Malignant cardiac phenotypic expression of Danon disease (LAMP2 cardiomyopathy)

Int J Cardiol. 2017 Oct 15:245:201-206. doi: 10.1016/j.ijcard.2017.06.031.

Abstract

Introduction: Danon disease is an X-linked lysosomal condition that causes a deficiency of lysosome-associated membrane protein 2 (LAMP2) gene. It is characterized clinically by a triad of skeletal myopathy, cardiomyopathy, and intellectual disability.

Methods: We examined clinical, echocardiographic, and genetic data on 5 patients with Danon disease, highlighting their clinical course and outcomes.

Results: All patients presented phenotypically with hypertrophic cardiomyopathy and later developed systolic dysfunction. The mean age at diagnosis was 19years (11-31years). All patients had diastolic dysfunction (mean e' of 5cm/s [3.5-6cm/s], mean E/e' of 17 [15-21]). Three patients required cardiac transplantation (ages 15, 27, and 42). Of the two deaths in this group, both were in women.

Conclusion: We highlight the aggressive cardiac phenotype of Danon disease in our clinical experience with rapid progression to end-stage cardiomyopathy; this progression occurred in both men and women. A timely diagnosis and an early referral for cardiac transplantation is crucial for improved outcomes.

Keywords: Danon disease; Echocardiography; Hypertrophic cardiomyopathy.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Female
  • Glycogen Storage Disease Type IIb / diagnostic imaging*
  • Glycogen Storage Disease Type IIb / genetics*
  • Glycogen Storage Disease Type IIb / surgery
  • Heart Transplantation / trends
  • Humans
  • Lysosomal-Associated Membrane Protein 2 / genetics*
  • Male
  • Phenotype*

Substances

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