve lesion caused by a 78:252-7. giant ganglion situated at the level of the 6 Ahmed I. Survival after herpes simplex type II distal thigh and damaging the tibial portion myelitis. Neurology 1988;38: 1500. 7 Klastersky J, Cappel R, Snoneck JM, Flament only. J, Thirty L. Ascending myelitis in associa- A 36 year old male right handed and tion with herpes simplex virus. N Engl J7 Med right footed state officer complained of pain 1972;287: 182-4. in his right calf for about six years especially when jogging and walking for more than 30 minutes. He was treated for a lumbar disc hernia, but a lumbar CT was unremarkable. Simultaneously he noted discomfort in his Peduncular hallucinosis and right hemi- right toes when wearing shoes; this was parkinsonism caused by left mesen- relieved by wearing orthopaedic sandals. cephalic infarction On first neurological examination he was found to have Lasegue's sign with his right Since the original description of Lhermittel leg at an angle of about 850. Reflexes and several causes of peduncular hallucinosis sensation in the lower extremities were nor- in mal. There was moderate paresis of the toe have been reported, but always relation Axial T2-weighted MR image obtained with a to a bilateral mesencephalic lesion. We 05-T unit shows an area of hyperintensity in flexors. Plain radiographs of the lower spine describe here a patient with prolonged vis- the left cerebral peduncle, consistent with showed six lumbar vertebrae and a fissured ual hallucinations and right hemiparkinson- infarction. vertebral arch of the first sacral segment.