Heart-hand syndrome of Slovenian type: a new kind of laminopathy

J Med Genet. 2008 Oct;45(10):666-71. doi: 10.1136/jmg.2008.060020. Epub 2008 Jul 8.

Abstract

Background: Heart-hand syndromes are a heterogeneous group of genetic disorders characterised by the association of congenital cardiac disease and limb deformities. Laminopathies are a group of diseases caused by mutations in the LMNA gene encoding A-type lamins.

Results: We report a new LMNA mutation (c.1609-12T>G, IVS9-12 T>G) that creates a new cryptic splicing site with the retention of 11 intronic nucleotides in the mRNA. This LMNA mutation segregates with a new type of heart-hand syndrome in a previously reported family suffering from adult onset progressive conduction system disease, atrial and ventricular tachyarrhythmias, sudden death, dilated cardiomyopathy, and brachydactyly with predominant foot involvement. Analysis of the fibroblasts of two affected family members identified for the first time a truncated lamin A/C protein resulting from the frame shift created by the new splicing site, together with nuclear envelope abnormalities confirming that this LMNA mutation is pathogenic.

Conclusions: This new heart-hand syndrome should therefore be considered as a new kind of laminopathy. As part of laminopathies with heart involvement, patients presenting with this phenotype and their relatives are at risk for developing sudden cardiac death and should beneficiate from appropriate LMNA genetic diagnosis.

Publication types

  • Case Reports
  • Letter
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Female
  • Frameshift Mutation
  • Heart Defects, Congenital / complications
  • Heart Defects, Congenital / genetics*
  • Heterozygote
  • Humans
  • Lamin Type A / genetics*
  • Limb Deformities, Congenital / complications
  • Limb Deformities, Congenital / genetics*
  • Male
  • Middle Aged
  • Pedigree
  • RNA Splice Sites
  • RNA, Messenger / chemistry

Substances

  • LMNA protein, human
  • Lamin Type A
  • RNA Splice Sites
  • RNA, Messenger