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Efficacy of vigabatrin therapy for tuberous sclerosis with infantile spasms

Sato Suzuki-Muromoto,, Mitsugu Uematsu, Hiroki Sato

    No to Hattatsu = Brain and Development
    |July 17, 2018
    PubMed

    Insights

    Vigabatrin (VGB) effectively treats infantile spasms in children with tuberous sclerosis (TS). A low-dose, short-term VGB protocol shows good tolerability and seizure control, minimizing risks.

    Area of Science:

    • Pediatric Neurology
    • Epileptology
    • Genetics

    Background:

    • Tuberous sclerosis (TS) is a genetic disorder often associated with epilepsy, particularly infantile spasms.
    • Infantile spasms in TS patients can be challenging to treat and may lead to developmental delays.
    • Vigabatrin (VGB) is an established anti-epileptic drug, but its use requires careful consideration of side effects.

    Purpose of the Study:

    • To assess the efficacy and tolerability of vigabatrin (VGB) in pediatric patients diagnosed with tuberous sclerosis (TS).
    • To specifically evaluate VGB's impact on infantile spasms and tonic seizures in children with TS.
    • To determine the safety profile of a modified VGB treatment protocol in this population.

    Main Methods:

    • A retrospective study of 17 children with TS treated at Tohoku University Hospital between April 2010 and May 2015.
    • Vigabatrin (VGB) was administered at a starting dose of 30 mg/kg/day, with a maximum treatment duration of 6 months.
    • Seizure types, reduction in seizure frequency, and adverse events were systematically recorded and analyzed.

    Main Results:

    • Vigabatrin (VGB) demonstrated positive seizure reduction, particularly in patients with infantile spasms.
    • Favorable predictors for seizure reduction included infantile spasms as the seizure type, lower maximum VGB dosage, younger age at treatment initiation, and earlier treatment post-diagnosis.
    • The most common adverse events were psychiatric symptoms (n=7) and electroretinogram (ERG) abnormalities (n=2), both reversible upon dose reduction.

    Conclusions:

    • Vigabatrin (VGB) is an effective and well-tolerated first-line treatment for infantile spasms in children with tuberous sclerosis (TS).
    • The implemented "low dosage and limited period" protocol successfully improved seizure control while mitigating potential VGB-related risks.
    • This approach offers a promising strategy for managing epilepsy in pediatric TS patients, balancing efficacy with safety.

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