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Ablation of cyclase-associated protein 2 (CAP2) leads to cardiomyopathy

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Ablation of cyclase-associated protein 2 (CAP2) leads to cardiomyopathy

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dc.contributor.author Peche, Vivek S. pl
dc.contributor.author Holak, Tadeusz [SAP14007479] pl
dc.contributor.author Burgute, Bhagyashri D. pl
dc.contributor.author Kosmas, Kosmas pl
dc.contributor.author Kale, Sushant P. pl
dc.contributor.author Wunderlich, Thomas F. pl
dc.contributor.author Elhamine, Fatiha pl
dc.contributor.author Stehle, Robert pl
dc.contributor.author Pfizer, Gabriele pl
dc.contributor.author Nohroudi, Klaus pl
dc.contributor.author Addicks, Klaus pl
dc.contributor.author Stöckigt, Florian pl
dc.contributor.author Schrickel, Jan W. pl
dc.contributor.author Gallinger, Julia pl
dc.contributor.author Schleicher, Michael pl
dc.contributor.author Noegel, Angelika A. pl
dc.date.accessioned 2015-06-18T07:19:43Z
dc.date.available 2015-06-18T07:19:43Z
dc.date.issued 2013 pl
dc.identifier.issn 1420-682X pl
dc.identifier.uri http://ruj.uj.edu.pl/xmlui/handle/item/9755
dc.language eng pl
dc.title Ablation of cyclase-associated protein 2 (CAP2) leads to cardiomyopathy pl
dc.type JournalArticle pl
dc.description.physical 527-543 pl
dc.description.additional Na publikacji autor podpisany: Tad A. Holak pl
dc.abstract.en Cyclase-associated proteins are highly con- served proteins that have a role in the regulation of actin dynamics. Higher eukaryotes have two isoforms, CAP1 and CAP2. To study the in vivo function of CAP2, we generated mice in which the CAP2 gene was inactivated by a gene-trap approach. Mutant mice showed a decrease in body weight and had a decreased survival rate. Further, they developed a severe cardiac defect marked by dilated cardiomyopathy (DCM) associated with drastic reduction in basal heart rate and prolongations in atrial and ventricular conduction times. Moreover, CAP2-deficient myofibrils exhibited reduced cooperativity of calcium- regulated force development. At the microscopic level, we observed disarrayed sarcomeres with development of fibrosis. We analyzed CAP2’s role in actin assembly and found that it sequesters G-actin and efficiently fragments filaments. This activity resides completely in its WASP homology domain. Thus CAP2 is an essential component of the myocardial sarcomere and is essential for physio- logical functioning of the cardiac system, and a deficiency leads to DCM and various cardiac defects. pl
dc.description.volume 70 pl
dc.description.number 3 pl
dc.identifier.doi 10.1007/s00018-012-1142-y pl
dc.identifier.eissn 1420-9071 pl
dc.title.journal Cellular and Molecular Life Sciences pl
dc.language.container eng pl
dc.affiliation Wydział Chemii : Zakład Chemii Organicznej pl
dc.subtype Article pl
dc.rights.original bez licencji pl
.pointsMNiSW [2013 A]: 40


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