Pathological evidence of vacuolar myelinopathy in a child following vigabatrin administration

Myles Horton1, Mubeen Rafay, Marc R Del Bigio

  • 1Section of Neurology, Department of Medicine, University of Manitoba, Winnipeg, Canada.

Journal of Child Neurology
|September 24, 2009
PubMed

Insights

Vigabatrin, an anti-seizure drug, caused white matter vacuolation and edema in a human infant, mirroring animal study findings. This case highlights potential risks in infants, especially those with prior myelin issues.

Area of Science:

  • Neuroscience
  • Pharmacology
  • Pediatric Neurology

Background:

  • Vigabatrin is a gamma-aminobutyric acid (GABA) aminotransferase inhibitor used to control seizures.
  • Animal studies linked vigabatrin to white matter vacuolation and intramyelinic edema.
  • These lesions had not been previously documented in human patients receiving vigabatrin.

Observation:

  • A child with cerebral palsy received vigabatrin for infantile spasms at nine months of age.
  • Neurologic function rapidly declined after vigabatrin initiation.
  • The child died three weeks after starting the medication.

Findings:

  • Neuropathological examination revealed white matter vacuolation and intramyelinic edema.
  • This is the first documented human case of vigabatrin-induced intramyelinic edema.
  • The findings align with observations from animal studies.

Implications:

  • This case validates concerns about vigabatrin's safety profile in infants.
  • It underscores potential risks for individuals with pre-existing myelin abnormalities.
  • Further caution is warranted when prescribing vigabatrin to vulnerable pediatric populations.

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