{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T16:00:53Z","timestamp":1772208053397,"version":"3.50.1"},"reference-count":46,"publisher":"The Company of Biologists","issue":"2","license":[{"start":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T00:00:00Z","timestamp":1772150400000},"content-version":"vor","delay-in-days":12,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["82070003"],"award-info":[{"award-number":["82070003"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["R01HD102186"],"award-info":[{"award-number":["R01HD102186"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["R01NS127879"],"award-info":[{"award-number":["R01NS127879"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["82270048"],"award-info":[{"award-number":["82270048"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100005326","name":"Yale University","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100005326","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,2,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>During embryogenesis, the establishment of left\u2013right (LR) asymmetry depends on directional fluid flow generated by motile cilia within the left\u2013right organizer (LRO). Disruption of this process can lead to laterality disorders such as situs inversus, heterotaxy, and congenital heart defects. Here, we identify CCDC57 as a regulator of ciliary function and LR patterning. Depletion of ccdc57 via morpholino oligonucleotides (MOs) led to abnormal cilia in the multiciliated cells of the embryonic epidermis of Xenopus. Additionally, LR markers, dand5 and pitx2c were misexpressed resulting in defects in normal rightward cardiac looping. Finally, we identified a patient with situs inversus carrying compound heterozygous CCDC57 missense variants. We tested these variants in Xenopus depleted of ccdc57. Wild-type human CCDC57 mRNA, but not the patient variants, rescued ciliary structure and function. These findings establish ccdc57 as a regulator of LR patterning and suggest its potential involvement in human laterality disorders.<\/jats:p>","DOI":"10.1242\/bio.062495","type":"journal-article","created":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T15:02:21Z","timestamp":1772204541000},"source":"Crossref","is-referenced-by-count":0,"title":["Ccdc57 regulates cilia and left-right patterning in\n                    <i>Xenopus<\/i>"],"prefix":"10.1242","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-1737-3493","authenticated-orcid":false,"given":"Binyi","family":"Yang","sequence":"first","affiliation":[{"name":"Second Xiangya Hospital, Central South University 1 Department of Pulmonary and Critical Care Medicine , , Changsha, Hunan 410011 ,","place":["China"]},{"name":"Yale University School of Medicine 2 Department of Pediatrics, Pediatric Genomics Discovery Program (PGDP) , , New Haven, CT 06510 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Emily K.","family":"Mis","sequence":"additional","affiliation":[{"name":"Yale University School of Medicine 2 Department of Pediatrics, Pediatric Genomics Discovery Program (PGDP) , , New Haven, CT 06510 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xianglin","family":"Zhou","sequence":"additional","affiliation":[{"name":"Second Xiangya Hospital, Central South University 1 Department of Pulmonary and Critical Care Medicine , , Changsha, Hunan 410011 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Faiza","family":"Aslam","sequence":"additional","affiliation":[{"name":"Yale University School of Medicine 2 Department of Pediatrics, Pediatric Genomics Discovery Program (PGDP) , , New Haven, CT 06510 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jie","family":"He","sequence":"additional","affiliation":[{"name":"Second Xiangya Hospital, Central South University 1 Department of Pulmonary and Critical Care Medicine , , Changsha, Hunan 410011 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiangyang","family":"Lu","sequence":"additional","affiliation":[{"name":"Second Xiangya Hospital, Central South University 1 Department of Pulmonary and Critical Care Medicine , , Changsha, Hunan 410011 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hui","family":"Fan","sequence":"additional","affiliation":[{"name":"Second Xiangya Hospital, Central South University 1 Department of Pulmonary and Critical Care Medicine , , Changsha, Hunan 410011 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ting","family":"Guo","sequence":"additional","affiliation":[{"name":"Second Xiangya Hospital, Central South University 1 Department of Pulmonary and Critical Care Medicine , , Changsha, Hunan 410011 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2999-0429","authenticated-orcid":false,"given":"Engin","family":"Deniz","sequence":"additional","affiliation":[{"name":"Yale University School of Medicine 2 Department of Pediatrics, Pediatric Genomics Discovery Program (PGDP) , , New Haven, CT 06510 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9501-651X","authenticated-orcid":false,"given":"Hong","family":"Luo","sequence":"additional","affiliation":[{"name":"Second Xiangya Hospital, Central South University 1 Department of Pulmonary and Critical Care Medicine , , Changsha, Hunan 410011 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9846-7076","authenticated-orcid":false,"given":"Mustafa K.","family":"Khokha","sequence":"additional","affiliation":[{"name":"Yale University School of Medicine 2 Department of Pediatrics, Pediatric Genomics Discovery Program (PGDP) , , New Haven, CT 06510 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2026,2,27]]},"reference":[{"key":"2026022710021401400_BIO062495C1","doi-asserted-by":"publisher","first-page":"130052","DOI":"10.1098\/rsob.130052","article-title":"Left-right asymmetry: cilia stir up new surprises in the node","volume":"3","author":"Babu","year":"2013","journal-title":"Open Biol."},{"key":"2026022710021401400_BIO062495C2","doi-asserted-by":"publisher","first-page":"151","DOI":"10.1016\/S0070-2153(08)00806-5","article-title":"Cilia multifunctional organelles at the center of vertebrate left-right asymmetry","volume":"85","author":"Basu","year":"2008","journal-title":"Curr. Top. Dev. Biol."},{"key":"2026022710021401400_BIO062495C3","doi-asserted-by":"publisher","first-page":"79","DOI":"10.1038\/ng.727","article-title":"The coiled-coil domain containing protein CCDC40 is essential for motile cilia function and left-right axis formation","volume":"43","author":"Becker-Heck","year":"2011","journal-title":"Nat. Genet."},{"key":"2026022710021401400_BIO062495C4","doi-asserted-by":"publisher","first-page":"464","DOI":"10.1016\/j.semcdb.2008.11.005","article-title":"Evolution of leftward flow","volume":"20","author":"Blum","year":"2009","journal-title":"Semin. Cell Dev. 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