Modest reductions of cardiac calsequestrin increase sarcoplasmic reticulum Ca2+ leak independent of luminal Ca2+ and trigger ventricular arrhythmias in mice

Circ Res. 2007 Sep 14;101(6):617-26. doi: 10.1161/CIRCRESAHA.107.157552. Epub 2007 Jul 26.

Abstract

Cardiac calsequestrin-null mice (Casq2-/-) display catecholaminergic ventricular tachycardia akin to humans with CASQ2 mutations. However, the specific contribution of Casq2 deficiency to the arrhythmia phenotype is difficult to assess because Casq2-/- mice also show significant reductions in the sarcoplasmic reticulum (SR) proteins junctin and triadin-1 and increased SR volume. Furthermore, it remains unknown whether Casq2 regulates SR Ca2+ release directly or indirectly by buffering SR luminal Ca2+. To address both questions, we examined heterozygous (Casq2+/-) mice, which have a 25% reduction in Casq2 but no significant decrease in other SR proteins. Casq2+/- mice (n=35) challenged with isoproterenol displayed 3-fold higher rates of ventricular ectopy than Casq2+/+ mice (n=31; P<0.05). Programmed stimulation induced significantly more ventricular tachycardia in Casq2+/- mice than in Casq2+/+ mice. Field-stimulated Ca2+ transients, cell shortening, L-type Ca2+ current, and SR volume were not significantly different in Casq2+/- and Casq2+/+ myocytes. However, in the presence of isoproterenol, SR Ca2+ leak was significantly increased in Casq2+/- myocytes (Casq2+/- 0.18+/-0.02 F(ratio) versus Casq2+/+ 0.11+/-0.01 F(ratio), n=57, 60; P<0.01), resulting in a significantly higher rate of spontaneous SR Ca2+ releases and triggered beats. SR luminal Ca2+ measured using Mag-Fura-2 was not altered by Casq2 reduction. As a result, the relationship between SR Ca2+ leak and SR luminal Ca2+ was significantly different between Casq2+/- and Casq2+/+ myocytes (P<0.01). Thus, even modest reductions in Casq2 increase SR Ca2+ leak and cause ventricular tachycardia susceptibility under stress. The underlying mechanism is likely the direct regulation of SR Ca2+ release channels by Casq2 rather than altered luminal Ca2+.

Publication types

  • Comparative Study
  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Calbindin 2
  • Calcium / metabolism
  • Calcium Channels / metabolism*
  • Calcium Signaling*
  • Calsequestrin / deficiency
  • Calsequestrin / genetics
  • Calsequestrin / metabolism*
  • Cardiac Pacing, Artificial
  • Diastole
  • Disease Models, Animal
  • Heart Rate
  • Ion Channel Gating*
  • Isoproterenol
  • Mice
  • Mice, Knockout
  • Mice, Transgenic
  • Myocardial Contraction
  • Myocytes, Cardiac / metabolism*
  • Myocytes, Cardiac / ultrastructure
  • S100 Calcium Binding Protein G / metabolism
  • Sarcoplasmic Reticulum / metabolism*
  • Sarcoplasmic Reticulum / ultrastructure
  • Tachycardia, Ventricular / chemically induced
  • Tachycardia, Ventricular / genetics
  • Tachycardia, Ventricular / metabolism*
  • Tachycardia, Ventricular / pathology
  • Tachycardia, Ventricular / physiopathology
  • Time Factors
  • Ventricular Function
  • Ventricular Premature Complexes / chemically induced
  • Ventricular Premature Complexes / genetics
  • Ventricular Premature Complexes / metabolism*
  • Ventricular Premature Complexes / pathology
  • Ventricular Premature Complexes / physiopathology

Substances

  • Calbindin 2
  • Calcium Channels
  • Calsequestrin
  • S100 Calcium Binding Protein G
  • casq2 protein, mouse
  • Isoproterenol
  • Calcium