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Turkiye Klinikleri J Ophthalmol 2018;27(1):72-7

OLGU SUNUMU DOI: 10.5336/ophthal.2016-51274

Isolated Unilateral Oculomotor Palsy Due to Hematoma in Mesencephalone

Servet ÇETİNKAYA, a ABS TRACT palsy causes reduced adduction, supraduction, infraduction, and Emine MESTAN b ptosis with or without . We report a case of isolated unilateral due to hematoma in mesencephalone. A 30-years- old male patient presented with sudden onset of left ptosis, vertical diplopia, and difficulty in reading with his left that had persisted for one week. aClinic of Ophthalmology, Direct and indirect light reflexes were negative on his left eye. There was incomplete ptosis on his Turkish Red Crescent Hospital, Konya left eyelid. In primary position, the left eye was deviated outward and downward. Supraduction of bDepartment of Neurology, the left eye was absent, infraduction and adduction were reduced. MRI demonstrated hematoma in Dumlupınar University Faculty of Medicine, mesencephalone. Three months later, patient’s symptoms decreased, MRI demonstrated resolution Kütahya of the hematoma and cavernoma in pons and gamma knife therapy was recommended. Isolated unilateral oculomotor nerve palsy due to hematoma related to cavernoma in mesencephalone is Re ce i ved: 09.03.2016 seen rarely and complete or incomplete recovery may be observed. Received in revised form: 16.06.2016 Ac cep ted: 17.06.2016 Keywords: Hematoma; mesencephalon; oculomotor nerve diseases Available online : 22.02.2018

Cor res pon den ce: Servet ÇETİNKAYA ÖZET Okülomotor sinir felci azalmış addüksiyon, supradüksiyon, infradüksiyon ve kapakta dü - Turkish Red Crescent Hospital, şüklüğe neden olur, midriasis de olaya eşlik edebilir. Biz, mezensefalondaki hematomdan kaynak - Clinic of Ophthalmology, Konya, lanan izole tek taraflı okülomotor sinir felci olan bir vakayı takdim ediyoruz. Bir hafta devam eden, TÜRKİYE/TURKEY ani başlangıçlı sol gözde kapakta düşüklük, dikey çift görme ve sol gözle yakını görememe şikaye - [email protected] tiyle polikliniğimize başvuran 30 yaşındaki erkek hastada, yapılan muayenede sol gözde direkt ve indirekt ışık reflekslerinin negatif olduğu gözlendi. Sol göz üst kapakta kısmi düşüklük mevcuttu. Primer pozisyonda sol göz dışa ve aşağı doğru kaymış durumdaydı. Sol gözün supradüksiyonu yoktu, addüksiyon ve infradüksiyonu ise azalmıştı. MR görüntülerinde mezensefalonda hematom izlen - mekteydi. Üç ay sonra hastanın belirti ve bulguları azaldı, MR görüntülerinde hematomda çözülme ve ponsta kavernom izlendi ve hastaya gamma knife terapisi önerildi. Mezensefalondaki hema - tomdan kaynaklanan kavernom ile ilişkili izole tek taraflı okülomotor sinir felci nadiren görülür ve tam veya kısmi iyileşme gerçekleşir.

Anah tar Ke li me ler: Hematom; mezensefalon; okülomotor sinir hastalıkları

he oculomotor nerve (the third cranial nerve) innervates the medial, inferior, superior recti muscles, the inferior oblique muscle, the lev - ator palpebrae superioris muscle, the muscle and the .1 The third nerve nucleus is located in the midbrain near the cerebral Cop yright © 2018 by Tür ki ye Kli nik le ri aqueduct at the level of the superior colliculus. Each extraocular muscle

