Abstract

Tardive dystonia, a persistent dystonia related to exposure to antipsychotic medication, is a relatively rare occurrence (Harenko 1967; Keegman and Rajput 1973; Burke et al. 1982). Burke et al. (1982) provide the most extensive description, discussing 42 cases and reviewing 15 previous reports. They noted the pharmacological management of the disorder to be difficult and rarely successful, with some improvement seen with dopamineblocking or -depleting agents (12 of 27 cases, 44%) or with anticholinergic agents (7 of 18 cases, 39%). We recently treated a patient with tardive dystonia who appeared to make a dramatic improvement with high-dose bromocriptine, a treatment used in other forms of dystonia (Stahl and Berger 1982). Because of the dearth of successful outcome in tardive dystonia, we thought it worthwhile to describe this case in detail. Mr. P is a 27-year-old single black man with a history of schizoaffective psychosis. During his first hospitalization at age 19, the patient was noted to manifest severe drooling and acute dystonia of the jaw musculature with either chlorpromazine, haloperidol, or fluphenazine. These symptoms were little improved by injections of diphenylhydramine 50 mg or oral benztropine mesylate up to 2 mg tid. Nevertheless, he continued to receive neuroleptics during his subsequent 14 hospitalizations. He was transferred to a chronic care facility in 1981, where he was treated with thiothixene 20 mg/day. In November 1982 the thiothixene dose was increased to 30 mg/day and a severe torticollis developed that did not respond to a 1-week withdrawal of antipsychotic medications along with oral or intramuscular benztropine 2 mg or diphenhydramine 50 mg. A neurological consultation led to the diagnosis of spasmodic torticollis and subsequent laboratory investigations including x-ray film of the spine, computed tomographic (CT) scan, electroencephalogram (EEG), and serum ceroloplasmin were all normal. Review of his history suggested no previous head trauma or loss of consciousness; however, there was a history of excessive alcohol intake during his early 20s. The patient was subsequently returned to antipsychotic medication, receiving in November 1982 thiothixene 40 mg/day along with amantadine 100 mg bid, in December 1982 chlorpromazine 800 mg/day, and in April 1983 thioridazine 400 mg/day along with diazepam 5 mg bid, and phenobarbital 30 mg tid. All these treatments were without significant effect on the patient's torticollis. In June 1983 the patient was diagnosed as having a tardive dystonia and all antipsychotic medications were discontinued. He was then treated with diazepam 15 mg tid alone and then in conjunction

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