Efficacy of vigabatrin therapy for tuberous sclerosis with infantile spasms

Suzuki Muromoto Sato, Numata Uematsu Yurika, Hino Fukuyo Naomi, Mitsugu Ucmatsu, Hiroki Sato, Yuko Sato, Tojo Nakayama, Atsuo Kikuchi, Tomoko Kobayashi, Shigeo Kurc

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Abstract

Objective: To evaluate the effects and tolerability of vigabatrin (VGB) in children with tuberous sclerosis (TS) with infantile spasms or tonic seizures. Methods: We examined the impact of VGB on a series of 17 children with TS visiting Tohoku University Hospital in Japan during April 2010 and May 2015. To minimize potential adverse effects. VGB was given to the patients for limited 6 months with titration from 30 mg/kg/day as an initial dose. Results: Main seizure types were classified into spasms (n = 10) or tonic seizures (n = 7). Seizure reduction was positively associated with seizure type of infantile spasms, lower maximum dosage, younger age on VGB administration, and earlier VGB treatment after the diagnosis. Seizure type of infantile spasm was an independent favorable predictor and also associated with long-Term seizure reduction. Major adverse events included psychiatric symptoms (n = 7) and clectrorctinogram (ERG) abnormalities (n = 2). All symptoms were recovered by reducing the dosage of VGB. Conclusion: VGB is effective and well tolerated as first-line treatment for TS children with infantile spasms. Our "low dosage and limited period" protocol is efficient for improving seizure control as well as minimizing the potential risks of VGB.

Original languageEnglish
Pages (from-to)413-419
Number of pages7
JournalNo To Hattatsu
Volume48
Issue number6
Publication statusPublished - 2016

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Clinical Neurology

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    Sato, S. M., Yurika, N. U., Naomi, H. F., Ucmatsu, M., Sato, H., Sato, Y., Nakayama, T., Kikuchi, A., Kobayashi, T., & Kurc, S. (2016). Efficacy of vigabatrin therapy for tuberous sclerosis with infantile spasms. No To Hattatsu, 48(6), 413-419.