Biophysical characterization of the short QT mutation hERG-N588K reveals a mixed gain-and loss-of-function.

نویسندگان

  • Morten Grunnet
  • Thomas G Diness
  • Rie Schultz Hansen
  • Soren-Peter Olesen
چکیده

The short QT syndrome is a newly discovered pro-arrhythmic condition, which may cause ventricular fibrillation and sudden death. Short QT can originate from the apparent gain-of-function mutation N588K in the hERG potassium channel that conducts repolarising I(Kr) current. The present study describes a profound biophysical characterization of HERG-N588K revealing both loss-of-function and gain-of-function properties of the mutant. Experiments were conducted after heterologous expression in both Xenopus laevis oocytes and mammalian cells and at both room temperature and at 37 degrees C. Also the impact of the beta-subunits KCNE2 was investigated. The most prominent loss-of-function property of HERG-N588K was reduced tail currents but also the activation properties was compromised. Based on these biophysical results we suggest that the general view of HERG-N588K being a gain-of-function is modified to a mixed gain- and loss-of-function mutation. This might also have impact on the pathological picture of the HERG-N588K channels ability to trigger arrhythmic events.

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Comparative effects of the short QT N588K mutation at 37 degrees C on hERG K+ channel current during ventricular, Purkinje fibre and atrial action potentials: an action potential clamp study.

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عنوان ژورنال:
  • Cellular physiology and biochemistry : international journal of experimental cellular physiology, biochemistry, and pharmacology

دوره 22 5-6  شماره 

صفحات  -

تاریخ انتشار 2008