Document Detail


Interplay between heart and skeletal muscle disease in heart failure: the 2011 George E. Brown Memorial Lecture.
MedLine Citation:
PMID:  22383709     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The study of single gene disorders often provides insight for more complex human disease. Mutations in the genes encoding the dystrophin protein complex cause muscular dystrophy and cardiomyopathy by destabilizing the plasma membrane of skeletal myofibers and cardiomyocytes. In these diseases, progressive skeletal muscle degeneration and weakness contribute to cardiac dysfunction. Moreover, the pace and pattern of muscle weakness, along with onset of cardiomyopathy, is highly variable even when associated with the same identical mutation. Using a mouse model of muscular dystrophy and cardiomyopathy, we identified genetic loci that modify muscle pathology and cardiac fibrosis. Distinct genetic modifiers were identified for diaphragm and abdominal musculature, and these genetic intervals differ from those that regulate pathology in the skeletal muscle of the limbs and the heart. One modifier gene was identified and highlights the importance of the transforming growth factor-β pathway in the pathogenesis of muscular dystrophy and cardiomyopathy. We determined that canonical transforming growth factor-β signaling contributes to heart and muscle dysfunction using a Drosophila model. Together, these studies demonstrate the value of using a genetically sensitized model to uncover pathways that regulate heart failure and muscle weakness.
Authors:
Elizabeth M McNally; Jeffery A Goldstein
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Publication Detail:
Type:  Lectures; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Circulation research     Volume:  110     ISSN:  1524-4571     ISO Abbreviation:  Circ. Res.     Publication Date:  2012 Mar 
Date Detail:
Created Date:  2012-03-02     Completed Date:  2012-05-03     Revised Date:  2013-04-15    
Medline Journal Info:
Nlm Unique ID:  0047103     Medline TA:  Circ Res     Country:  United States    
Other Details:
Languages:  eng     Pagination:  749-54     Citation Subset:  IM    
Affiliation:
Department of Medicine, University of Chicago, Chicago, IL 60637, USA. emcnally@uchicago.edu
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MeSH Terms
Descriptor/Qualifier:
Animals
Cardiomyopathies / physiopathology
Disease Models, Animal
Drosophila
Heart / physiopathology*
Heart Failure / physiopathology*
Mice
Muscle, Skeletal / physiopathology*
Muscular Dystrophies / physiopathology
Signal Transduction / physiology
Transforming Growth Factor beta / physiology
Grant Support
ID/Acronym/Agency:
P01 NS072027-01A1/NS/NINDS NIH HHS; R01 AR050703-02/AR/NIAMS NIH HHS; R01 HL061322-10S1/HL/NHLBI NIH HHS; R01 HL061322-11/HL/NHLBI NIH HHS
Chemical
Reg. No./Substance:
0/Transforming Growth Factor beta
Comments/Corrections

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