Effectiveness of vigabatrin in West syndrome associated with tuberous sclerosis

Naoki Ando, Shinji Fujimoto, Tatsuya Ishikawa, Satoru Kobayashi, Ayako Hattori, Tetsuya Ito, Hajime Togari

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Abstract

Vigabatrin (VGB) is one of the most effective anti-epileptic drugs for tonic spasms, those accompanied with tuberous sclerosis complex (TSC), but is not available in Japan. We treated 7 patients with West syndrome (WS) and TSC with VGB. In these patients, VGB treatment was started at 5-65 months of age. Six patients (86%) had complete cessation of tonic spasms. Of these, 3 patients had complete cessation within 24 hours after VGB treatment. The mean initial dosage of VGB was 36.2 mg·kg-1·day-1, and the mean maintenance dosage was 38.4 mg·kg-1·day -1. At the beginning of VGB treatment, 3 patients had hypsarrhythmia, 2 had focal discharge with generahzation, and 2 had only focal discharge on electroencephalography. Hypsarrhythmia disappeared within 4-8 weeks after VGB treatment. Behavioral problems and sleep difficulty were observed in 6 patients. Visual field examination revealed no abnormalities in 3 patients. We hope that patients with WS and TSC can be treated with VGB as soon as possible in Japan.

Original languageEnglish
Pages (from-to)444-448
Number of pages5
JournalNo To Hattatsu
Volume42
Issue number6
Publication statusPublished - 01-12-2010
Externally publishedYes

All Science Journal Classification (ASJC) codes

  • Pediatrics, Perinatology, and Child Health
  • Clinical Neurology

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    Ando, N., Fujimoto, S., Ishikawa, T., Kobayashi, S., Hattori, A., Ito, T., & Togari, H. (2010). Effectiveness of vigabatrin in West syndrome associated with tuberous sclerosis. No To Hattatsu, 42(6), 444-448.