72 Servet ÇETİNKAYA et al. Turkiye Klinikleri J Ophthalmol 2018;27(1):72-7 that receives innervation from the third nerve has rect and indirect light reflexes were positive on his a corresponding subnucleus. A single central nu - right eye, and negative on his left eye. Left cleus (central caudal nucleus) innervates both lev - was mydriatic and unresponsive to light. There was ator palpebrae muscles. Distinct, bilateral subnuclei incomplete ptosis on his left eyelid. In primary po - exist for the extraocular muscles. An additional bi - sition, the left eye was deviated outward and lateral subnucleus, the Edinger-Westphal nucleus, downward. Supraduction of the left eye was absent, provides parasympathetic input to the ciliary body infraduction and adduction were reduced. All and pupillary sphincter. Each subnucleus inner - movements of the right eye were normal, as shown vates the ipsilateral corresponding extraocular in Figure 1. Forced duction test was negative. Pa - muscle, with two exceptions, the single central tient’s diplopia complaint was getting worse, when caudal nucleus sends projections to both levator he was forced to look upwards. His visual field muscles and the superior rectus subnucleus inner - analysis revealed no abnormalities. He had no 2 vates contralateral superior rectus muscle . other neurological signs or symptoms. Oculomotor fascicles travel ventrally through Complete blood count and biochemical analy - the midbrain, each passing through the red nu - sis of the blood were normal. MRI demonstrated cleus, substantia nigra and crus cerebri before they hematoma in mesencephalone on T1 and T2 exit into subarachnoid space of the interpeduncu - weighted images, as shown in Figure 2. Patient also lar fossa. Then the third nerve enters the cavernous has arachnoid cyst seen in anterior part of the left sinus, wherein it is separated into superior and in - temporal lobe on the MRI images, but that was not ferior divisions. The superior division innervates related to the patient’s clinical findings. Patient was the superior rectus and levator muscles, while the consulted to Neurology and Neurosurgery Clinics inferior division supplies the inferior rectus, infe - and he was followed-up by neurologists and neu - rior oblique, medial rectus, ciliary body and pupil - rosurgeons, too. Since central caudal nucleus (the lary sphincter muscles. After entering the superior levator palpebralis nucleus) projects bilaterally and orbital fissure, the oculomotor nerve lies within the the superior rectus subnucleus projects contralat - and muscle cone and reaches the erally, in our patient the pupillary involvement, respective muscles .3 unilateral ptosis and unilateral reduction in the Damage to the third cranial nerve causes re - movements of inferior, superior,medial recti and duced adduction, supraduction, infraduction, and inferior oblique muscles, indicate that the fascicles 4 ptosis with or without mydriasis . The aim of this within the midbrain were affected. case report is to represent the course of this rare case. A gradual improvement of oculomotor nerve functions was observed during the subsequent fol - CASE REPORT low-ups. Meanwhile he did not take any treat - ments. Three months later, patient’s symptoms A 30-years-old male patient presented with sudden decreased but didn’t resolve completely. Left pupil onset of left ptosis, vertical diplopia and difficulty was responsive to light but lazy, ptosis was recov - in reading with his left eye that had persisted for ered, eye movements were better, but still they one week. He had no systemic diseases. He never were reduced, as shown in Figure 3. MRI at that used tobacco or alcohol. On examination, he was time demonstrated resolution of the hematoma conscious and well oriented, his vital signs were and cavernoma or cavernous angioma (arterio -ve - normal. nous malformation) in pons on T1 and T2 His uncorrected visual acuities were 20/20 on weighted images, shown in Figure 4. Neurosur - both , his intraocular pressures were 16 mmHg geons recommended gamma knife therapy (radia - in right eye and 15 mmHg in left eye respectively, tion therapy) for the patient to eradicate or and examination was normal bilaterally. Di - minimize the lesion.

73 Servet ÇETİNKAYA et al. Turkiye Klinikleri J Ophthalmol 2018;27(1):72-7

FIGURE 1: The primary position and the movements of right and left eyes of the patient.

FIGURE 2: T1 and T2 weighted MRI images.

74 Servet ÇETİNKAYA et al. Turkiye Klinikleri J Ophthalmol 2018;27(1):72-7

FIGURE 3: The primary position and the movements of right and left eyes of the patient after three months.

FIGURE 4: T1 and T2 weighted MRI images after three months.

betes, hypertension and atherosclerosis, and hemor - DISCUSSION rhage from arterio -venous malformation and hy - Oculomotor nerve palsy may be congenital or ac - pertension, demyelinization and compression by quired. Most acquired causes in adults are vascular intracranial aneurysms or other mass lesions like pri - diseases such as ischemia or infarction due to dia - mary or metastatic tumors. Infectious, inflammatory

