Genetic deletion of CMG2 exacerbates systemic-to-pulmonary shunt-induced pulmonary arterial hypertension

FASEB J. 2021 Apr;35(4):e21421. doi: 10.1096/fj.202000299R.

Abstract

Pulmonary arterial hypertension (PAH) secondary to congenital heart disease (CHD-PAH) with systemic-to-pulmonary shunt (SPS) is characterized by proliferative vascular remodeling. Capillary morphogenesis gene-2 (CMG2) plays a key role in cell proliferation and apoptosis. This study aimed to determine the role of CMG2 in the pathogenesis of SPS-induced PAH. CMG2 levels were significantly downregulated in pulmonary arterioles from patients with Eisenmenger syndrome and rats with SPS-induced PAH. CMG2 was highly expressed in several cells including human pulmonary arterial smooth muscle cells (HPASMCs). CMG2-/- rats exhibited more severe PAH and pulmonary vascular remodeling than wild-type rats when exposed to SPS for 8 weeks. Overexpression of CMG2 significantly inhibited proliferation and promoted apoptosis of HPASMCs, while knockdown of CMG2 promoted cell proliferation and inhibited cell apoptosis. Next-generation sequencing and subsequent validation results suggested that PI3K-AKT was the most prominent signaling pathway regulated by differentially expressed genes (DEGs) in CMG2-/- rat lungs. Our work identified a novel role for CMG2 in SPS-induced PAH based on the findings that CMG2 deficiency exacerbates SPS-induced vascular remodeling in the development of PAH, indicating that CMG2 might act as a potential target for the treatment of CHD-PAH.

Keywords: capillary morphogenesis gene-2; congenital heart disease; pulmonary vascular remodeling.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Animals
  • Apoptosis
  • Case-Control Studies
  • Cell Line
  • Cell Proliferation
  • Down-Regulation
  • Eisenmenger Complex / metabolism*
  • Endothelial Cells
  • Gene Deletion
  • Gene Expression Regulation
  • Humans
  • Male
  • Middle Aged
  • Muscle, Smooth, Vascular
  • Myocytes, Smooth Muscle
  • Pulmonary Arterial Hypertension / genetics*
  • Pulmonary Arterial Hypertension / pathology*
  • Rats
  • Receptors, Peptide
  • Vascular Remodeling / physiology
  • Young Adult

Substances

  • ANTXR2 protein, human
  • Receptors, Peptide