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Mol. Life Sci."],"published-print":{"date-parts":[[2024,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>\n                    Mutations in cysteine and glycine-rich protein 3 (CSRP3)\/muscle LIM protein (MLP), a key regulator of striated muscle function, have been linked to hypertrophic cardiomyopathy (HCM) and dilated cardiomyopathy (DCM) in patients. However, the roles of CSRP3 in heart development and regeneration are not completely understood. In this study, we characterized a novel zebrafish gene-trap line,\n                    <jats:italic>gSAIzGFFM218A<\/jats:italic>\n                    , which harbors an insertion in the\n                    <jats:italic>csrp3<\/jats:italic>\n                    genomic locus, heterozygous fish served as a\n                    <jats:italic>csrp3<\/jats:italic>\n                    expression reporter line and homozygous fish served as a\n                    <jats:italic>csrp3<\/jats:italic>\n                    mutant line. We discovered that\n                    <jats:italic>csrp3<\/jats:italic>\n                    is specifically expressed in larval ventricular cardiomyocytes (CMs) and that\n                    <jats:italic>csrp3<\/jats:italic>\n                    deficiency leads to excessive trabeculation, a common feature of CSRP3-related HCM and DCM. We further revealed that\n                    <jats:italic>csrp3<\/jats:italic>\n                    expression increased in response to different cardiac injuries and was regulated by several signaling pathways vital for heart regeneration. Csrp3 deficiency impeded zebrafish heart regeneration by impairing CM dedifferentiation, hindering sarcomere reassembly, and reducing CM proliferation while aggravating apoptosis. Csrp3 overexpression promoted CM proliferation after injury and ameliorated the impairment of ventricle regeneration caused by pharmacological inhibition of multiple signaling pathways. Our study highlights the critical role of Csrp3 in both zebrafish heart development and regeneration, and provides a valuable animal model for further functional exploration that will shed light on the molecular pathogenesis of CSRP3-related human cardiac diseases.\n                  <\/jats:p>","DOI":"10.1007\/s00018-024-05189-0","type":"journal-article","created":{"date-parts":[[2024,3,31]],"date-time":"2024-03-31T15:01:21Z","timestamp":1711897281000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["A novel gene-trap line reveals the dynamic patterns and essential roles of cysteine and glycine-rich protein 3 in zebrafish heart development and regeneration"],"prefix":"10.1007","volume":"81","author":[{"given":"Shuzhang","family":"Liang","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yating","family":"Zhou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yue","family":"Chang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jiayi","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Min","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peng","family":"Gao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qi","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hong","family":"Yu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Koichi","family":"Kawakami","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jinmin","family":"Ma","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6594-450X","authenticated-orcid":false,"given":"Ruilin","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,3,31]]},"reference":[{"issue":"1","key":"5189_CR1","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1016\/j.gene.2015.04.077","volume":"566","author":"E Vafiadaki","year":"2015","unstructured":"Vafiadaki E, Arvanitis DA, Sanoudou D (2015) Muscle LIM protein: master regulator of cardiac and skeletal muscle functions. 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