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

来源：

JOURNAL OF PEDIATRIC EPILEPSY | 2013年 / 2卷 / 04期

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

页码：255 / 258

页数：4

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