Extremely low-dose vigabatrin for West syndrome with tuberous sclerosis

被引:0
|
作者
Hino-Fukuyo, Naomi [1 ]
Sato, Yuko [1 ]
Kakisaka, Yosuke [1 ]
Endo, Wakaba [1 ]
Kubota, Yuki [1 ]
Kikuchi, Atsuo [1 ]
Kobayashi, Tomoko [1 ]
Haginoya, Kazuhiro [1 ,2 ]
Uematsu, Mitsugu [1 ]
Numata, Yurika [1 ]
Doi, Hiroshi [3 ]
Mori, Masato [4 ]
Osaka, Hitoshi [3 ,4 ]
Kure, Shigeo [1 ]
机构
[1] Tohoku Univ, Sch Med, Dept Pediat, Sendai, Miyagi, Japan
[2] Takuto Rehabil Ctr Children, Dept Pediat Neurol, Sendai, Miyagi, Japan
[3] Tohoku Univ, Sch Med, Dept Ophthalmol, Sendai, Miyagi, Japan
[4] Jichi Med Univ, Sch Med, Dept Pediat, Yamanashi, Japan
关键词
West syndrome; infantile spasms; tuberous screlosis complex; vigabatrin;
D O I
10.3233/PEP-14064
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Treatment of west syndrome in patients with tuberous sclerosis, the relevant effective period and doses of vigabatrin (VGB) to avoid serious side effects still needs further investigation. We report on a Japanese girl who showed good results with a very low dose of VGB. Tonic spasms appeared at 5 mo of age. Adrenocorticotropic hormone therapy resulted in incomplete seizure control. VGB at the lowest practical dose (30 mg/kg/d) showed complete control after 3 d. With reduction of the dose to 10 mg/kg/d, side effects such as hyperactivity, irritability, and sleep disturbances improved. She was seizure- free for the next 6 mo with an improved developmental quotient. Ophthalmological evaluation revealed no abnormality. The present case illustrates that low- dose VGB therapy (10 mg/kg/d) has fewer side effects and may bring prompt seizure control in west syndrome with tuberous sclerosis.
引用
收藏
页码:255 / 258
页数:4
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