{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"institution":[{"name":"bioRxiv"}],"indexed":{"date-parts":[[2026,7,30]],"date-time":"2026-07-30T14:00:42Z","timestamp":1785420042518,"version":"3.56.0"},"posted":{"date-parts":[[2025,5,17]]},"group-title":"Developmental Biology","reference-count":29,"publisher":"openRxiv","license":[{"start":{"date-parts":[[2025,5,17]],"date-time":"2025-05-17T00:00:00Z","timestamp":1747440000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"award":["NIGMS - T32GM148739"],"award-info":[{"award-number":["NIGMS - T32GM148739"]}],"id":[{"id":"https:\/\/ror.org\/01cwqze88","id-type":"ROR","asserted-by":"publisher"}]},{"award":["NIAMS - R01AR071486"],"award-info":[{"award-number":["NIAMS - R01AR071486"]}],"id":[{"id":"https:\/\/ror.org\/01cwqze88","id-type":"ROR","asserted-by":"publisher"}]},{"award":["NIAMS - R01AR066070"],"award-info":[{"award-number":["NIAMS - R01AR066070"]}],"id":[{"id":"https:\/\/ror.org\/01cwqze88","id-type":"ROR","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"accepted":{"date-parts":[[2025,5,17]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                <jats:p>\n                  <jats:italic>Cfap298<\/jats:italic>\n                  is a highly conserved gene required for ciliary motility and dynein arm assembly. It plays a known role in Left-Right (LR) patterning in zebrafish and is linked to human ciliopathies. Here we describe a new\n                  <jats:italic>Cfap298<\/jats:italic>\n                  mutant allele,\n                  <jats:italic>\n                    Cfap298\n                    <jats:sup>\u03943aa<\/jats:sup>\n                  <\/jats:italic>\n                  , which selectively interferes with LR axis establishment in mice. Mutant embryos display a range of laterality defects including\n                  <jats:italic>situs solitus<\/jats:italic>\n                  ,\n                  <jats:italic>situs inversus<\/jats:italic>\n                  , and\n                  <jats:italic>situs ambiguous,<\/jats:italic>\n                  as indicated by abnormal heart, lung, and stomach positioning. At embryonic day 8.5, mutant embryos display abnormal\n                  <jats:italic>Nodal<\/jats:italic>\n                  ,\n                  <jats:italic>Pitx2<\/jats:italic>\n                  , and\n                  <jats:italic>Lefty1<\/jats:italic>\n                  expression patterns in the lateral plate mesoderm and midline, consistent with an early disruption in LR symmetry breaking. In mice, LR asymmetry is established by leftward fluid flow in the node, generated by planar-polarized cilia. Although\n                  <jats:italic>cfap298<\/jats:italic>\n                  mutations are reported to affect cell polarity, we did not observe changes in cilia position, length, or planar cell polarity protein localization within the node, suggesting that\n                  <jats:italic>\n                    Cfap298\n                    <jats:sup>\u03943aa<\/jats:sup>\n                  <\/jats:italic>\n                  functions at the level of cilia motility. Consistently, motile cilia lining the trachea of\n                  <jats:italic>\n                    Cfap298\n                    <jats:sup>\u03943aa<\/jats:sup>\n                  <\/jats:italic>\n                  mutants fail to beat. Expression of the\n                  <jats:italic>\n                    Cfap298\n                    <jats:sup>\u03943aa<\/jats:sup>\n                  <\/jats:italic>\n                  variant in zebrafish fails to rescue the LR defects of\n                  <jats:italic>cfap298<\/jats:italic>\n                  (\n                  <jats:italic>kurly)<\/jats:italic>\n                  loss-of-function mutants. These results confirm that\n                  <jats:italic>Cfap298<\/jats:italic>\n                  functions in LR axis formation in mammals and uncover a novel region of CFAP298 protein with a conserved and essential role in cilia motility.\n                <\/jats:p>\n                <jats:sec>\n                  <jats:title>Summary Statement<\/jats:title>\n                  <jats:p>\n                    A novel\n                    <jats:italic>Cfap298<\/jats:italic>\n                    mutation disrupts left-right patterning in mice by impairing ciliary motility, revealing a conserved protein region essential for this function across species.\n                  <\/jats:p>\n                <\/jats:sec>","DOI":"10.1101\/2025.05.16.654593","type":"posted-content","created":{"date-parts":[[2025,5,17]],"date-time":"2025-05-17T23:00:11Z","timestamp":1747522811000},"source":"Crossref","is-referenced-by-count":0,"title":["A Conserved Domain of\n                  <i>Cfap298<\/i>\n                  Governs Left-Right Symmetry Breaking in Vertebrates"],"prefix":"10.64898","author":[{"given":"Marvin","family":"Cortez","sequence":"first","affiliation":[{"name":"Department of Molecular Biology, Princeton University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6283-0531","authenticated-orcid":false,"given":"Cullen B","family":"Young","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology, Princeton University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6993-5312","authenticated-orcid":false,"given":"Katherine A","family":"Little","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology, Princeton University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0173-1887","authenticated-orcid":false,"given":"Daniel T","family":"Grimes","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology, Princeton University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5464-259X","authenticated-orcid":false,"given":"Danelle","family":"Devenport","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology, Princeton University"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6620-5015","authenticated-orcid":false,"given":"Rebecca D","family":"Burdine","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology, Princeton University"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"54368","reference":[{"key":"2026041122251310000_2025.05.16.654593v1.1","doi-asserted-by":"publisher","DOI":"10.1038\/S41586-024-07487-W"},{"key":"2026041122251310000_2025.05.16.654593v1.2","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2010.07.042"},{"key":"2026041122251310000_2025.05.16.654593v1.3","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0008999"},{"key":"2026041122251310000_2025.05.16.654593v1.4","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.08.015"},{"key":"2026041122251310000_2025.05.16.654593v1.5","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2016.06.030"},{"key":"2026041122251310000_2025.05.16.654593v1.6","doi-asserted-by":"publisher","DOI":"10.1016\/j.semcdb.2020.07.005"},{"key":"2026041122251310000_2025.05.16.654593v1.7","doi-asserted-by":"publisher","DOI":"10.1016\/j.gde.2019.05.005"},{"key":"2026041122251310000_2025.05.16.654593v1.8","doi-asserted-by":"publisher","DOI":"10.1038\/ncb1784"},{"key":"2026041122251310000_2025.05.16.654593v1.9","doi-asserted-by":"crossref","unstructured":"Francis, R. and Lo, C . 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