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We created a zebrafish muscular laminopathy model using CRISPR\/Cas9 technology to target the zebrafish\n                      <jats:italic>lmna<\/jats:italic>\n                      gene.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Heterozygous and homozygous\n                      <jats:italic>lmna<\/jats:italic>\n                      mutants present skeletal muscle damage at 1\u00a0day post\u2010fertilization (dpf), and mobility impairment at 4 to 7 dpf. Cardiac structure and function analyses between 1 and 7 dpf show mild and transient defects in the\n                      <jats:italic>lmna<\/jats:italic>\n                      mutants compared to wild type (WT). Quantitative RT\u2010PCR analysis of genes implicated in striated muscle laminopathies show a decrease in\n                      <jats:italic>jun<\/jats:italic>\n                      and\n                      <jats:italic>nf\u03bab2<\/jats:italic>\n                      expression in 7 dpf homozygous\n                      <jats:italic>lmna<\/jats:italic>\n                      mutants compared to WT. Homozygous\n                      <jats:italic>lmna<\/jats:italic>\n                      mutants have a 1.26\u2010fold protein increase in activated Erk 1\/2, kinases associated with striated muscle laminopathies, compared to WT at 7 dpf. Activated Protein Kinase C alpha (Pkc \u03b1), a kinase that interacts with lamin A\/C and Erk 1\/2, is also upregulated in 7 dpf homozygous\n                      <jats:italic>lmna<\/jats:italic>\n                      mutants compared to WT.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      This study presents an animal model of skeletal muscle laminopathy where heterozygous and homozygous\n                      <jats:italic>lmna<\/jats:italic>\n                      mutants exhibit prominent skeletal muscle abnormalities during the first week of development. Furthermore, this is the first animal model that potentially implicates Pkc \u03b1 in muscular laminopathies.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.427","type":"journal-article","created":{"date-parts":[[2021,10,4]],"date-time":"2021-10-04T06:54:12Z","timestamp":1633330452000},"page":"645-661","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":16,"title":["A\n                    <scp>CRISPR<\/scp>\n                    \/Cas9 zebrafish lamin A\/C mutant model of muscular laminopathy"],"prefix":"10.1002","volume":"251","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6379-2135","authenticated-orcid":false,"given":"Hannah A.","family":"Nicolas","sequence":"first","affiliation":[{"name":"Department of Biology, Faculty of Science University of Ottawa  Ottawa Ontario 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