A novel missense mutation causing a G487R substitution in the S2-S3 loop of human ether-à-go-go-related gene channel

J Cardiovasc Electrophysiol. 2012 Nov;23(11):1246-53. doi: 10.1111/j.1540-8167.2012.02383.x. Epub 2012 Jul 5.

Abstract

Introduction: Mutations of human ether-à-go-go-related gene (hERG), which encodes a cardiac K(+) channel responsible for the acceleration of the repolarizing phase of an action potential and the prevention of premature action potential regeneration, often cause severe arrhythmic disorders. We found a novel missense mutation of hERG that results in a G487R substitution in the S2-S3 loop of the channel subunit [hERG(G487R)] from a family and determined whether this mutant gene could induce an abnormality in channel function.

Methods and results: We made whole-cell voltage-clamp recordings from HEK-293T cells transfected with wild-type hERG [hERG(WT)], hERG(G487R), or both. We measured hERG channel-mediated current as the "tail" of a depolarization-elicited current. The current density of the tail current and its voltage- and time-dependences were not different among all the cell groups. The time-courses of deactivation, inactivation, and recovery from inactivation and their voltage-dependences were not different among all the cell groups. Furthermore, we performed immunocytochemical analysis using an anti-hERG subunit antibody. The ratio of the immunoreactivity of the plasma membrane to that of the cytoplasm was not different between cells transfected with hERG(WT), hERG(G487R), or both.

Conclusion: hERG(G487R) can produce functional channels with normal gating kinetics and cell-surface expression efficiency with or without the aid of hERG(WT). Therefore, neither the heterozygous nor homozygous inheritance of hERG(G487R) is thought to cause severe cardiac disorders. hERG(G487R) would be a candidate for a rare variant or polymorphism of hERG with an amino acid substitution in the unusual region of the channel subunit.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Action Potentials
  • Amino Acid Sequence
  • Amino Acid Substitution
  • Animals
  • Arrhythmias, Cardiac / genetics*
  • Arrhythmias, Cardiac / metabolism
  • Arrhythmias, Cardiac / mortality
  • Cell Membrane / metabolism
  • Death, Sudden, Cardiac / etiology*
  • ERG1 Potassium Channel
  • Ether-A-Go-Go Potassium Channels / chemistry
  • Ether-A-Go-Go Potassium Channels / genetics*
  • Ether-A-Go-Go Potassium Channels / metabolism
  • Female
  • Genetic Predisposition to Disease
  • HEK293 Cells
  • Humans
  • Immunohistochemistry
  • Ion Channel Gating
  • Kinetics
  • Male
  • Molecular Sequence Data
  • Mutation, Missense*
  • Patch-Clamp Techniques
  • Phenotype
  • Potassium / metabolism
  • Protein Subunits
  • Transfection
  • Young Adult

Substances

  • ERG1 Potassium Channel
  • Ether-A-Go-Go Potassium Channels
  • KCNH2 protein, human
  • Protein Subunits
  • Potassium