Unrestrained p38 MAPK activation in Dusp1/4 double-null mice induces cardiomyopathy

Circ Res. 2013 Jan 4;112(1):48-56. doi: 10.1161/CIRCRESAHA.112.272963. Epub 2012 Sep 19.

Abstract

Rationale: Mitogen-activated protein kinases (MAPKs) are activated in the heart by disease-inducing and stress-inducing stimuli, where they participate in hypertrophy, remodeling, contractility, and heart failure. A family of dual-specificity phosphatases (DUSPs) directly inactivates each of the MAPK terminal effectors, potentially serving a cardioprotective role.

Objective: To determine the role of DUSP1 and DUSP4 in regulating p38 MAPK function in the heart and the effect on disease.

Methods and results: Here, we generated mice and mouse embryonic fibroblasts lacking both Dusp1 and Dusp4 genes. Although single nulls showed no molecular effects, combined disruption of Dusp1/4 promoted unrestrained p38 MAPK activity in both mouse embryonic fibroblasts and the heart, with no change in the phosphorylation of c-Jun N-terminal kinases or extracellular signal-regulated kinases at baseline or with stress stimulation. Single disruption of either Dusp1 or Dusp4 did not result in cardiac pathology, although Dusp1/4 double-null mice exhibited cardiomyopathy and increased mortality with aging. Pharmacological inhibition of p38 MAPK with SB731445 ameliorated cardiomyopathy in Dusp1/4 double-null mice, indicating that DUSP1/4 function primarily through p38 MAPK in affecting disease. At the cellular level, unrestrained p38 MAPK activity diminished cardiac contractility and Ca2+ handling, which was acutely reversed with a p38 inhibitory compound. Poor function in Dusp1/4 double-null mice also was partially rescued by phospholamban deletion.

Conclusions: Our data demonstrate that Dusp1 and Dusp4 are cardioprotective genes that play a critical role in the heart by dampening p38 MAPK signaling that would otherwise reduce contractility and induce cardiomyopathy.

Publication types

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

MeSH terms

  • Animals
  • Calcium / metabolism
  • Calcium-Binding Proteins / deficiency
  • Calcium-Binding Proteins / genetics
  • Cardiomyopathies / diagnosis
  • Cardiomyopathies / enzymology*
  • Cardiomyopathies / genetics
  • Cardiomyopathies / physiopathology
  • Cardiomyopathies / prevention & control
  • Cells, Cultured
  • Disease Models, Animal
  • Dual Specificity Phosphatase 1 / deficiency*
  • Dual Specificity Phosphatase 1 / genetics
  • Enzyme Activation
  • Fibroblasts / enzymology
  • Gene Expression Regulation
  • Hemodynamics
  • Mice
  • Mice, 129 Strain
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Myocardial Contraction
  • Myocytes, Cardiac / drug effects
  • Myocytes, Cardiac / enzymology*
  • Myocytes, Cardiac / pathology
  • Phosphorylation
  • Protein Kinase Inhibitors / pharmacology
  • Protein Tyrosine Phosphatases
  • Signal Transduction*
  • Time Factors
  • p38 Mitogen-Activated Protein Kinases / antagonists & inhibitors
  • p38 Mitogen-Activated Protein Kinases / metabolism*

Substances

  • Calcium-Binding Proteins
  • Protein Kinase Inhibitors
  • phospholamban
  • p38 Mitogen-Activated Protein Kinases
  • Dual Specificity Phosphatase 1
  • Dusp1 protein, mouse
  • MKP2 protein, mouse
  • Protein Tyrosine Phosphatases
  • Calcium