Long QT syndrome and dilated cardiomyopathy with SCN5A p.R1193Q polymorphism: cardioverter-defibrillator implantation at 27 months

Pacing Clin Electrophysiol. 2012 Aug;35(8):e243-6. doi: 10.1111/j.1540-8159.2012.03409.x. Epub 2012 Apr 22.

Abstract

Cardiac sodium channel dysfunction associated with the SCN5A gene presents with mixed phenotypes, including long QT syndrome type 3, sinus node dysfunction, and dilated cardiomyopathy (DCM). We report a Korean case of an overlap syndrome of cardiac sodium channelopathy with SCN5A p.R1193Q polymorphism, treated by the placement of an intrapericardial implantable cardioverter-defibrillator (ICD) at the age of 27 months. Although the patient received two appropriate life-saving shocks for ventricular fibrillations, he eventually died of DCM progression. However, this case shows that intrapericardial ICD implantation is feasible in young children with a high risk for sudden cardiac death.

Publication types

  • Case Reports

MeSH terms

  • Cardiomyopathy, Dilated / genetics
  • Cardiomyopathy, Dilated / therapy*
  • Channelopathies / genetics
  • Child, Preschool
  • Defibrillators, Implantable*
  • Disease Progression
  • Fatal Outcome
  • Humans
  • Long QT Syndrome / genetics*
  • Long QT Syndrome / therapy*
  • Male
  • NAV1.5 Voltage-Gated Sodium Channel / genetics*
  • Polymorphism, Genetic*
  • Ventricular Fibrillation / genetics
  • Ventricular Fibrillation / therapy

Substances

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