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Pearls & Oy-sters: Acute Obstructive Hydrocephalus Co-Occurrence With Vigabatrin-Associated MRI Changes in an Infant

Alexandra Gamber1, Hugh Garton2, Hemant Parmar3

  • 1Division of Neurology, Department of Pediatrics, University of Michigan, Ann Arbor.

Neurology
|September 26, 2025
PubMed

Insights

Vigabatrin treatment for infantile spasms can cause brain abnormalities (VABAM). In a case of lissencephaly, these abnormalities led to severe complications, including hydrocephalus, highlighting a rare but serious risk.

Area of Science:

  • Neurology
  • Pediatric Neurology
  • Neuroradiology

Background:

  • Lissencephaly is a severe brain malformation.
  • Hypsarrhythmia is a type of epilepsy often treated with vigabatrin.
  • Vigabatrin-associated brain abnormalities on MRI (VABAM) are known but typically reversible.

Purpose of the Study:

  • To report a rare, life-threatening complication of VABAM.
  • To discuss the potential influence of lissencephaly on VABAM.
  • To review the pathophysiology of VABAM.

Main Methods:

  • Case report of a 4-month-old infant with lissencephaly.
  • Treatment with vigabatrin for hypsarrhythmia.
  • Clinical and imaging follow-up including MRI.

Main Results:

  • The infant developed hypotonia and respiratory failure.
  • MRI revealed VABAM with acute obstructive hydrocephalus.
  • Hydrocephalus required emergent extraventricular drainage.

Conclusions:

  • VABAM can present with severe, life-threatening complications beyond typical reversible changes.
  • Lissencephaly may influence the presentation or severity of VABAM.
  • Careful monitoring is crucial in infants with lissencephaly treated with vigabatrin.

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