Vigabatrin Therapy for Infantile Spasms in a Case of Cardiofaciocutaneous Syndrome with Cardiac Hypertrophy Developing during Adrenocorticotropic Hormone Treatment

被引:2
|
作者
Hatori, Takayuki [1 ]
Sugiyama, Yohei [1 ]
Yamashita, Shinichiro [1 ]
Hirakubo, Yuka [1 ]
Nonaka, Kazuhito [1 ]
Ichihashi, Ko [1 ]
机构
[1] Jichi Med Univ, Saitama Med Ctr, Dept Pediat, 1-847 Amanuma Cho, Saitama 3308503, Japan
关键词
cardiofaciocutaneous syndrome; infantile spasms; hypertrophic cardiomyopathy; vigabatrin; FACIO-CUTANEOUS SYNDROME; VISUAL-FIELD LOSS; CLINICAL-FEATURES; MUTATIONS; PREVALENCE; EPILEPSY;
D O I
10.1272/jnms.83.167
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
In a patient with cardiofaciocutaneous syndrome complicated by intractable infantile spasms (West syndrome), cardiac hypertrophy developed during adrenocorticotropic hormone treatment. Various types of antiepileptic drugs, intravenous immunoglobulin, thyrotropin releasing hormone, and a ketogenic diet were ineffective in this case. However, vigabatrin both decreased clinical seizures and improved electroencephalogram findings. Although vigabatrin has not been approved for use in Japan, the results in the present case suggest that this drug should be considered as an alternative therapy for cases of infantile spasms associated with syndromes involving cardiomyopathy or its potential risk factors, such as cardiofaciocutaneous syndrome.
引用
收藏
页码:167 / 171
页数:5
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