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Clinical Specialist, Department of Neuroscience, Neurology Division, Annunziata Hospital Cosenza, Italy
Researcher, Chair of Pharmacology, Department of Experimental and Clinical Medicine, Faculty of Medicine and Surgery, University Magna Graecia of Catanzaro, Clinical Pharmacology and Pharmacovigilance Unit, Mater Domini University Hospital, Via T. Campanella 115, 88100 Catanzaro, Italy, fax 39-0961-774424, luca_gallelli{at}hotmail.com
Chair of Pharmacology, Department of Experimental and Clinical Medicine, Faculty of Medicine and Surgery, University Magna Graecia of Catanzaro, Clinical Pharmacology and Pharmacovigilance Unit, Mater Domini University Hospital
Full Professor, Chair of Pharmacology, Department of Experimental and Clinical Medicine, Faculty of Medicine and Surgery, University Magna Graecia of Catanzaro, Clinical Pharmacology and Pharmacovigilance Unit, Mater Domini University Hospital
Published Online, October 2, 2007. www.theannals.com, DOI 10.1345/aph.1K233
Case Report. A 76-year-old woman with a 4 month history of movement disorders presented on April 23, 2006, to the neurological division of Cosenza Hospital for a consultation. She had a history of moderate hypertension and carotid atherosclerosis and was being treated with enalapril 20 mg/day and aspirin 100 mg/day. There was no history of alcohol or drug abuse. She was oriented and presented bilateral choreiform movements of the upper limbs and neck. The remainder of the physical examination was unremarkable. Routine biochemical and hematologic tests were normal, and no auto-antibodies (antiphospholipid, antinucleus, antiDNA) were documented.
A magnetic resonance imaging scan of the brain disclosed bilateral ischemic lesions in basal ganglia. Psychiatric evaluation and Mini-Mental State Examination did not reveal cognitive impairment (score 27/30) or other psychiatric manifestations. Testing protocols for Huntington's disease did not detect CAG-repeat expansion in the huntingtin (IT15) gene.
A diagnosis of vascular generalized chorea was established, and on April 27, haloperidol (1 mg every 8 h, up to 5 mg every 12 h) was started. However, due to the persistence of involuntary movements, haloperidol was discontinued and clonazepam 0.5 mg every 8 hours was begun. The appearance of somnolence one week later prompted discontinuation of clonazepam and initiation of topiramate (25 mg every 12 h, up to 50 mg every 12 h after 2 wk [June 4]), with an improvement in choreiform movements in about one week. The patient remained in good control of involuntary movements and sporadic presentations for 3 months. To evaluate whether improvement was related to pharmacologic treatment, topiramate was gradually withdrawn beginning on September 7 over a period of 7 days; at that time, there was a significant worsening of choreiform movements. Therefore, on October 4, topiramate 50 mg every 12 hours was restarted, with an improvement in involuntary movements.
As of April 2007, 6 months after the restart of topiramate treatment, no significant change in the frequency of choreiform movements had been observed. Moreover, neither adverse events nor topiramate dose adjustments had been recorded.
Discussion. It has been postulated that hyperkinetic movement
disorder is caused by decreased transmission of 
-aminobutyric acid
(GABA) in the indirect basal ganglia
pathways.4 In
our patient, haloperidol treatment did not show clinical efficacy and
clonazepam treatment induced the development of somnolence. On the other hand,
topiramate, which enhanced GABA
activity,5
induced an improvement in symptoms.
We suggest that topiramate could represent a useful therapeutic option in the treatment of symptomatic generalized chorea. However, prospective clinical trials should be undertaken to confirm these results.
References
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