Transforming growth factor β receptor inhibition prevents ventricular fibrosis in a mouse model of progressive cardiac conduction disease

Cardiovasc Res. 2017 Apr 1;113(5):464-474. doi: 10.1093/cvr/cvx026.

Abstract

Aims: Loss-of-function mutations in SCN5A, the gene encoding NaV1.5 channel, have been associated with inherited progressive cardiac conduction disease (PCCD). We have proposed that Scn5a heterozygous knock-out (Scn5a+/-) mice, which are characterized by ventricular fibrotic remodelling with ageing, represent a model for PCCD. Our objectives were to identify the molecular pathway involved in fibrosis development and prevent its activation.

Methods and results: Our study shows that myocardial interstitial fibrosis occurred in Scn5a+/- mice only after 45 weeks of age. Fibrosis was triggered by transforming growth factor β (TGF-β) pathway activation. Younger Scn5a+/- mice were characterized by a higher connexin 43 expression than wild-type (WT) mice. After the age of 45 weeks, connexin 43 expression decreased in both WT and Scn5a+/- mice, although the decrease was larger in Scn5a+/- mice. Chronic inhibition of cardiac sodium current with flecainide (50 mg/kg/day p.o) in WT mice from the age of 6 weeks to the age of 60 weeks did not lead to TGF-β pathway activation and fibrosis. Chronic inhibition of TGF-β receptors with GW788388 (5 mg/kg/day p.o.) in Scn5a+/- mice from the age of 45 weeks to the age of 60 weeks prevented the occurrence of fibrosis. However, current data could not detect reduction in QRS duration with GW788388.

Conclusion: Myocardial fibrosis secondary to a loss of NaV1.5 is triggered by TGF-β signalling pathway. Those events are more likely secondary to the decreased NaV1.5 sarcolemmal expression rather than the decreased Na+ current per se. TGF-β receptor inhibition prevents age-dependent development of ventricular fibrosis in Scn5a+/- mouse.

Keywords: CTGF; Connexin 43; GW788388; NaV1.5; Smad2/3.

MeSH terms

  • Age Factors
  • Animals
  • Anti-Arrhythmia Agents / pharmacology
  • Arrhythmias, Cardiac / drug therapy*
  • Arrhythmias, Cardiac / genetics
  • Arrhythmias, Cardiac / metabolism
  • Arrhythmias, Cardiac / physiopathology
  • Benzamides / pharmacology*
  • Cardiomyopathies / genetics
  • Cardiomyopathies / metabolism
  • Cardiomyopathies / physiopathology
  • Cardiomyopathies / prevention & control*
  • Connexin 43 / metabolism
  • Disease Models, Animal
  • Female
  • Fibrosis
  • Flecainide / pharmacology
  • Genetic Predisposition to Disease
  • Heart Conduction System / drug effects*
  • Heart Conduction System / metabolism
  • Heart Conduction System / physiopathology
  • Heart Rate
  • Heart Ventricles / drug effects*
  • Heart Ventricles / metabolism
  • Heart Ventricles / physiopathology
  • Heterozygote
  • Kinetics
  • Male
  • Membrane Potentials
  • Mice, 129 Strain
  • Mice, Knockout
  • NAV1.5 Voltage-Gated Sodium Channel / deficiency
  • NAV1.5 Voltage-Gated Sodium Channel / genetics
  • Phenotype
  • Pyrazoles / pharmacology*
  • Receptors, Transforming Growth Factor beta / antagonists & inhibitors*
  • Receptors, Transforming Growth Factor beta / metabolism
  • Signal Transduction / drug effects
  • Transforming Growth Factor beta / metabolism
  • Ventricular Remodeling / drug effects*
  • Voltage-Gated Sodium Channel Blockers / pharmacology

Substances

  • 4-(4-(3-(pyridin-2-yl)-1H-pyrazol-4-yl)pyridin-2-yl)-N-(tetrahydro-2H-pyran-4-yl)benzamide
  • Anti-Arrhythmia Agents
  • Benzamides
  • Connexin 43
  • GJA1 protein, mouse
  • NAV1.5 Voltage-Gated Sodium Channel
  • Pyrazoles
  • Receptors, Transforming Growth Factor beta
  • Scn5a protein, mouse
  • Transforming Growth Factor beta
  • Voltage-Gated Sodium Channel Blockers
  • Flecainide