Oculo-Facial-Skeletal Myorhythmia in Whipple Disease: Treatment with Ceftriaxone

Brief Reports15 March 1990Oculo-Facial-Skeletal Myorhythmia in Whipple Disease: Treatment with CeftriaxoneCharles H. Adler, MD, PhD, Steven L. Galetta, MDCharles H. Adler, MD, PhDSearch for more papers by this author, Steven L. Galetta, MDSearch for more papers by this authorAuthor, Article, and Disclosure Informationhttps://doi.org/10.7326/0003-4819-76-3-112-6-467 SectionsAboutPDF ToolsAdd to favoritesDownload CitationsTrack CitationsPermissions ShareFacebookTwitterLinkedInRedditEmail ExcerptWhipple disease is a chronic, systemic disorder resulting in steatorrhea, weight loss, arthralgias, fever, and neurologic symptoms (1). Central nervous system manifestations may occur without systemic involvement (2-4), and include headache, seizures, meningitis, myoclonus, dementia, and supranuclear ophthalmoplegia (1-4). A unique, possibly pathognomonic, movement disorder, involving a slow (1 Hz) convergent-divergent pendular nystagmus, associated with synchronous, rhythmic movements of the mouth and other body parts, has been called oculomasticatory or oculo-facial-skeletal myorhythmia. It has previously been reported in six patients with biopsy-proven Whipple disease (5-9). We report a patient with Whipple disease with amnesia, meningitis, supranuclear ophthalmoparesis, and oculo-facial-skeletal myorhythmia...References1. ComerBrandtAbissi GLC. Whipple's disease: a review. Am J Gastroenterol. 1983;78:107-14. MedlineGoogle Scholar2. PallisLewis CP. Neurology of gastrointestinal disease. In: Vinken PJ, Bruyn SW, eds. The Handbook of Neurology. Amsterdam: North Holland Publishing Company; 1980:39 (Part II): 449-68. Google Scholar3. PollockLewisKendall SPB. Whipple's disease confined to the nervous system. J Neurol Neurosurg Psychiatry. 1981;44:1104-9. CrossrefMedlineGoogle Scholar4. AdamsRhynerDayDeArmondSmuckler MPJSE. Whippie's disease confined to the central nervous system. Ann Neurol. 1987;21:104-8. CrossrefMedlineGoogle Scholar5. Van BogaertLafonPagesLabauge LRPR. Sur une encephalite subaigue non classable, principalement characterisee par des myorythmies oculo-facio-cervicales. Rev Neurol (Paris). 1963;109:443-53. Google Scholar6. KnoxBaylessPitman DTF. Neurologic disease in patients with treated Whipple's disease. Medicine (Baltimore). 1976;55:467-76. CrossrefMedlineGoogle Scholar7. SchwartzSelhorstOchs MJA. Oculomasticatory myorhythmia: a unique movement disorder occurring in Whipple's disease. Ann Neurol. 1986;20:677-83. CrossrefMedlineGoogle Scholar8. GrottaPettigrewSchmidtFriedmanSnatic JLWGS. Oculomasticatory myorhythmia [Letter]. Ann Neurol. 1987;22:395-6. CrossrefMedlineGoogle Scholar9. Hausser-HauwRoulletRobertMarteau CERR. Oculo-facioskeletal myorhythmia as a cerebral complication of systemic Whipple's disease. Mov Disord. 1988;3:179-84. CrossrefMedlineGoogle Scholar10. KeinathMerrellVlietstraDobbins RDRW. Antibiotic treatment and relapse in Whipple's disease: long-term follow-up of 88 patients. Gastroenterology. 1985;88:1867-73. CrossrefMedlineGoogle Scholar This content is PDF only. To continue reading please click on the PDF icon. Author, Article, and Disclosure InformationAffiliations: From the Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania. For current author addresses, see end of text. 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