Transgenic overexpression of caveolin-3 in the heart induces a cardiomyopathic phenotype

被引:55
作者
Aravamudan, B
Volonte, D
Ramani, R
Gursoy, E
Lisanti, MP
London, B
Galbiati, F [1 ]
机构
[1] Univ Pittsburgh, Sch Med, Dept Pharmacol, Pittsburgh, PA 15261 USA
[2] Univ Pittsburgh, Sch Med, Cardiovasc Inst, Pittsburgh, PA 15261 USA
关键词
D O I
10.1093/hmg/ddg313
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
Caveolins are structural protein components of caveolar membrane domains. Caveolin-3, a muscle-specific member of the caveolin family, is expressed in skeletal muscle tissue and in the heart. The multiple roles that caveolin-3 plays in cellular physiology are becoming more apparent. We have shown that lack of caveolin-3 expression in skeletal muscle resembles limb-girdle muscular dystrophy-1C. In contrast, we have demonstrated that overexpression of caveolin-3 in skeletal muscle tissue promotes defects similar to those seen in Duchenne muscular dystrophy (DMD). Thus, a tight regulation of caveolin-3 expression is fundamental for normal muscle functions. Since caveolin-3 is also endogenously expressed in cardiac myocytes, and cardiomyopathies are observed in DMD patients, we looked at the effects of overexpression of caveolin-3 on cardiac structure and function by characterizing caveolin-3 transgenic mice. Our results indicate that overexpression of caveolin-3 causes severe cardiac tissue degeneration, fibrosis and a reduction in cardiac functions. We also show that dystrophin and its associated glycoproteins are down-regulated in caveolin-3 transgenic heart. In addition, we demonstrate that the activity of nitric oxide synthase (NOS) is down-regulated by high levels of caveolin-3 in the heart. Taken together, these results indicate that overexpression of caveolin-3 is sufficient to induce severe cardiomyopathy. In addition, these findings suggest that caveolin-3 transgenic mice may represent a valid mouse model for studying the molecular mechanisms underlying cardiomyopathies associated with Duchenne muscular dystrophy.
引用
收藏
页码:2777 / 2788
页数:12
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[11]   Caveolin-3 in myocardium and skeletal muscle in heart failure [J].
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Murakami, T ;
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Ohi, H ;
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[18]   Protective role of cardiac-specific overexpression of caveolin-3 in cirrhotic cardiomyopathy [J].
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