{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,3]],"date-time":"2026-02-03T18:23:10Z","timestamp":1770142990733,"version":"3.49.0"},"reference-count":18,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2024,5,9]],"date-time":"2024-05-09T00:00:00Z","timestamp":1715212800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["1P01HD104436"],"award-info":[{"award-number":["1P01HD104436"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["1R01HD081562"],"award-info":[{"award-number":["1R01HD081562"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["U01 HD043478"],"award-info":[{"award-number":["U01 HD043478"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Genesis"],"published-print":{"date-parts":[[2024,6]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Cilia play a key role in the regulation of signaling pathways required for embryonic development, including the proper formation of the neural tube, the precursor to the brain and spinal cord. Forward genetic screens were used to generate mouse lines that display neural tube defects (NTD) and secondary phenotypes useful in interrogating function. We describe here the <jats:italic>L3P<\/jats:italic> mutant line that displays phenotypes of disrupted Sonic hedgehog signaling and affects the initiation of cilia formation. A point mutation was mapped in the <jats:italic>L3P<\/jats:italic> line to the gene <jats:italic>Rsg1<\/jats:italic>, which encodes a GTPase\u2010like protein. The mutation lies within the GTP\u2010binding pocket and disrupts the highly conserved G1 domain. The mutant protein and other centrosomal and IFT proteins still localize appropriately to the basal body of cilia, suggesting that RSG1 GTPase activity is not required for basal body maturation but is needed for a downstream step in axonemal elongation.<\/jats:p>","DOI":"10.1002\/dvg.23602","type":"journal-article","created":{"date-parts":[[2024,5,9]],"date-time":"2024-05-09T11:35:00Z","timestamp":1715254500000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["<scp>RSG1<\/scp> is required for cilia\u2010dependent neural tube closure"],"prefix":"10.1002","volume":"62","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1611-4320","authenticated-orcid":false,"given":"David","family":"Engelhardt","sequence":"first","affiliation":[{"name":"Department of Molecular Cellular and Development Biology, University of Colorado  Boulder Colorado USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amber","family":"Marean","sequence":"additional","affiliation":[{"name":"Molecular Biology Graduate Program University of Colorado Anschutz Medical Campus  Aurora Colorado USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David","family":"McKean","sequence":"additional","affiliation":[{"name":"Cells, Stem Cells and Developmental Biology Graduate Program University of Colorado Anschutz Medical Campus  Aurora Colorado USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Juliette","family":"Petersen","sequence":"additional","affiliation":[{"name":"Molecular Biology Graduate Program University of Colorado Anschutz Medical Campus  Aurora Colorado USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9959-0594","authenticated-orcid":false,"given":"Lee","family":"Niswander","sequence":"additional","affiliation":[{"name":"Department of Molecular Cellular and Development Biology, University of Colorado  Boulder Colorado USA"},{"name":"Molecular Biology Graduate Program University of Colorado Anschutz Medical Campus  Aurora Colorado USA"},{"name":"Cells, Stem Cells and Developmental Biology Graduate Program University of Colorado Anschutz Medical Campus  Aurora Colorado USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2024,5,9]]},"reference":[{"key":"e_1_2_2_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tcb.2016.12.001"},{"key":"e_1_2_2_3_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201604048"},{"key":"e_1_2_2_4_1","doi-asserted-by":"publisher","DOI":"10.1186\/1755-8417-2-3"},{"key":"e_1_2_2_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/10018"},{"key":"e_1_2_2_6_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.277.5329.1109"},{"key":"e_1_2_2_7_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.200707181"},{"key":"e_1_2_2_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncb1966"},{"key":"e_1_2_2_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20676"},{"key":"e_1_2_2_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.1182"},{"key":"e_1_2_2_11_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature02061"},{"key":"e_1_2_2_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncb1251"},{"key":"e_1_2_2_13_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-60327-292-6_7"},{"key":"e_1_2_2_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20686"},{"key":"e_1_2_2_15_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.151.3.709"},{"key":"e_1_2_2_16_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.biochem.66.1.639"},{"key":"e_1_2_2_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.3558"},{"key":"e_1_2_2_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2005.02.027"},{"key":"e_1_2_2_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20164"}],"container-title":["genesis"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/dvg.23602","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,25]],"date-time":"2024-06-25T11:26:43Z","timestamp":1719314803000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/dvg.23602"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,5,9]]},"references-count":18,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2024,6]]}},"alternative-id":["10.1002\/dvg.23602"],"URL":"https:\/\/doi.org\/10.1002\/dvg.23602","archive":["Portico"],"relation":{},"ISSN":["1526-954X","1526-968X"],"issn-type":[{"value":"1526-954X","type":"print"},{"value":"1526-968X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,5,9]]},"assertion":[{"value":"2024-03-18","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-04-22","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-05-09","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e23602"}}