Long-term follow-up study of vigabatrin in pretreated children with West syndrome

Seizure. 1998 Aug;7(4):293-7. doi: 10.1016/s1059-1311(98)80021-6.

Abstract

A multicentre, long-term, open-label, add-on study of vigabatrin was undertaken in 23 pretreated children with infantile spasms. After 3 months of vigabatrin therapy 11 of the 23 patients had become seizure-free. At this time two-thirds of these 11 children still received other antiepileptic drugs (AEDs) in addition to vigabatrin (mostly valproic acid and/or dexamethasone). After a mean follow-up time of 5 1/4 years (range: 4 1/4-6 1/2) 72% of 18 evaluable patients (two children died, three were lost to follow-up) revealed seizure freedom for at least 1 year. The mean duration of vigabatrin therapy had been 2 1/2 years (range: 2 weeks to 4 3/4 years). Two-thirds of the 18 children continued to take AEDs, three of them undergoing vigabatrin monotherapy. Relapses of infantile spasms had occurred in 14% of the children. The rate of vigabatrin side effects (10%) was low. At follow-up, the EEG of 13 and the 18 patients demonstrated focal or multifocal epileptic discharges. Fifty-five percent had developed another epilepsy (focal epilepsy, secondary generalized epilepsy or myoclonic-astatic epilepsy). With respect to mental functions, three children were normal or slightly retarded, four showed moderate retardation and 11 revealed severe or very severe retardation. This long-term result is comparable to that in ACTH studies with unselected patients. The conclusions are: (1) vigabatrin is an effective drug for the short-term and long-term treatment of refractory infantile spasms; (2) the relapse rate is low; (3) vigabatrin is well tolerated; (4) with respect to secondary epilepsies and mental functions the long-term outcome in these pretreated children is similar to that in earlier studies with ACTH or corticosteroids.

Publication types

  • Clinical Trial
  • Comparative Study
  • Multicenter Study

MeSH terms

  • Adrenocorticotropic Hormone / administration & dosage
  • Anti-Anxiety Agents / administration & dosage
  • Anticonvulsants / therapeutic use*
  • Benzodiazepines*
  • Child, Preschool
  • Clobazam
  • Dexamethasone / administration & dosage
  • Drug Therapy, Combination
  • Electroencephalography
  • Female
  • Glucocorticoids / administration & dosage
  • Humans
  • Infant
  • Male
  • Premedication
  • Prospective Studies
  • Pyridoxine / administration & dosage
  • Spasms, Infantile / drug therapy*
  • Treatment Outcome
  • Valproic Acid / administration & dosage
  • Vigabatrin
  • gamma-Aminobutyric Acid / analogs & derivatives*
  • gamma-Aminobutyric Acid / therapeutic use

Substances

  • Anti-Anxiety Agents
  • Anticonvulsants
  • Glucocorticoids
  • Benzodiazepines
  • Clobazam
  • gamma-Aminobutyric Acid
  • Valproic Acid
  • Dexamethasone
  • Adrenocorticotropic Hormone
  • Vigabatrin
  • Pyridoxine