Novel LAMP-2 mutation in a family with Danon disease presenting with hypertrophic cardiomyopathy

Circ J. 2009 Feb;73(2):376-80. doi: 10.1253/circj.cj-08-0241. Epub 2008 Dec 5.

Abstract

Danon disease is an X-linked dominant multisystem disorder that includes hypertrophic cardiomyopathy with skeletal myopathy, and results from mutations in the gene encoding the lysosome-associated membrane protein-2 (LAMP-2). To date, over 20 different mutations in LAMP2 have been identified. Three members of a family, a male proband (18 years old) and 2 sisters (15 and 20 years old) were studied. Their mother had been diagnosed with dilated cardiomyopathy at the age of 39 years, and died from advanced heart failure at the age of 43 years. The proband developed marked concentric hypertrophy at the age of 5 years and DNA analyses revealed a novel hemizygous frameshift mutation (c.573delA) in exon 5. The 2 affected sisters were also heterozygous for the same mutation. Functional analyses of this novel LAMP2 mutation are mandatory.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adolescent
  • Cardiomyopathy, Hypertrophic / diagnosis
  • Cardiomyopathy, Hypertrophic / genetics*
  • Echocardiography
  • Electrocardiography
  • Frameshift Mutation / genetics*
  • Glycogen Storage Disease Type IIb / diagnosis
  • Glycogen Storage Disease Type IIb / genetics*
  • Humans
  • Hypertrophy, Left Ventricular / diagnostic imaging
  • Lysosomal Membrane Proteins / genetics*
  • Lysosomal-Associated Membrane Protein 2
  • Male
  • Pedigree

Substances

  • LAMP2 protein, human
  • Lysosomal-Associated Membrane Protein 2
  • Lysosomal Membrane Proteins