| Literature DB >> 18555174 |
Prinyarat Burusnukul1, Emily C de Los Reyes, Jacqueline Yinger, Daniel R Boué.
Abstract
Danon disease is an X-linked cardioskeletal myopathy, originally reported as "lysosomal glycogen storage disease with normal acid maltase," resulting from a primary deficiency of lysosome-associated membrane protein-2 because of mutations in the lysosome-associated membrane protein-2 gene. Classic clinical features in males include cardiomyopathy (100%, eventually), myopathy (90%), and mental retardation (70%), but mostly of a mild degree. We report on an unusual presentation in a patient with autism, motor delay, and a normal cardiac evaluation. The presence of multiorgan involvement, including elevated liver enzymes, abnormal cranial magnetic resonance imaging, and diffuse hypotonia with swallowing difficulties, prompted a muscle biopsy. A quadriceps muscle biopsy was performed, and the findings were most suspicious for a glycogen storage-type disease. Subsequently, a pathogenic lysosome-associated membrane protein-2 mutation was found. To our knowledge, there are no previous clinical reports of autism in children with Danon disease.Entities:
Mesh:
Year: 2008 PMID: 18555174 DOI: 10.1016/j.pediatrneurol.2008.03.011
Source DB: PubMed Journal: Pediatr Neurol ISSN: 0887-8994 Impact factor: 3.372