We report a man who developed oculogyric crisis one month after disulfiram intoxication. Brain MRI showed lesions involving bilateral globus pallidus and left substantia nigra. In our patient, neuronal discharges from pathologically reorganized basal ganglia circuit to the midbrain ocular motor center might lead to tonic deviation of the eyesope

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Journal of Movement Disorders

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     48 Copyright ⓒ 2009 The Korean Movement Disorder Society  Journal of Movement Disorders 2009;2:48-49 ISSN 2005-940X CASE REPORT Oculogyric Crisis Associated with Disulfiram-Induced Pallidonigral Lesion    파킨슨2(1) (파킨슨21-13) / 서하영 / 초교 /4.14전 화：717-5511, 전 송：717-5515 E-mail：ml@smileml.com 140-846 서울 용산구 원효로 1가 12-15(중앙 B/D)  We report a man who developed oculogyric crisis one month after disulfiram intoxication. Brain MRI showed lesions involving bilateral globus pallidus and left substantia nigra. In our patient, neuronal discharges from pathologically reorganized basal ganglia circuit to the mid-brain ocular motor center might lead to tonic deviation of the eyes.   Journal of Movement Disorders 2009;2:48-49 Key Words: Disulfiram, Oculogyric crisis.  Jae Hyeok Leea Chul Hyoung Lyoob Jin Goo Leec Myung Sik Leeb aDepartment of Neurology,  Pusan National University  Yangsan Hospital, Yangsan, Korea bDepartment of Neurology,  Gangnam Severance Hospital,  Yonsei University College of Medicine,  Seoul, Korea cDepartment of Neurology,  Incheon Choongang General Hospital,  Incheon, Korea     Received March 16, 2009 Revised April 3, 2009 Accepted March 25, 2009  Corresponding author Myung Sik Lee, MD, PhD Department of Neurology, Gangnam Severance Hospital, Yonsei University College of Medicine, 612 Eonju-ro, Gangnam-gu, Seoul 135-720, Korea Tel +82-2-2019-3322 Fax +82-2-3462-5904 E-mail mslee@yuhs.ac.   Oculogyric crisis (OGC) is characterized by a transient forceful upward conjugate devia-tion of the eyes. It can be seen in patients with postencephalitic parkinsonism,1 neuropetic induced acute or tardive dystonia,2 Wilson’s disease,3 and lesions involving uni-or bilateral lentiform nuclei.4,5  Disulfiram intoxication may produce a lesion at the globus pallidus and putamen, and causeparkinsonism and dystonia.6,7 We describe a male patient who attempted suicide with a highdose of disulfiram and developed oculogyric crisis, in addition to parkinsonism and dystoina. MRI brain scan studies showed lesions in the bilateral globus pallidus and left substantia nigra.  Case Report  A 15-year old boy attempted suicide by taking a high dose of disulfiram (20 mg). Upon ar-rival at the hospital, he was alert and vital signs were stable. He had not been treated neuro-leptics. One month after, he suddenly developed an acute insult consisted of anxiety attack and subsequent retrocollis and forceful upward deviation of the eyes. During the attack, he could not close his eyes and speak, making only grunting noises. He could not move his eyes voluntarily at all, but horizontal oculocephalic reflexes could be elicited. The attack was oc-casionally accompanied by dystonic posturing of all four limbs. The frequency, duration and intensity of the attacks increased gradually over a period of eight years, occurring more than 10 times a day and lasted up to 2 hours. Neurological examinations between the attacks re-vealed mild rigidity of the right arm and leg. There was a delay in initiation of saccadic eye movements. He had no motor weakness or sensory deficits, but he needed a help to walk due to foot dystonia. Deep tendon reflexes were normoactive and symmetric. Plantar reflexeswere flexor bilaterally. Routine laboratory tests were all normal. Ictal electroencephalography studies were unremarkable. T2-weighted brain MRI studies showed high signal intensity le-sions at the bilateral globus pallidus and left substantia nigra (Figure 1). Levodopa treatment was not effective, but the frequency of the attacks decreased markedly after trihexypheni-dyl hydrochloride (2.5 mg tid) treatment. Also, the foot dystonia improved partially and the patient could walk a short distance without assistance.  Discussion  Disulfiram is metabolized to cyanide disulfide (CS2) and produce lesions at the globus      Oculogyric Crisis after Disulfiram Intoxication █ Lee JH, et al. www.kmds.or.kr 49 pallidus and substantia nigra pars reticulata in monkeys.7 In huans, pallidal or lenticular lesions after disulfiram intoxication have been reported.6,7 However, to our knowledge, lesion at the substantia nigra after disulfiram intoxication is rarely docu-mented on brain MRI studies.  OGC occurs frequently in association with neuroleptic treat-ment, postencephalitic parkinsonism, and focal brain lesions at the putamen or globus pallidus.1,2,4,5 These findings suggest that OGC can be caused by basal ganglia dysfunction, partic-ularly of the dopaminergic system. In our patient, neuronal dis-charges from pathologically reorganized basal ganglia circuit to the midbrain ocular motor center might lead to tonic devia-tion of the eyes.  We reported a patient who developed disulfiram induced parkinsonism, dystonia, and OGC, responding markedly to the anticholinergics treatment.  REFERENCES 1. Onuaguluchi G. Crises in post-encephalitic parkinsonism. Brain 1961; 84:395-414. 2. Leigh RJ, Foley JM, Remler BF, Civil RH. Oculogyric crisis: a syn-drome of thought disorder and ocular deviation. Ann Neurol 1987;22: 13-17. 3. Lee MS, Kim YD, Lyoo CH. Oculogyric crisis as an initial manifesta-tion of Wilson’s disease. Neurology 1999;52:1714-1715. 4. Kim JS, Kim HK, Im JH, Lee MC. Oculogyric crisis and abnormal magnetic resonance imaging signals in bilateral lentiform nuclei. Mov Disord 1996:11:756-758.  5. Schoser BG, Groden C. Subacute onset of oculogyric crises and gen-eralized dystonia following intranasal administration of heroin. Addic-tion1999;94:431-414. 6. Krauss JK, Mohadjer M, Wakhloo AK, Mundinger F. Dystonia and akinesia due to pallidoputaminal lesions after disulfiram intoxication. Mov Disord 1991;6:166-170. 7. Laplane D, Attal N, Sauron B, de Billy A, Dubois B. Lesions of basal ganglia due to disulfiram neurotoxicity. J Neurol Neurosurg Psychi-atry 1992;55:925-929.   A B Figure. 1. T2-weighted brain MRI shows increased signal intensity inboth globus pallidus (A) and left midbrain adjacent substantia nigra(B). 

Oculogyric crisis associated with disulfiram-induced pallidonigral lesion

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Oculogyric crisis associated with disulfiram-induced pallidonigral lesion

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