75 Servet ÇETİNKAYA et al. Turkiye Klinikleri J Ophthalmol 2018;27(1):72-7 and traumatic causes are less likely .5-14 In our patient rological signs such as pyramidal tract dysfunction, the cause was hematoma due to hemorrhage from ab normalities of coordination and impaired con - cavernoma (arterio -venous malformation ). sciousness, because of the large number of struc - Identification of a total third nerve palsy is tures located nearby, isolated palsies are very 2,15 typically straightforward on examination, when rare . Due to unilateral ptosis and unilateral ex - there is ptosis, an eye that is deviated ‘down and traocular muscular involvement in our patient, out’ and a dilated pupil .[1,5] Our patient had total the lesion was thought to be fascicular in origin. third nerve palsy, since he had ptosis, ‘down and And other cranial , motor, sensory, coor - out’ deviation and a dilated unreactive pupil. But dination and reflex systems were spared in our it was incomplete third nerve palsy, because in - patient. fraduction and adduction of the left eye were not Edinger-Westphal nucleus provides para- absent but reduced and the ptosis was not com - sympathetic input to the pupillary sphincter and plete. ciliary body. This nucleus and its fascicles are lo - Systemic hypertension and arterio -venous cated at the top of the nuclear complex. Bilateral malformations represent the most common etiolo - pupillary involvement with nuclear oculomotor gies of primary hematomas of the mesen - palsy, therefore, is only expected in lesions with 3,15 cephalone .15 De Mendonca et al. 16 reported a extension to the rostral midbrain . Pupil-spar - 70-years-old hypertensive woman suffering from ing oculomotor palsy is most likely caused by mi - sudden onset of bilateral blepharoptosis, within crovascular ischemia resulting from diabetes, few hours she developed focal signs attributable to hypertension and atherosclerosis .5 Unilateral dif - a lesion of the mesencephalone and a stuporous ficulty in reading and unreactive pupil in our pa - state, from which she did not recover, neu - tient were due to the destruction of the ropathological examination showed a hematoma in pupillomotor fibers located medially among the mesencephalone . Ramirez-Moreno et al. presented oculomotor fascicles. a case of hypertensive woman with an oculomotor Neurosurgical intervention (surgical evacua - nerve syndrome associated with limitation of tion) may be required in severe cases of mesen - horizantal gaze of the contralateral eye and reac - cephalic hematoma .18 Careful observation for at tive head inclination due to mesencephalic least 6 months is essential before planning a stra - 17 hematoma. Durward et al. reported two patients bismus and/or ptosis surgery, as some patients will presenting with a mesencephalic hematoma, the show a partial or full spontaneous recovery. Most etiology was a proven arteriovenous malformation oculomotor nerve palsy due to microangiopathy re - in one case and the other one was suspected to be 18 solves completely in the first few months. When due to hypertensive arteriolar rupture. In our pa - strabismus surgery is needed, the extent and sever - tient, the etiology was also arteriovenous malfor - ity of extraocular muscle involvement and the mation (cavernoma in pons) . presence of aberrant regeneration play a crucial The crossed projection of the superior rectus role in surgical planning .5 For our patient, neuro - subnucleus indicates that unilateral third nerve le - surgeons recommended gamma knife therapy, sions with contralateral superior rectus involve - which is a radiation therapy. ment obligate a nuclear lesion, whereas a third nerve palsy with no contralateral superior rectus abnormality can not be caused by a nuclear lesion, CONCLUSION furthermore, unilateral ptosis also shows that the In conclusion, isolated unilateral oculomotor nerve lesion is not nuclear, because a single central nu - palsy due to hematoma related to cavernoma in cleus (central caudal nucleus), innervates both le - mesencephalone is seen very rarely and complete vator palpebrae muscles. Nuclear and fascicular or incomplete recovery may be observed in such nerve palsies often are associated with other neu - cases.

76 Servet ÇETİNKAYA et al. Turkiye Klinikleri J Ophthalmol 2018;27(1):72-7

Source of Finance members of the potential conflicts of interest, counseling, ex - During this study, no financial or spiritual support was re - pertise, working conditions, share holding and similar situa - ceived neither from any pharmaceutical company that has a tions in any firm. direct connection with the research subject, nor from a com - Authorship Contributions pany that provides or produces medical instruments and ma - terials which may negatively affect the evaluation process of Idea/Concept: Servet Çetinkaya; Design: Servet Çetinkaya; this study. Control/Supervision: Emine Mestan; Data Collection and/or Processing: Servet Çetinkaya; Analysis and/or Interpretation: Conflict of Interest Servet Çetinkaya; Literature Review: Servet Çetinkaya; Writ- No conflicts of interest between the authors and / or family ing the Article: Servet Çetinkaya; Critical Review: Emine Mes - members of the scientific and medical committee members or tan.

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