IKs Gain- and Loss-of-Function in Early-Onset Lone Atrial Fibrillation

J Cardiovasc Electrophysiol. 2015 Jul;26(7):715-23. doi: 10.1111/jce.12666. Epub 2015 May 4.

Abstract

Introduction: Atrial fibrillation (AF) is the most frequent cardiac arrhythmia. The potassium current IKs is essential for cardiac repolarization. Gain-of-function mutation in KCNQ1, the gene encoding the pore-forming α-subunit of the IKs channel (KV 7.1), was the first ion channel dysfunction to be associated with familial AF. We hypothesized that early-onset lone AF is associated with a high prevalence of mutations in KCNQ1.

Methods and results: We bidirectionally sequenced the entire coding sequence of KCNQ1 in 209 unrelated patients with early-onset lone AF (<40 years) and investigated the identified mutations functionally in a heterologous expression system. We found 4 nonsynonymous KCNQ1 mutations (A46T, R195W, A302V, and R670K) in 4 unrelated patients (38, 31, 39, and 36 years, respectively). None of the mutations were present in the control group (n = 416 alleles). No other mutations were found in genes previously associated with AF. The mutations A46T, R195W, and A302V have previously been associated with long-QT syndrome. In line with previous reports, we found A302V to display a pronounced loss-of-function of the IKs current, while the other mutants exhibited a gain-of-function phenotype.

Conclusions: Mutations in the IKs channel leading to gain-of-function have previously been described in familial AF, yet this is the first time a loss-of-function mutation in KCNQ1 is associated with early-onset lone AF. These findings suggest that both gain-of-function and loss-of-function of cardiac potassium currents enhance the susceptibility to AF.

Keywords: cardiac arrhythmia; genetics; lone atrial fibrillation; molecular biology; potassium channel; trafficking; two-electrode voltage clamp.

Publication types

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

MeSH terms

  • Action Potentials
  • Adolescent
  • Adult
  • Atrial Fibrillation / diagnosis
  • Atrial Fibrillation / genetics*
  • Atrial Fibrillation / metabolism
  • Atrial Fibrillation / physiopathology
  • Atrial Fibrillation / therapy
  • Case-Control Studies
  • Cell Line
  • DNA Mutational Analysis
  • Denmark
  • Electrocardiography
  • Female
  • Genetic Predisposition to Disease
  • Heart Rate
  • Humans
  • KCNQ1 Potassium Channel / genetics*
  • KCNQ1 Potassium Channel / metabolism
  • Male
  • Mutation*
  • Myocytes, Cardiac / metabolism
  • Phenotype
  • Potassium / metabolism
  • Transfection
  • Young Adult

Substances

  • KCNQ1 Potassium Channel
  • KCNQ1 protein, human
  • Potassium