Vascular endothelium-targeted Sirt7 gene therapy rejuvenates blood vessels and extends life span in a Hutchinson-Gilford progeria model

Sci Adv. 2020 Feb 19;6(8):eaay5556. doi: 10.1126/sciadv.aay5556. eCollection 2020 Feb.

Abstract

Vascular dysfunction is a typical characteristic of aging, but its contributing roles to systemic aging and the therapeutic potential are lacking experimental evidence. Here, we generated a knock-in mouse model with the causative Hutchinson-Gilford progeria syndrome (HGPS) LmnaG609G mutation, called progerin. The Lmnaf/f ;TC mice with progerin expression induced by Tie2-Cre exhibit defective microvasculature and neovascularization, accelerated aging, and shortened life span. Single-cell transcriptomic analysis of murine lung endothelial cells revealed a substantial up-regulation of inflammatory response. Molecularly, progerin interacts and destabilizes deacylase Sirt7; ectopic expression of Sirt7 alleviates the inflammatory response caused by progerin in endothelial cells. Vascular endothelium-targeted Sirt7 gene therapy, driven by an ICAM2 promoter, improves neovascularization, ameliorates aging features, and extends life span in Lmnaf/f ;TC mice. These data support endothelial dysfunction as a primary trigger of systemic aging and highlight gene therapy as a potential strategy for the clinical treatment of HGPS and age-related vascular dysfunction.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cellular Senescence
  • Disease Models, Animal
  • Endothelial Cells
  • Endothelium, Vascular / metabolism*
  • Gene Expression Profiling
  • Genetic Therapy* / methods
  • Humans
  • Longevity* / genetics
  • Mice
  • Mice, Knockout
  • Platelet Endothelial Cell Adhesion Molecule-1 / metabolism
  • Progeria / genetics*
  • Progeria / metabolism*
  • Progeria / therapy
  • Single-Cell Analysis
  • Sirtuins / genetics*
  • Vasodilation

Substances

  • Platelet Endothelial Cell Adhesion Molecule-1
  • Sirt7 protein, mouse
  • Sirtuins