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Here we show that mouse embryos that lack the small guanosine triphosphatase RSG1 die at embryonic day 12.5, with developmental abnormalities characteristic of decreased cilia-dependent Hedgehog signaling. Rsg1 mutant embryos have fewer primary cilia than wild-type embryos, but the cilia that form are of normal length and traffic Hedgehog pathway proteins within the cilium correctly. Rsg1 mother centrioles recruit proteins required for cilia initiation and dock onto ciliary vesicles, but axonemal microtubules fail to elongate normally. RSG1 localizes to the mother centriole in a process that depends on tau tubulin kinase 2 (TTBK2), the CPLANE complex protein Inturned (INTU), and its own GTPase activity. The data suggest a specific role for RSG1 in the final maturation of the mother centriole and ciliary vesicle that allows extension of the ciliary axoneme.<\/jats:p>","DOI":"10.1083\/jcb.201604048","type":"journal-article","created":{"date-parts":[[2017,10,16]],"date-time":"2017-10-16T10:10:19Z","timestamp":1508148619000},"page":"413-427","update-policy":"https:\/\/doi.org\/10.1083\/jcb.crossmarkpolicy","source":"Crossref","is-referenced-by-count":34,"title":["The small GTPase RSG1 controls a final step in primary cilia initiation"],"prefix":"10.1083","volume":"217","author":[{"given":"Stephanie O.","family":"Agbu","sequence":"first","affiliation":[{"name":"Developmental Biology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 1"},{"name":"Biochemistry, Cell and Molecular Biology Program, Weill Graduate School of Medical Sciences of Cornell University, New York, NY 2"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9945-3768","authenticated-orcid":true,"given":"Yinwen","family":"Liang","sequence":"additional","affiliation":[{"name":"Developmental Biology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 1"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0099-0879","authenticated-orcid":true,"given":"Aimin","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Biology, Eberly College of Science, The Pennsylvania State University, University Park, PA 3"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1657-2161","authenticated-orcid":true,"given":"Kathryn V.","family":"Anderson","sequence":"additional","affiliation":[{"name":"Developmental Biology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 1"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2017,10,16]]},"reference":[{"key":"2023072309235383300_bib1","doi-asserted-by":"publisher","first-page":"379","DOI":"10.1016\/j.tcb.2016.12.001","article-title":"From planar cell polarity to ciliogenesis and back: The curious tale of the PPE and CPLANE proteins","volume":"27","author":"Adler","year":"2017","journal-title":"Trends Cell Biol."},{"key":"2023072309235383300_bib2","doi-asserted-by":"publisher","first-page":"320","DOI":"10.1006\/dbio.2002.0811","article-title":"Mouse GLI3 regulates Fgf8 expression and apoptosis in the developing neural tube, face, and limb bud","volume":"251","author":"Aoto","year":"2002","journal-title":"Dev. 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