Intraventricular baclofen infusion for dystonia. Report of two cases.
Two children with generalized dystonia were given continuous intraventricular baclofen (IVB) infusions. The first was a 12-year-old girl with mixed dystonia and spasticity caused by methylmalonic aciduria whose spinal anatomy precluded administration of intrathecal baclofen (ITB). The second was an 11-year-old boy whose dystonia was associated with cerebral palsy and had not improved significantly in response to conventional ITB treatment. The girl improved dramatically with IVB; the boy experienced no greater improvement from IVB than from ITB. No adverse effects were noted in either child during 2 to 6 months of infusion. These are the first reports of the use of IVB in this clinical setting. The results warrant additional investigation.
Albright
A Leland
AL
Department of Neurosurgery, Children's Hospital of Pittsburgh, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania 15213, USA. leland.albright@chp.edu
D001418:Baclofen / Q000008:administration & dosage*; D002648:Child; D004421:Dystonia / Q000188:drug therapy* / Q000209:etiology; D005260:Female; D006801:Humans; D015918:Infusion Pumps, Implantable; D007263:Infusions, Parenteral; D008297:Male; D009125:Muscle Relaxants, Central / Q000008:administration & dosage*; D020542:Third Ventricle; D016896:Treatment Outcome
D009125:Muscle Relaxants, Central; D001418:Baclofen