{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T06:39:36Z","timestamp":1791614376232,"version":"4.3.4"},"reference-count":55,"publisher":"The Company of Biologists","issue":"6","license":[{"start":{"date-parts":[[2024,3,28]],"date-time":"2024-03-28T00:00:00Z","timestamp":1711584000000},"content-version":"vor","delay-in-days":13,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100000024","name":"Canadian Institutes of Health Research","doi-asserted-by":"crossref","award":["CIHR230929"],"award-info":[{"award-number":["CIHR230929"]}],"id":[{"id":"10.13039\/501100000024","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100030922","name":"Institut de recherche, Centre universitaire de sant\u00e9 McGill","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100030922","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Fonds de recherche du Qu\u00e9bec - Sant\u00e9"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,3,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>The primary cilium decorates most eukaryotic cells and regulates tissue morphogenesis and maintenance. Structural or functional defects of primary cilium result in ciliopathies, congenital human disorders affecting multiple organs. Pathogenic variants in the ciliogenesis and planar cell polarity effectors (CPLANE) genes FUZZY, INTU and WDPCP disturb ciliogenesis, causing severe ciliopathies in humans and mice. Here, we show that the loss of Fuzzy in mice results in defects of primary cilia, accompanied by increased RhoA activity and excessive actin polymerization at the basal body. We discovered that, mechanistically, Fuzzy interacts with and recruits the negative actin regulator ARHGAP35 (also known as p190A RhoGAP) to the basal body. We identified genetic interactions between the two genes and found that a mutant ArhGAP35 allele increases the severity of phenotypic defects observed in Fuzzy\u2212\/\u2212 mice. Based on our findings, we propose that Fuzzy regulates ciliogenesis by recruiting ARHGAP35 to the basal body, where the latter likely restricts actin polymerization and modifies the actin network. Our study identifies a mechanism whereby CPLANE proteins control both actin polymerization and primary cilium formation.<\/jats:p>","DOI":"10.1242\/dev.202322","type":"journal-article","created":{"date-parts":[[2024,3,28]],"date-time":"2024-03-28T11:26:22Z","timestamp":1711625182000},"source":"Crossref","is-referenced-by-count":7,"title":["The CPLANE protein Fuzzy regulates ciliogenesis by suppressing actin polymerization at the base of the primary cilium via p190A RhoGAP"],"prefix":"10.1242","volume":"151","author":[{"given":"Rhythm","family":"Sharma","sequence":"first","affiliation":[{"name":"McGill University 1 Department of Medicine and Department of Physiology , , Montreal H4A 3J1, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rita","family":"Kalot","sequence":"additional","affiliation":[{"name":"McGill University 1 Department of Medicine and Department of Physiology , , Montreal H4A 3J1, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yossef","family":"Levin","sequence":"additional","affiliation":[{"name":"McGill University 1 Department of Medicine and Department of Physiology , , Montreal H4A 3J1, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sima","family":"Babayeva","sequence":"additional","affiliation":[{"name":"Metabolic Disorders and Complications Program, Research Institute of the McGill University Health Centre 2 , , Montreal H4A 3J1, QC , Canada"},{"name":"McGill University 2 , , Montreal H4A 3J1, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nadezda","family":"Kachurina","sequence":"additional","affiliation":[{"name":"Metabolic Disorders and Complications Program, Research Institute of the McGill University Health Centre 2 , , Montreal H4A 3J1, QC , Canada"},{"name":"McGill University 2 , , Montreal H4A 3J1, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chen-Feng","family":"Chung","sequence":"additional","affiliation":[{"name":"McGill University 1 Department of Medicine and Department of Physiology , , Montreal H4A 3J1, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2483-2165","authenticated-orcid":false,"given":"Karen J.","family":"Liu","sequence":"additional","affiliation":[{"name":"Centre for Craniofacial and Regenerative Biology, King's College London 3 , London SE1 9RT , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maxime","family":"Bouchard","sequence":"additional","affiliation":[{"name":"Rosalind and Morris Goodman Cancer Institute 4 , Department of Medicine of the McGill University, , Montreal H3A 1A3, QC , Canada"},{"name":"McGill University 4 , Department of Medicine of the McGill University, , Montreal H3A 1A3, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9836-8246","authenticated-orcid":false,"given":"Elena","family":"Torban","sequence":"additional","affiliation":[{"name":"McGill University 1 Department of Medicine and Department of Physiology , , Montreal H4A 3J1, QC , Canada"},{"name":"Metabolic Disorders and Complications Program, Research Institute of the McGill University Health Centre 2 , , Montreal H4A 3J1, QC , Canada"},{"name":"McGill University 2 , , Montreal H4A 3J1, QC , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,3,28]]},"reference":[{"key":"2024032811255864200_DEV202322C1","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":"2024032811255864200_DEV202322C2","doi-asserted-by":"publisher","first-page":"829","DOI":"10.1093\/genetics\/137.3.829","article-title":"The Drosophila tissue polarity gene inturned functions prior to wing hair morphogenesis in the regulation of hair polarity and number","volume":"137","author":"Adler","year":"1994","journal-title":"Genetics"},{"key":"2024032811255864200_DEV202322C3","doi-asserted-by":"publisher","first-page":"413","DOI":"10.1083\/jcb.201604048","article-title":"The small GTPase RSG1 controls a final step in primary cilia initiation","volume":"217","author":"Agbu","year":"2018","journal-title":"J. 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