LNK deficiency promotes acute aortic dissection and rupture

JCI Insight. 2018 Oct 18;3(20):e122558. doi: 10.1172/jci.insight.122558.

Abstract

Aortic dissection (AD) is a life-threatening vascular disease with limited treatment strategies. Here, we show that loss of the GWAS-identified SH2B3 gene, encoding lymphocyte adaptor protein LNK, markedly increases susceptibility to acute AD and rupture in response to angiotensin (Ang) II infusion. As early as day 3 following Ang II infusion, prior to the development of AD, Lnk-/- aortas display altered mechanical properties, increased elastin breaks, collagen thinning, enhanced neutrophil accumulation, and increased MMP-9 activity compared with WT mice. Adoptive transfer of Lnk-/- leukocytes into Rag1-/- mice induces AD and rupture in response to Ang II, demonstrating that LNK deficiency in hematopoietic cells plays a key role in this disease. Interestingly, treatment with doxycycline prevents the early accumulation of aortic neutrophils and significantly reduces the incidence of AD and rupture. PrediXcan analysis in a biobank of more than 23,000 individuals reveals that decreased expression of SH2B3 is significantly associated with increased frequency of AD-related phenotypes (odds ratio 0.81). Thus, we identified a role for LNK in the pathology of AD in experimental animals and humans and describe a new model that can be used to inform both inherited and acquired forms of this disease.

Keywords: Adaptor proteins; Cardiovascular disease; Inflammation; Innate immunity; Vascular Biology.

Publication types

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

MeSH terms

  • Adaptor Proteins, Signal Transducing / deficiency*
  • Adaptor Proteins, Signal Transducing / genetics*
  • Angiotensin II / administration & dosage
  • Animals
  • Aorta / drug effects
  • Aorta / pathology*
  • Aortic Dissection / chemically induced
  • Aortic Dissection / genetics*
  • Aortic Dissection / pathology
  • Aortic Rupture / chemically induced
  • Aortic Rupture / genetics*
  • Aortic Rupture / pathology
  • Disease Models, Animal
  • Female
  • Genetic Predisposition to Disease
  • Homeodomain Proteins / genetics
  • Homeodomain Proteins / metabolism
  • Humans
  • Male
  • Mice
  • Mice, Knockout

Substances

  • Adaptor Proteins, Signal Transducing
  • Homeodomain Proteins
  • Lnk protein, mouse
  • SH2B3 protein, human
  • Angiotensin II
  • RAG-1 protein