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Pearls & Oy-sters: Acute Obstructive Hydrocephalus Co-Occurrence With Vigabatrin-Associated MRI Changes in an Infant With LIS1-Related Classical Lissencephaly

Year: 2025

Type: article

Abstract: We present a case of a 4-month-old infant with classical type I lissencephaly treated with vigabatrin for hypsarrhythmia. The patient subsequently developed hypotonia and respiratory failure. Imaging revealed vigabatrin-associated brain abnormalities on MRI (VABAM), with acute obstructive hydrocephalus requiring emergent extraventricular drainage. VABAM involve a predictable pattern of restricted diffusion, are generally clinically silent, and are reversible on cessation of the medication. This case presents a rare, life-threatening complication in association with VABAM. In this report, we review the pathophysiology of this phenomenon and the potential influence of lissencephaly.

Source: Neurology

DOI: 10.1212/wnl.0000000000214181

Authors: Alexandra Gamber, Hugh Garton, Hemant Parmar, Eric Armour, Giulia M. Benedetti

Institution: University of Michigan

Language: English

Topic: Fetal and Pediatric Neurological Disorders

Subfield: Pediatrics, Perinatology and Child Health

Field: Medicine

Domain: Health Sciences

SDG: Good health and well-being

Open Access status: bronze

OpenAlex ID: W4414578992