Macrocyclic MEK1/2 inhibitor with efficacy in a mouse model of cardiomyopathy caused by lamin A/C gene mutation

Bioorg Med Chem. 2017 Feb 1;25(3):1004-1013. doi: 10.1016/j.bmc.2016.12.014. Epub 2016 Dec 9.

Abstract

Signaling mediated by extracellular signal-regulated kinases 1 and 2 (ERK1/2) is involved in numerous cellular processes. Mitogen-activated protein kinase kinases (MEK1/2) catalyze the phosphorylation of ERK1/2, converting it into an active kinase that regulates the expression of numerous genes and cellular processes. Inhibitors of MEK1/2 have demonstrated preclinical and clinical efficacy in certain cancers and types of cardiomyopathy. We report the synthesis of a novel, allosteric, macrocyclic MEK1/2 inhibitor that potently inhibits ERK1/2 activity in cultured cells and tissues of mice after systemic administration. Mice with dilated cardiomyopathy caused by a lamin A/C gene mutation have abnormally increased cardiac ERK1/2 activity. In these mice, this novel MEK1/2 inhibitor is well tolerated, improves left ventricular systolic function, decreases left ventricular fibrosis, has beneficial effects on skeletal muscle structure and pathology and prolongs survival. The novel MEK1/2 inhibitor described herein may therefore find clinical utility in the treatment of this rare cardiomyopathy, other types of cardiomyopathy and cancers in humans.

Keywords: Cardiomyopathy; Emery-Dreifuss muscular dystrophy; Extracellular signal-regulated kinase; Lamin; MEK inhibitor; Mitogen-activated protein kinase.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Animals
  • Cardiomyopathy, Dilated / drug therapy*
  • Cardiomyopathy, Dilated / genetics
  • Disease Models, Animal*
  • Dose-Response Relationship, Drug
  • Lamin Type A / genetics*
  • Macrocyclic Compounds / administration & dosage
  • Macrocyclic Compounds / chemistry
  • Macrocyclic Compounds / pharmacology*
  • Mice
  • Mice, Transgenic
  • Mitogen-Activated Protein Kinase 1 / antagonists & inhibitors*
  • Mitogen-Activated Protein Kinase 1 / metabolism
  • Molecular Structure
  • Mutation
  • Protein Kinase Inhibitors / administration & dosage
  • Protein Kinase Inhibitors / chemistry
  • Protein Kinase Inhibitors / pharmacology*
  • Structure-Activity Relationship

Substances

  • Lamin Type A
  • Macrocyclic Compounds
  • Protein Kinase Inhibitors
  • Mitogen-Activated Protein Kinase 1