Replacement of antipsychotic and antiepileptic medication by L-alpha-methyldopa in a woman with velocardiofacial syndrome.
We report the case of a 23-year-old woman with velocardiofacial syndrome (VCFS) and a history of psychosis and seizures. She had been treated with conventional antipsychotic and antiepileptic drugs for 10 and 3 years, respectively. However, she continued to experience occasional hallucinations and paroxysmal jerking of the extremities. L-alpha-methyldopa 500 mg b.i.d. (later reduced to 250 mg t.i.d.) was added to her regimen. Hallucinations and seizures stopped shortly. Over the course of approximately 1 year, the previous medications were discontinued without recurrence of psychotic and epileptic symptoms. Eventually, improved mental functions and behaviour enabled her transition from living in a licensed residential facility to sharing a private residence with a partner. VCFS is associated with haploinsufficiency of catecholamine-methyltransferase, leading to excessive extraneuronal catecholamine concentrations. Alpha-Methyldopa inhibits catecholamine neurotransmission in a variety of ways. It is possible that the drug compensated for genetically disturbed catecholamine transmission thus achieving beneficial effects in this case.
O'Hanlon
James F
JF
Tri-Counties Regional Center, Santa Barbara, California 93103, USA. jameso@tri-counties.org
Ritchie
Rand C
RC
Smith
Edward A
EA
Patel
Rashiklal
R
D000015:Abnormalities, Multiple / Q000235:genetics*; D000316:Adrenergic alpha-Agonists / Q000627:therapeutic use*; D000328:Adult; D000927:Anticonvulsants / Q000627:therapeutic use*; D014150:Antipsychotic Agents / Q000627:therapeutic use*; D019465:Craniofacial Abnormalities / Q000235:genetics*; D005260:Female; D006801:Humans; D008750:Methyldopa / Q000627:therapeutic use*; D010641:Phenotype; D011618:Psychotic Disorders / Q000188:drug therapy* / Q000235:genetics; D012640:Seizures / Q000188:drug therapy / Q000235:genetics; D013577:Syndrome
D000316:Adrenergic alpha-Agonists; D000927:Anticonvulsants; D014150:Antipsychotic Agents; D008750:Methyldopa