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

Authors :
Shinichiro Yamashita
Takayuki Hatori
Ko Ichihashi
Yuka Hirakubo
Kazuhito Nonaka
Yohei Sugiyama
Source :
Journal of Nippon Medical School. 83:167-171
Publication Year :
2016
Publisher :
Medical Association of Nippon Medical School, 2016.

Abstract

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.

Details

ISSN :
13473409 and 13454676
Volume :
83
Database :
OpenAIRE
Journal :
Journal of Nippon Medical School
Accession number :
edsair.doi.dedup.....c388e6184643586784fd6c824672ba65
Full Text :
https://doi.org/10.1272/jnms.83.167