Literature DB >> 31427533

Disrupted mechanobiology links the molecular and cellular phenotypes in familial dilated cardiomyopathy.

Sarah R Clippinger1, Paige E Cloonan1, Lina Greenberg1, Melanie Ernst1, W Tom Stump1, Michael J Greenberg2.   

Abstract

Familial dilated cardiomyopathy (DCM) is a leading cause of sudden cardiac death and a major indicator for heart transplant. The disease is frequently caused by mutations of sarcomeric proteins; however, it is not well understood how these molecular mutations lead to alterations in cellular organization and contractility. To address this critical gap in our knowledge, we studied the molecular and cellular consequences of a DCM mutation in troponin-T, ΔK210. We determined the molecular mechanism of ΔK210 and used computational modeling to predict that the mutation should reduce the force per sarcomere. In mutant cardiomyocytes, we found that ΔK210 not only reduces contractility but also causes cellular hypertrophy and impairs cardiomyocytes' ability to adapt to changes in substrate stiffness (e.g., heart tissue fibrosis that occurs with aging and disease). These results help link the molecular and cellular phenotypes and implicate alterations in mechanosensing as an important factor in the development of DCM.

Entities:  

Keywords:  cardiomyopathy; contractility; mechanobiology; muscle; troponin

Mesh:

Substances:

Year:  2019        PMID: 31427533      PMCID: PMC6731759          DOI: 10.1073/pnas.1910962116

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  85 in total

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6.  Ca(2+)-desensitizing effect of a deletion mutation Delta K210 in cardiac troponin T that causes familial dilated cardiomyopathy.

Authors:  S Morimoto; Q-W Lu; K Harada; F Takahashi-Yanaga; R Minakami; M Ohta; T Sasaguri; I Ohtsuki
Journal:  Proc Natl Acad Sci U S A       Date:  2002-01-02       Impact factor: 11.205

7.  Single-molecule mechanics of R403Q cardiac myosin isolated from the mouse model of familial hypertrophic cardiomyopathy.

Authors:  M J Tyska; E Hayes; M Giewat; C E Seidman; J G Seidman; D M Warshaw
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