Cardiac and skeletal muscle manifestations in the G608G mouse model of Hutchinson-Gilford progeria syndrome

Aging Cell. 2024 Oct;23(10):e14259. doi: 10.1111/acel.14259. Epub 2024 Jul 3.

Abstract

Hutchinson-Gilford progeria syndrome (HGPS) is a rare premature aging disorder resulting from de novo mutations in the lamin A gene. Children with HGPS typically pass away in their teenage years due to cardiovascular diseases such as atherosclerosis, myocardial infarction, heart failure, and stroke. In this study, we characterized the G608G HGPS mouse model and explored cardiac and skeletal muscle function, along with senescence-associated phenotypes in fibroblasts. Homozygous G608G HGPS mice exhibited cardiac dysfunction, including decreased cardiac output and stroke volume, and impaired left ventricle relaxation. Additionally, skeletal muscle exhibited decreased isometric tetanic torque, muscle atrophy, and increased fibrosis. HGPS fibroblasts showed nuclear abnormalities, decreased proliferation, and increased expression of senescence markers. These findings provide insights into the pathophysiology of the G608G HGPS mouse model and inform potential therapeutic strategies for HGPS.

Keywords: G608G HGPS mouse model; Hutchinson‐Gilford progeria syndrome; cardiomyopathy; fibroblasts; myopathy; senescence.

MeSH terms

  • Animals
  • Disease Models, Animal*
  • Fibroblasts / metabolism
  • Fibroblasts / pathology
  • Humans
  • Lamin Type A / genetics
  • Lamin Type A / metabolism
  • Male
  • Mice
  • Muscle, Skeletal* / metabolism
  • Muscle, Skeletal* / pathology
  • Myocardium / metabolism
  • Myocardium / pathology
  • Progeria* / genetics
  • Progeria* / metabolism
  • Progeria* / pathology

Substances

  • Lamin Type A