Literature DB >> 35157045

Sunitinib inhibits STAT3 phosphorylation in cardiac muscle and prevents cardiomyopathy in the mdx mouse model of Duchenne muscular dystrophy.

Ariany Oliveira-Santos1, Marisela Dagda1, Dean J Burkin1.   

Abstract

Duchenne muscular dystrophy (DMD) is a fatal X-linked genetic disorder affecting approximately 1 in 5000 male births worldwide. DMD is caused by mutations in the dystrophin gene. Dystrophin is essential for maintaining muscle cell membrane integrity and stability by linking the cytoskeleton to the extracellular matrix, which protects myofibers from contraction-induced damage. Loss of dystrophin leads to mechanically induced skeletal and cardiac muscle damage. Although the disease is not evident in DMD patients at birth, muscular dystrophy rapidly progresses and results in respiratory and cardiac muscle failure as early as the teenage years. Premature death in DMD patients is due to cardiac arrhythmias and left ventricular dysfunction. Currently, there is no effective treatment for DMD-related cardiac failure. Recently, we have shown that a Food and Drug Administration-approved small molecule, sunitinib, a multi-targeted tyrosine kinase inhibitor can mitigate skeletal muscle disease through an increase in myogenic capacity, cell membrane integrity, and improvement of skeletal muscle function via regulation of STAT3-related signaling pathway. Chronic activation of STAT3 has been shown to promote cardiac hypertrophy and failure. In this study, we examined the effects of long-term sunitinib treatment on cardiac pathology and function. Our results showed sunitinib treatment reduced STAT3 phosphorylation in the heart muscle of mdx mice, improved cardiac electrical function, increased cardiac output and stroke volume, decreased ventricular hypertrophy, reduced cardiomyocytes membrane damage, fibrotic tissue deposition and slightly decreased cardiac inflammation. Together, our studies support the idea that sunitinib could serve as a novel treatment to slow cardiomyopathy progression in DMD. One Sentence Summary In this study, we determined if sunitinib, a Food and Drug Administration-approved drug, could reduce the pathology and improve cardiac function in an animal model for DMD.
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Year:  2022        PMID: 35157045      PMCID: PMC9307308          DOI: 10.1093/hmg/ddac042

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   5.121


  78 in total

Review 1.  Duchenne muscular dystrophy.

Authors:  Eppie M Yiu; Andrew J Kornberg
Journal:  J Paediatr Child Health       Date:  2015-03-09       Impact factor: 1.954

2.  Increased connective tissue growth factor associated with cardiac fibrosis in the mdx mouse model of dystrophic cardiomyopathy.

Authors:  Carol G Au; Tanya L Butler; Megan C Sherwood; Jonathan R Egan; Kathryn N North; David S Winlaw
Journal:  Int J Exp Pathol       Date:  2010-12-01       Impact factor: 1.925

3.  Evidence-based path to newborn screening for Duchenne muscular dystrophy.

Authors:  Jerry R Mendell; Chris Shilling; Nancy D Leslie; Kevin M Flanigan; Roula al-Dahhak; Julie Gastier-Foster; Kelley Kneile; Diane M Dunn; Brett Duval; Alexander Aoyagi; Cindy Hamil; Maha Mahmoud; Kandice Roush; Lauren Bird; Chelsea Rankin; Heather Lilly; Natalie Street; Ram Chandrasekar; Robert B Weiss
Journal:  Ann Neurol       Date:  2012-03       Impact factor: 10.422

4.  Decreased myocardial nNOS, increased iNOS and abnormal ECGs in mouse models of Duchenne muscular dystrophy.

Authors:  B L Bia; P J Cassidy; M E Young; J A Rafael; B Leighton; K E Davies; G K Radda; K Clarke
Journal:  J Mol Cell Cardiol       Date:  1999-10       Impact factor: 5.000

Review 5.  Of mice and men: from early NMR studies of the heart to physiological genomics.

Authors:  G K Radda
Journal:  Biochem Biophys Res Commun       Date:  1999-12-29       Impact factor: 3.575

6.  In vivo MRI characterization of progressive cardiac dysfunction in the mdx mouse model of muscular dystrophy.

Authors:  Daniel J Stuckey; Carolyn A Carr; Patrizia Camelliti; Damian J Tyler; Kay E Davies; Kieran Clarke
Journal:  PLoS One       Date:  2012-01-03       Impact factor: 3.240

7.  Sunitinib prevents cachexia and prolongs survival of mice bearing renal cancer by restraining STAT3 and MuRF-1 activation in muscle.

Authors:  Francesca Pretto; Carmen Ghilardi; Michele Moschetta; Andrea Bassi; Alessandra Rovida; Valentina Scarlato; Laura Talamini; Fabio Fiordaliso; Cinzia Bisighini; Giovanna Damia; Maria Rosa Bani; Rosanna Piccirillo; Raffaella Giavazzi
Journal:  Oncotarget       Date:  2015-02-20

8.  Increased Circulating Levels of Interleukin-6 Induce Perturbation in Redox-Regulated Signaling Cascades in Muscle of Dystrophic Mice.

Authors:  Laura Pelosi; Laura Forcina; Carmine Nicoletti; Bianca Maria Scicchitano; Antonio Musarò
Journal:  Oxid Med Cell Longev       Date:  2017-08-06       Impact factor: 6.543

Review 9.  Cardiac Pathophysiology and the Future of Cardiac Therapies in Duchenne Muscular Dystrophy.

Authors:  Tatyana A Meyers; DeWayne Townsend
Journal:  Int J Mol Sci       Date:  2019-08-22       Impact factor: 5.923

Review 10.  Biomarkers for Duchenne muscular dystrophy: myonecrosis, inflammation and oxidative stress.

Authors:  Miranda D Grounds; Jessica R Terrill; Basma A Al-Mshhdani; Marisa N Duong; Hannah G Radley-Crabb; Peter G Arthur
Journal:  Dis Model Mech       Date:  2020-03-02       Impact factor: 5.758

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  1 in total

Review 1.  The role of the dystrophin glycoprotein complex in muscle cell mechanotransduction.

Authors:  Darren Graham Samuel Wilson; Andrew Tinker; Thomas Iskratsch
Journal:  Commun Biol       Date:  2022-09-27
  1 in total

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