title: Acute focal dystonia induced by a tricyclic antidepressant in a patient with Wilson disease: a case report.
abstract: The authors present the case of a 19-year-old patient with Wilson disease (WD) who developed symptoms of acute focal dystonia of the left hand (a 'starfish' hand presentation) shortly after treatment with the tricyclic antidepressant clomipramine. The diagnosis of WD was made 8 months earlier based on abnormal copper metabolism parameters and was confirmed by genetic testing. Initially, the patient presented with akathisia, sialorrhea, oromandibular dystonia (occasionally grimacing) and slight dysarthria. The patient's symptoms diminished after treatment with d-penicillamine was initiated. No further deterioration was observed after copper-chelating therapy was started. The authors diagnosed acute focal dystonia induced by clomipramine. Botulinum toxin and intensive rehabilitation was initiated; complete regression of hand dystonia was observed. Based on the case, the authors suggest that care should be exercised with regard to starting medications that could potentially impact the extrapyramidal system in WD patients.
authors.affiliation: Tomasz Litwin, Instytut Psychiatrii i Neurologii, II Klinika Neurologii, Sobieskiego 9, 02-957 Warszawa, phone: +48 22 458 25 68, fax: +48 22 842 40 23, e-mail: tomlit@medprakt.pl.
authors.forename: T
authors.initials: T
authors.lastname: Litwin
authors.forename: G
authors.initials: G
authors.lastname: Chabik
authors.forename: A
authors.initials: A
authors.lastname: Członkowska
mesh_terms: D000929:Antidepressive Agents, Tricyclic / Q000009:adverse effects*; D000931:Antidotes / Q000627:therapeutic use; D001905:Botulinum Toxins / Q000627:therapeutic use; D002614:Chelating Agents / Q000627:therapeutic use*; D002997:Clomipramine / Q000009:adverse effects*; D003300:Copper / Q000627:therapeutic use; D004421:Dystonia / Q000139:chemically induced*; D006225:Hand; D006527:Hepatolenticular Degeneration / Q000175:diagnosis* / Q000188:drug therapy*; D006801:Humans; D008297:Male; D009460:Neurologic Examination / Q000379:methods; D010396:Penicillamine / Q000627:therapeutic use*; D012074:Remission Induction; D016896:Treatment Outcome; D055815:Young Adult
chemical_list: D000929:Antidepressive Agents, Tricyclic; D000931:Antidotes; D002614:Chelating Agents; D003300:Copper; D001905:Botulinum Toxins; D010396:Penicillamine; D002997:Clomipramine