Losartan Attenuates Degradation of Aorta and Lung Tissue Micromechanics in a Mouse Model of Severe Marfan Syndrome

Ann Biomed Eng. 2016 Oct;44(10):2994-3006. doi: 10.1007/s10439-016-1616-4. Epub 2016 Apr 18.

Abstract

Marfan syndrome (MFS) is an autosomal dominant disease of the connective tissue due to mutations in the fibrillin-1 gene (FBN1). This study aimed at characterizing microelastic properties of the ascending aortic wall and lung parenchyma tissues from wild type (WT) and age-matched Fbn1 hypomorphic mice (Fbn1(mgR/mgR) mice) to identify tissue-specific biomechanical effects of aging and disease in MFS. Atomic force microscopy was used to indent lung parenchyma and aortic wall tissues, using Hybrid Eshelby Decomposition analysis to extract layer-specific properties of the intima and media. The intima stiffened with age and was not different between WT and Fbn1(mgR/mgR) tissues, whereas the media layer of MFS aortas showed progressive structural and mechanical degradation with a modulus that was 50% softer than WT by 3.5 months of age. Similarly, MFS mice displayed progressive structural and mechanical deterioration of lung tissue, which was over 85% softer than WT by 3.5 months of age. Chronic treatment with the angiotensin type I receptor antagonist, losartan, attenuated the aorta and lung tissue degradation, resulting in structural and mechanical properties not significantly different from age-matched WT controls. By revealing micromechanical softening of elastin-rich aorta and lung tissues with disease progression in fibrillin-1 deficient mice, our findings support the use of losartan as a prophylactic treatment that may abrogate the life-threatening symptoms of MFS.

Keywords: Atomic force microscopy; Losartan; Lung biomechanics; Marfan syndrome; Vascular biomechanics.

Publication types

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

MeSH terms

  • Animals
  • Aorta* / metabolism
  • Aorta* / pathology
  • Aorta* / physiopathology
  • Disease Models, Animal
  • Fibrillin-1 / genetics
  • Fibrillin-1 / metabolism
  • Humans
  • Losartan / pharmacology*
  • Lung* / metabolism
  • Lung* / pathology
  • Lung* / physiopathology
  • Marfan Syndrome* / drug therapy
  • Marfan Syndrome* / genetics
  • Marfan Syndrome* / metabolism
  • Marfan Syndrome* / physiopathology
  • Mice
  • Mice, Transgenic

Substances

  • Fbn1 protein, mouse
  • Fibrillin-1
  • Losartan