Literature DB >> 8853067

Case report: lysosomal glycogen storage disease with normal acid maltase: an unusual form of hypertrophic cardiomyopathy with rapidly progressive heart failure.

H F Tse1, T W Shek, Y T Tai, Y K Lau, L Ma.   

Abstract

A 14-year-old boy with mild mental retardation, myopathy, and nonobstructive hypertrophic cardiomyopathy (HCM) with clinical and histopathologic features consistent with lysosomal glycogen storage disease with normal acid maltase is described. The case illustrates the aggressive nature of the cardiomyopathy of this syndrome. This condition is associated with malignant ventricular arrhythmias, relentlessly progressive ventricular dilatation, dysfunction, and sudden death. It is important to recognize this unusual and malignant form of HCM to precipitate low early diagnosis by muscle biopsy. Patients with this condition would be excellent candidates for life-saving heart transplant as the myopathy and mental retardation are mild and nonprogressive. The underlying biochemical defect and mode of inheritance of this syndrome are unclear. However, a significant proportion are genetically related and thus, relatives may benefit from family screening.

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Year:  1996        PMID: 8853067     DOI: 10.1097/00000441-199610000-00006

Source DB:  PubMed          Journal:  Am J Med Sci        ISSN: 0002-9629            Impact factor:   2.378


  1 in total

1.  Familial X-linked cardiomyopathy (Danon disease): diagnostic confirmation by mutation analysis of the LAMP2gene.

Authors:  Christian Balmer; Diana Ballhausen; Nils U Bosshard; Beat Steinmann; Eugen Boltshauser; Urs Bauersfeld; Andrea Superti-Furga
Journal:  Eur J Pediatr       Date:  2005-05-12       Impact factor: 3.183

  1 in total

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