Cardiac dysfunction in Pkd1-deficient mice with phenotype rescue by galectin-3 knockout

Kidney Int. 2016 Sep;90(3):580-97. doi: 10.1016/j.kint.2016.04.028. Epub 2016 Jul 27.

Abstract

Alterations in myocardial wall texture stand out among ADPKD cardiovascular manifestations in hypertensive and normotensive patients. To elucidate their pathogenesis, we analyzed the cardiac phenotype in Pkd1(cond/cond)Nestin(cre) (CYG+) cystic mice exposed to increased blood pressure, at 5 to 6 and 20 to 24 weeks of age, and Pkd1(+/-) (HTG+) noncystic mice at 5-6 and 10-13 weeks. Echocardiographic analyses revealed decreased myocardial deformation and systolic function in CYG+ and HTG+ mice, as well as diastolic dysfunction in older CYG+ mice, compared to their Pkd1(cond/cond) and Pkd1(+/+) controls. Hearts from CYG+ and HTG+ mice presented reduced polycystin-1 expression, increased apoptosis, and mild fibrosis. Since galectin-3 has been associated with heart dysfunction, we studied it as a potential modifier of the ADPKD cardiac phenotype. Double-mutant Pkd1(cond/cond):Nestin(cre);Lgals3(-/-) (CYG-) and Pkd1(+/-);Lgals3(-/-) (HTG-) mice displayed improved cardiac deformability and systolic parameters compared to single-mutants, not differing from the controls. CYG- and HTG- showed decreased apoptosis and fibrosis. Analysis of a severe cystic model (Pkd1(V/V); VVG+) showed that Pkd1(V/V);Lgals3(-/-) (VVG-) mice have longer survival, decreased cardiac apoptosis and improved heart function compared to VVG+. CYG- and VVG- animals showed no difference in renal cystic burden compared to CYG+ and VVG+ mice. Thus, myocardial dysfunction occurs in different Pkd1-deficient models and suppression of galectin-3 expression rescues this phenotype.

Keywords: ADPKD; Pkd1-deficiency; apoptosis; cardiac dysfunction; cardiomyopathy; galectin-3.

Publication types

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

MeSH terms

  • Animals
  • Apoptosis / genetics
  • Cardiovascular Diseases / complications
  • Cardiovascular Diseases / genetics*
  • Disease Models, Animal
  • Echocardiography
  • Fibrosis
  • Galectin 3 / genetics*
  • Humans
  • Hypertension / etiology
  • Kidney / metabolism
  • Kidney / pathology
  • Mice
  • Mice, Knockout
  • Myocardium / pathology*
  • Phenotype
  • Polycystic Kidney, Autosomal Dominant / complications
  • Polycystic Kidney, Autosomal Dominant / genetics*
  • TRPP Cation Channels / genetics*
  • TRPP Cation Channels / metabolism

Substances

  • Galectin 3
  • Lgals3 protein, mouse
  • TRPP Cation Channels
  • polycystic kidney disease 1 protein