{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T05:03:49Z","timestamp":1772168629208,"version":"3.50.1"},"reference-count":35,"publisher":"The Company of Biologists","issue":"20","license":[{"start":{"date-parts":[[2025,10,31]],"date-time":"2025-10-31T00:00:00Z","timestamp":1761868800000},"content-version":"vor","delay-in-days":16,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["T32GM148739"],"award-info":[{"award-number":["T32GM148739"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01AR066070"],"award-info":[{"award-number":["R01AR066070"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01AR071486"],"award-info":[{"award-number":["R01AR071486"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100006734","name":"Princeton University","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100006734","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,10,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Cfap298 is a highly conserved gene required for ciliary motility and dynein arm assembly, with known roles in left\u2013right (LR) patterning in zebrafish and links to human ciliopathies. Here, we describe a Cfap298 mutant allele, Cfap298\u0394\u0394S, which selectively disrupts LR axis establishment in mice. Mutant embryos display organ laterality defects and abnormal Nodal, Pitx2 and Lefty1 expression, consistent with an early disruption in LR symmetry breaking. LR asymmetry is established by leftward fluid flow in the node, generated by planar-polarized cilia. Although cfap298 mutations are reported to affect planar polarity, we did not observe changes in cilia position, length or CELSR1 localization within the node, suggesting that Cfap298\u0394\u0394S functions at the level of cilia motility. Accordingly, cilia lining the trachea of Cfap298\u0394\u0394S mutants fail to beat or beat incorrectly. Expression of the Cfap298\u0394\u0394S variant in zebrafish partially rescues body curvature defects but fails to rescue LR defects of cfap298 (kurly) loss-of-function mutants. These results confirm a conserved role for Cfap298 in mammalian LR patterning and identify a previously unreported region of CFAP298 with a conserved and essential role in cilia motility.<\/jats:p>","DOI":"10.1242\/jcs.264129","type":"journal-article","created":{"date-parts":[[2025,9,16]],"date-time":"2025-09-16T07:48:55Z","timestamp":1758008935000},"source":"Crossref","is-referenced-by-count":2,"title":["A conserved domain of\n                    <i>Cfap298<\/i>\n                    governs left\u2013right symmetry breaking in vertebrates"],"prefix":"10.1242","volume":"138","author":[{"given":"Marvin","family":"Cortez","sequence":"first","affiliation":[{"name":"Princeton University Department of Molecular Biology , , Princeton, NJ 08544,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6283-0531","authenticated-orcid":false,"given":"Cullen B.","family":"Young","sequence":"additional","affiliation":[{"name":"Princeton University Department of Molecular Biology , , Princeton, NJ 08544,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6993-5312","authenticated-orcid":false,"given":"Katherine A.","family":"Little","sequence":"additional","affiliation":[{"name":"Princeton University Department of Molecular Biology , , Princeton, NJ 08544,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0173-1887","authenticated-orcid":false,"given":"Daniel T.","family":"Grimes","sequence":"additional","affiliation":[{"name":"Princeton University Department of Molecular Biology , , Princeton, NJ 08544,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5464-259X","authenticated-orcid":false,"given":"Danelle","family":"Devenport","sequence":"additional","affiliation":[{"name":"Princeton University Department of Molecular Biology , , Princeton, NJ 08544,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6620-5015","authenticated-orcid":false,"given":"Rebecca D.","family":"Burdine","sequence":"additional","affiliation":[{"name":"Princeton University Department of Molecular Biology , , Princeton, NJ 08544,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2025,10,31]]},"reference":[{"key":"2025103109150981300_JCS264129C1","doi-asserted-by":"publisher","first-page":"493","DOI":"10.1038\/s41586-024-07487-w","article-title":"Accurate structure prediction of biomolecular interactions with AlphaFold 3","volume":"630","author":"Abramson","year":"2024","journal-title":"Nature"},{"key":"2025103109150981300_JCS264129C2","doi-asserted-by":"publisher","first-page":"773","DOI":"10.1016\/j.cell.2010.07.042","article-title":"Cell flow reorients the axis of planar polarity in the wing epithelium of Drosophila","volume":"142","author":"Aigouy","year":"2010","journal-title":"Cell"},{"key":"2025103109150981300_JCS264129C3","doi-asserted-by":"publisher","first-page":"e8999","DOI":"10.1371\/journal.pone.0008999","article-title":"Planar cell polarity enables posterior localization of nodal cilia and left-right axis determination during mouse and Xenopus embryogenesis","volume":"5","author":"Antic","year":"2010","journal-title":"PLoS ONE"},{"key":"2025103109150981300_JCS264129C4","doi-asserted-by":"publisher","first-page":"672","DOI":"10.1016\/j.ajhg.2013.08.015","article-title":"Zebrafish ciliopathy screen plus human mutational analysis identifies C21orf59 and CCDC65 defects as causing primary ciliary dyskinesia","volume":"93","author":"Austin-Tse","year":"2013","journal-title":"Am. 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