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We used genome editing to ablate CNTROB in hTERT-RPE1 cells and observed an increased frequency of monocentriolar and acentriolar cells. Using a novel monoclonal antibody, we found that centrobin primarily localizes to daughter centrioles but also associates with mother centrioles upon serum starvation. Strikingly, centrobin loss abrogated primary ciliation upon serum starvation. Ultrastructural analysis of centrobin nulls revealed defective axonemal extension after mother centriole docking. Ciliogenesis required a C-terminal portion of centrobin that interacts with CP110 and tubulin. We also depleted centrobin in zebrafish embryos to explore its roles in an entire organism. Centrobin-depleted embryos showed microcephaly, with curved and shorter bodies, along with marked defects in laterality control, morphological features that indicate ciliary dysfunction. Our data identify new roles for centrobin as a positive regulator of vertebrate ciliogenesis.<\/jats:p>","DOI":"10.1083\/jcb.201706095","type":"journal-article","created":{"date-parts":[[2018,2,13]],"date-time":"2018-02-13T14:06:31Z","timestamp":1518530791000},"page":"1205-1215","update-policy":"https:\/\/doi.org\/10.1083\/jcb.crossmarkpolicy","source":"Crossref","is-referenced-by-count":34,"title":["Centrobin controls primary ciliogenesis in vertebrates"],"prefix":"10.1083","volume":"217","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5548-7915","authenticated-orcid":true,"given":"Yetunde Adesanya","family":"Ogungbenro","sequence":"first","affiliation":[{"name":"Centre for Chromosome Biology, School of Natural Sciences, National University of Ireland Galway, Galway, Ireland 1"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teresa Casar","family":"Tena","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Molecular Biology, Ulm University, Ulm, Germany 3"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4047-6487","authenticated-orcid":true,"given":"David","family":"Gaboriau","sequence":"additional","affiliation":[{"name":"Centre for Chromosome Biology, School of Natural Sciences, National University of Ireland Galway, Galway, Ireland 1"},{"name":"Facility for Imaging by Light Microscopy, Imperial College London, London, England, UK 4"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pierce","family":"Lalor","sequence":"additional","affiliation":[{"name":"Anatomy, School of Medicine, National University of Ireland Galway, Galway, Ireland 2"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Dockery","sequence":"additional","affiliation":[{"name":"Anatomy, School of Medicine, National University of Ireland Galway, Galway, Ireland 2"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2714-965X","authenticated-orcid":true,"given":"Melanie","family":"Philipp","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Molecular Biology, Ulm University, Ulm, Germany 3"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2401-7029","authenticated-orcid":true,"given":"Ciaran G.","family":"Morrison","sequence":"additional","affiliation":[{"name":"Centre for Chromosome Biology, School of Natural Sciences, National University of Ireland Galway, Galway, Ireland 1"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2018,2,13]]},"reference":[{"key":"2023072315022235400_bib1","doi-asserted-by":"publisher","first-page":"246","DOI":"10.1083\/jcb.54.2.246","article-title":"The three-dimensional structure of the basal body from the rhesus monkey oviduct","volume":"54","author":"Anderson","year":"1972","journal-title":"J. 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