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Phenytoin-induced dystonia and choreoathetosis in two retarded epileptic children

From the Departments of Pediatrics, Neurology, and Neurosurgery (Neurology), and the Division of Neurology, St. Louis Children's Hospital, St. Louis.

Similar movement disorders developed in two 8-year-old retarded children while they were receiving phenytoin. Seizures subsequent to a diphtheria-pertussis-tetanus immunization had developed in each child at 1 to 2 months of age. A static encephalopathy ensued, characterized by mental retardation, ataxia, spasticity, and a mixed seizure disorder. Intermittent dystonia and choreoathetosis developed insidiously while serum phenytoin concentrations were in the therapeutic range. Sustained dystonia and choreoathetosis developed 2 hours after an oral provocation with phenytoin. The baseline abnormalities on the electroencephalogram remained unchanged during the choreoathetosis. Recognizable metabolic abnormalities known to be associated with similar movement disorders were excluded. It was concluded from these studies that the movement disorder is secondary to phenytoin and can occur at therapeutic serum concentrations. Phenytoin is a central anticholinergic agent and a central stimulant of serotonin, and may induce movement disorders as a result of altering these neurotransmitters in the brain. The variable expression of these movement disorders may relate to the nature of the preexisting striatal insult.

This study was supported in part by USPHS grant No. NS-05633; the Clinical Research Center Grant MO-1 RR00036-15; and the Allen P. and Josephine B. Green Foundation, Mexico, Missouri.

Presented in part at the twenty-seventh annual meeting of the American Academy of Neurology, Bal Harbour, Florida, May 1975.

Received for publication August 4, 1975.

Reprint requests should be addressed to Dr. Chalhub at St. Louis Children's Hospital, 500 S. Kingshighway, St. Louis, MO 63110.

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