{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,18]],"date-time":"2026-06-18T14:06:37Z","timestamp":1781791597821,"version":"3.54.5"},"reference-count":62,"publisher":"Wiley","license":[{"start":{"date-parts":[[2025,12,19]],"date-time":"2025-12-19T00:00:00Z","timestamp":1766102400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100007786","name":"National Center for Child Health and Development","doi-asserted-by":"publisher","award":["R01HD099031"],"award-info":[{"award-number":["R01HD099031"]}],"id":[{"id":"10.13039\/100007786","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Dynamics"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Actin filament organization in cardiomyocytes critically depends on the formin Fhod3, but a role for Fhod3 in skeletal muscle development has not yet been described.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We demonstrate here that in zebrafish mutated for one of two\n                      <jats:italic>fhod3<\/jats:italic>\n                      paralog genes,\n                      <jats:italic>fhod3a<\/jats:italic>\n                      , skeletal muscle of the trunk appears normal through 2\u2009days post\u2010fertilization, but afterward exhibits myofibril damage, including gaps between myofibrils and myofibril fragmentation. Despite the progressive nature of the myofibril damage,\n                      <jats:italic>fhod3a<\/jats:italic>\n                      mutants differ from muscular dystrophy models in that damage is exacerbated by inhibition of muscle activity, and\n                      <jats:italic>fhod3a<\/jats:italic>\n                      mutants show no evidence of sarcolemma disruption. Rather, myofibril damage appears to coincide with growth of the contractile apparatus. We find neither the second\n                      <jats:italic>fhod3<\/jats:italic>\n                      paralog,\n                      <jats:italic>fhod3b<\/jats:italic>\n                      , nor the related\n                      <jats:italic>fhod1<\/jats:italic>\n                      contribute to embryonic skeletal muscle development, but fish individually mutated for\n                      <jats:italic>fhod3a<\/jats:italic>\n                      ,\n                      <jats:italic>fhod3b<\/jats:italic>\n                      , or\n                      <jats:italic>fhod1<\/jats:italic>\n                      are viable and appear grossly normal as adults. This may reflect redundancy in adults, as all three are expressed in many adult organs.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      These results indicate a\n                      <jats:italic>fhod3\u2010<\/jats:italic>\n                      encoded formin is dispensable for initial myofibril assembly in skeletal muscle but promotes myofibril stability during muscle fiber growth. This is the first demonstration in a vertebrate that Fhod3 contributes to skeletal muscle development.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.70108","type":"journal-article","created":{"date-parts":[[2025,12,19]],"date-time":"2025-12-19T14:13:07Z","timestamp":1766153587000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Fhod3 in zebrafish supports myofibril stability during growth of embryonic skeletal muscle"],"prefix":"10.1002","author":[{"given":"Aubrie","family":"Russell","sequence":"first","affiliation":[{"name":"Department of Cell and Developmental Biology State University of New York Upstate Medical University  Syracuse New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tracy","family":"Eng","sequence":"additional","affiliation":[{"name":"Department of Cell and Developmental Biology State University of New York Upstate Medical University  Syracuse New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey D.","family":"Amack","sequence":"additional","affiliation":[{"name":"Department of Cell and Developmental Biology State University of New York Upstate Medical University  Syracuse New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Pruyne","sequence":"additional","affiliation":[{"name":"Department of Cell and Developmental Biology State University of New York Upstate Medical University  Syracuse New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,12,19]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/cphy.c160033"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1097\/00003677\u2010200304000\u201000004"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrm2816"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.4161\/21541248.2014.973765"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1242\/bio.20121370"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.m109.059303"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201005060"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.7554\/elife.42144"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1161\/circgenetics.112.965277"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.7554\/elife.16540"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.e24\u201004\u20100145"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1072309"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncb834"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/s0092\u20108674(04)00210\u20107"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.200211078"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.adn9560"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature03251"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1038\/nsmb.2462"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.m117.800888"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0148472"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1021\/bi026520j"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0405902101"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.104048"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a006684"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/1097\u20100177(2000)9999:9999<::aid\u2010dvdy1065>3.0.co;2\u2010a"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.30860"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2023.11.001"},{"key":"e_1_2_9_29_1","unstructured":"ThisseB ThisseC.Fast release clones: a high throughput expression analysis. 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