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Vigabatrin-associated brain abnormalities on MRI in tuberous sclerosis complex patients with infantile spasms: are

Lin Wan1,2,3, Wen He1,2, Yang-Yang Wang1,2

  • 1Department of Pediatrics, PLA General Hospital, Beijing, China.

Therapeutic Advances in Neurological Disorders
|January 5, 2023
PubMed

Insights

Vigabatrin-associated brain abnormalities (VABAM) occurred less frequently in infantile spasms patients with tuberous sclerosis complex (TSC). Concomitant rapamycin treatment may reduce the risk of VABAM in these patients.

Area of Science:

  • Neurology
  • Pediatrics
  • Radiology

Background:

  • Vigabatrin (VGB) is a primary treatment for infantile spasms (IS), particularly in tuberous sclerosis complex (TSC).
  • Vigabatrin-associated brain abnormalities on MRI (VABAM) are known adverse events.
  • Understanding VABAM occurrence and risk factors is crucial for patient safety.

Purpose of the Study:

  • To determine the incidence of VABAM in children with IS caused by TSC (IST).
  • To identify risk factors associated with VABAM in IST patients.
  • To explore the potential protective effect of rapamycin therapy.

Main Methods:

  • Retrospective cohort study of 172 children with IS treated with VGB.
  • Cerebral MRI review to identify VABAM.
  • Comparison between IST patients and those with IS from other etiologies.
  • Logistic regression and survival analysis to identify risk/protective factors.

Main Results:

  • VABAM was observed in 22.1% of all IS patients receiving VGB.
  • The incidence of VABAM was significantly lower in IST patients (10%) compared to other IS etiologies (32.6%).
  • Concomitant rapamycin therapy was associated with a significantly lower risk of VABAM in IST patients.

Conclusions:

  • Infantile spasms patients with TSC exhibit a lower incidence of VABAM.
  • Rapamycin may be a protective factor against VABAM development.
  • Further research is warranted to elucidate the mechanisms and confirm rapamycin's protective role.
Abstract

Keywords:
VABAMincidenceinfantile spasmsrapamycintuberous sclerosis complex

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