Mutation Load of Multiple Ion Channel Gene Mutations in Brugada Syndrome

Cardiology. 2017;137(4):256-260. doi: 10.1159/000471792. Epub 2017 May 12.

Abstract

Brugada syndrome is a primary arrhythmic syndrome that accounts for 20% of all sudden cardiac death cases in individuals with a structurally normal heart. Pathogenic variants associated with Brugada syndrome have been identified in over 19 genes, with SCN5A as a pivotal gene accounting for nearly 30% of cases. In contrast to other arrhythmogenic channelopathies (such as long QT syndrome), digenic inheritance has never been reported in Brugada syndrome. Exploring 66 cardiac genes using a new custom next-generation sequencing panel, we identified a double heterozygosity for pathogenic mutations in SCN5A and TRPM4 in a Brugada syndrome patient. The parents were heterozygous for each variation. This novel finding highlights the role of mutation load in Brugada syndrome and strongly suggests the adoption of a gene panel to obtain an accurate genetic diagnosis, which is mandatory for risk stratification, prevention, and therapy.

Keywords: Brugada syndrome; Digenic inheritance; Mutation load.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Brugada Syndrome / complications
  • Brugada Syndrome / genetics*
  • Child
  • Child, Preschool
  • Electrocardiography
  • Family
  • Female
  • Heterozygote
  • Humans
  • Long QT Syndrome / genetics
  • Male
  • Middle Aged
  • Mutation Rate
  • NAV1.5 Voltage-Gated Sodium Channel / genetics*
  • TRPM Cation Channels / genetics*

Substances

  • NAV1.5 Voltage-Gated Sodium Channel
  • SCN5A protein, human
  • TRPM Cation Channels
  • TRPM4 protein, human