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Topiramate Effect in Opsoclonus-Myoclonus-Ataxia Syndrome

Thiago Dias Fernandes, MD; Rodrigo Bazan, MD; Luiz Eduardo Betting, MD, PhD; Fernando Coronetti Gomes  da Rocha, MD, PhD
Arch Neurol. 2012;69(1):133. doi:10.1001/archneurol.2011.717.
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A 34-year-old woman with no comorbidities developed severe truncal ataxia and opsoclonus progressively over 4 days. The patient was admitted 20 days after onset of symptoms. During hospitalization, she was extensively investigated for the etiology of opsoclonus-myoclonus-ataxia syndrome (OMS). All ancillary tests performed gave normal results, including cerebrospinal fluid examination; serology for infectious diseases and collagenosis; brain and spine magnetic resonance imaging; thoracic, abdominal, and pelvic computerized tomography; and screening for gynecological neoplasias. Topiramate treatment was initiated at a starting dose of 50 mg/d and was titrated up to 150 mg/d over 10 days. During this period, the patient had a remarkable improvement of symptoms (video). After a few days, the topiramate dose was gradually decreased to 50 mg/d. However, after 1 week, ataxia and the opsoclonus clearly worsened. After reestablishing the medication dose to 150 mg/d, symptoms improved again.

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