Oculofacial-skeletal myorhythmia in central nervous system Whipple's disease: Additional case and review of the literature

Authors

  • Dr. David A. Simpson,

    Corresponding author
    1. Michigan Institute for Neurological Disorders, Farmington Hills, Michigan, U.S.A.
    2. Garden City Osteopathic Hospital, Garden City, Michigan, U.S.A.
    • Michigan Institute for Neurological Disorders, 28595 Orchard Lake Road, Suite 200, Farmington Hills, MI 48334, U.S.A.
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  • Robin Wishnow,

    1. Michigan Institute for Neurological Disorders, Farmington Hills, Michigan, U.S.A.
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  • Richard B. Gargulinski,

    1. Michigan Institute for Neurological Disorders, Farmington Hills, Michigan, U.S.A.
    2. Oakland General Hospital, Madison Heights, Michigan, U.S.A.
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  • Anne M. Pawlak

    1. Michigan Institute for Neurological Disorders, Farmington Hills, Michigan, U.S.A.
    2. Garden City Osteopathic Hospital, Garden City, Michigan, U.S.A.
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Abstract

A case of oculofacial-skeletal myorhythmia associated with cerebral Whipple'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 variety of broad-spectrum antibiotic drugs, and antiepileptic, muscle relaxant, antispastic, and psychotropic drugs. The current patient's mental status improved after starting intravenous ceftriaxone but improvement of the abnormal movements occurred only after the addition of oral valproate. Current treatment consists of intravenous trimethoprim-sulfamethoxazole for 2 weeks followed by oral trimethoprim-sulfamethoxazole twice daily for 1 year. Presented here is an alternative treatment of intravenous ceftriazone followed by oral trimethoprim-sulfamethoxazole in combination with valproate for abnormal movements not responsive to antibiotic drugs alone.

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