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Vigabatrin Therapy for Infantile Spasms in a Case of Cardiofaciocutaneous Syndrome with Cardiac Hypertrophy Developing during Adrenocorticotropic Hormone Treatment
- 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.
- Subjects :
- Heart Defects, Congenital
0301 basic medicine
Pediatrics
medicine.medical_specialty
medicine.medical_treatment
Cardiomyopathy
Thyrotropin-releasing hormone
Cardiomegaly
Adrenocorticotropic hormone
Cardiofaciocutaneous syndrome
Vigabatrin
Electrocardiography
03 medical and health sciences
0302 clinical medicine
Adrenocorticotropic Hormone
Ectodermal Dysplasia
Humans
Medicine
business.industry
Infant, Newborn
Hypertrophic cardiomyopathy
Facies
Infant
West Syndrome
General Medicine
medicine.disease
Failure to Thrive
030104 developmental biology
Child, Preschool
Anesthesia
Female
business
Spasms, Infantile
030217 neurology & neurosurgery
Ketogenic diet
medicine.drug
Subjects
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