Whole-exome sequencing identifies Y1495X of SCN5A to be associated with familial conduction disease and sudden death

Sci Rep. 2014 Jul 10:4:5616. doi: 10.1038/srep05616.

Abstract

SCN5A mutations have been reported to underlie a variety of inherited arrhythmias, while the complex overlapping phenotype, especially with congenital heart disease (CHD), is rarely reported. The 48-year-old proband underwent a recent syncope during rest. A CHD (tetralogy of Fallot) and conduction disease was revealed by echocardiogram and ultrasonic cardiogram examination. We combined whole-exome sequencing (WES) and bioinformatics strategies to identify the pathogenic gene for this autosomal-dominant cardiac conduction disease (CCD) in a multi-generation pedigree. We examined four members of this family, including three affected and one unaffected. A novel nonsense mutation (Y1495X) in SCN5A was identified in the affected family members. This mutation is predicted to generate a truncated SCN5A protein, which could result in the loss of sodium current, a defined mechanism of SCN5A related arrhythmias. Our study provides evidence that WES is a highly effective approach for genetic analyses of rare clinical phenotypes. Our study also offers accurate genetic testing information for those yet clinically negative relatives.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Arrhythmias, Cardiac / etiology*
  • Arrhythmias, Cardiac / genetics*
  • Brugada Syndrome
  • Cardiac Conduction System Disease
  • Child
  • Child, Preschool
  • Codon, Nonsense / genetics
  • Death, Sudden / etiology*
  • Exome / genetics*
  • Female
  • Genetic Predisposition to Disease / genetics*
  • Heart Conduction System / abnormalities*
  • Humans
  • Male
  • Middle Aged
  • NAV1.5 Voltage-Gated Sodium Channel / genetics*
  • Pedigree
  • Phenotype
  • Sodium / metabolism

Substances

  • Codon, Nonsense
  • NAV1.5 Voltage-Gated Sodium Channel
  • SCN5A protein, human
  • Sodium