Drug-induced long-QT syndrome associated with a subclinical SCN5A mutation

Circulation. 2002 Sep 3;106(10):1269-74. doi: 10.1161/01.cir.0000027139.42087.b6.

Abstract

Background: Subclinical mutations in genes associated with the congenital long-QT syndromes (LQTS) have been suggested as a risk factor for drug-induced LQTS and accompanying life-threatening arrhythmias. Recent studies have identified genetic variants of the cardiac K+ channel genes predisposing affected individuals to acquired LQTS. We have identified a novel Na+ channel mutation in an individual who exhibited drug-induced LQTS.

Methods and results: An elderly Japanese woman with documented QT prolongation and torsade de pointes during treatment with the prokinetic drug cisapride underwent mutational analysis of LQTS-related genes. A novel missense mutation (L1825P) was identified within the C-terminus region of the cardiac Na+ channel (SCN5A). The L1825P channel heterologously expressed in tsA-201 cells showed Na+ current with slow decay and a prominent tetrodotoxin-sensitive noninactivating component, similar to the gain-of-function phenotype most commonly observed for SCN5A-associated congenital LQTS (LQT3). In addition, L1825P exhibited loss of function Na+ channel features characteristic of Brugada syndrome. Peak Na+ current density observed in cells expressing L1825P was significantly diminished, and the voltage dependence of activation and inactivation was shifted toward more positive and negative potentials, respectively.

Conclusions: This study demonstrates that subclinical mutations in the LQTS-related gene SCN5A may predispose certain individuals to drug-induced cardiac arrhythmias.

Publication types

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

MeSH terms

  • Aged
  • Amino Acid Sequence
  • Base Sequence
  • Cell Line
  • Cisapride / adverse effects
  • Electric Conductivity
  • Electrocardiography
  • Female
  • Genetic Predisposition to Disease*
  • Humans
  • Ion Channel Gating
  • Long QT Syndrome / chemically induced*
  • Long QT Syndrome / diagnosis
  • Long QT Syndrome / genetics*
  • Mutation, Missense*
  • NAV1.5 Voltage-Gated Sodium Channel
  • Pedigree
  • Polymorphism, Single-Stranded Conformational
  • Potassium Channels / genetics
  • Sodium Channels / chemistry
  • Sodium Channels / genetics*
  • Sodium Channels / physiology

Substances

  • NAV1.5 Voltage-Gated Sodium Channel
  • Potassium Channels
  • SCN5A protein, human
  • Sodium Channels
  • Cisapride