OCULOFACIAL-SKELETAL MYORHYTHMIA IN CENTRAL-NERVOUS-SYSTEM WHIPPLES-DISEASE - ADDITIONAL CASE AND REVIEW OF THE LITERATURE

A case of oculofacial-skeletal myorhythmia associated with cerebral Wh ipple's disease is presented. This peculiar abnormal movement disorder consists of a convergent-divergent pendular nystagmus associated with a synchronous, rhythmic movement of the mouth, jaw, and extremities. The movements in previous cases have responded inconsistently to a var iety of broad-spectrum antibiotic drugs, and antiepileptic, muscle rel axant, antispastic, and psychotropic drugs. The current patient's ment al status improved after starting intravenous ceftriaxone but improvem ent of the abnormal movements occurred only after the addition of oral valproate. Current treatment consists of intravenous trimethoprim-sul famethoxazole for 2 weeks followed by oral trimethoprim-sulfamethoxazo le twice daily for 1 year. Presented here is an alternative treatment of intravenous ceftriazone followed by oral trimethoprimsulfamethoxazo le in combination with valproate for abnormal movements not responsive to antibiotic drugs alone.