{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,22]],"date-time":"2026-09-22T01:56:32Z","timestamp":1790042192499,"version":"4.0.1"},"reference-count":27,"publisher":"The Company of Biologists","issue":"6","license":[{"start":{"date-parts":[[2020,9,2]],"date-time":"2020-09-02T00:00:00Z","timestamp":1599004800000},"content-version":"vor","delay-in-days":3053,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-sa\/3.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2012,6,15]]},"abstract":"<jats:title>Summary<\/jats:title>\n               <jats:p>The primary cilium is a microtubule-based structure protruded from the basal body analogous to the centriole. CPAP (centrosomal P4.1-associated protein) has previously been reported to be a cell cycle-regulated protein that controls centriole length. Mutations in CPAP cause primary microcephaly (MCPH) in humans. Here, using a cell-based system that we established to monitor cilia formation in neuronal CAD (Cath.a-differentiated) cells and hippocampal neurons, we found that CPAP is required for cilia biogenesis. Overexpression of wild-type CPAP promoted cilia formation and induced longer cilia. In contrast, an exogenously expressed CPAP-377EE mutant that lacks tubulin-dimer binding significantly inhibited cilia formation and caused cilia shortening. Furthermore, depletion of CPAP inhibited ciliogenesis and such effect was effectively rescued by expression of wild-type CPAP, but not by the CPAP-377EE mutant. Taken together, our results suggest that CPAP is a positive regulator of ciliogenesis whose intrinsic tubulin-dimer binding activity is required for cilia formation in neuronal cells.<\/jats:p>","DOI":"10.1242\/bio.20121388","type":"journal-article","created":{"date-parts":[[2012,4,27]],"date-time":"2012-04-27T04:50:50Z","timestamp":1335502250000},"page":"559-565","source":"Crossref","is-referenced-by-count":31,"title":["CPAP is required for cilia formation in neuronal cells"],"prefix":"10.1242","volume":"1","author":[{"given":"Kuo-Sheng","family":"Wu","sequence":"first","affiliation":[{"name":"Program in Molecular Medicine, National Yang-Ming University and Academia Sinica, Nankang, Taipei 115, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tang K.","family":"Tang","sequence":"additional","affiliation":[{"name":"Program in Molecular Medicine, National Yang-Ming University and Academia Sinica, Nankang, Taipei 115, Taiwan"},{"name":"Institute of Biomedical Sciences, Academia Sinica, Nankang, Taipei 115, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2012,4,24]]},"reference":[{"key":"2021080923294685000_b1","doi-asserted-by":"publisher","first-page":"848","DOI":"10.1002\/cne.22793","article-title":"Development and distribution of neuronal cilia in mouse neocortex.","volume":"520","author":"Arellano","year":"2012","journal-title":"J. Comp. Neurol."},{"key":"2021080923294685000_b2","doi-asserted-by":"publisher","first-page":"1375","DOI":"10.1016\/j.cell.2006.05.025","article-title":"Flies without centrioles.","volume":"125","author":"Basto","year":"2006","journal-title":"Cell"},{"key":"2021080923294685000_b3","doi-asserted-by":"publisher","first-page":"1095","DOI":"10.1002\/jnr.21209","article-title":"Hippocampal neurons possess primary cilia in culture.","volume":"85","author":"Berbari","year":"2007","journal-title":"J. Neurosci. Res."},{"key":"2021080923294685000_b4","doi-asserted-by":"publisher","first-page":"307","DOI":"10.1016\/j.tig.2011.05.004","article-title":"Centrosomes and cilia in human disease.","volume":"27","author":"Bettencourt-Dias","year":"2011","journal-title":"Trends Genet."},{"key":"2021080923294685000_b5","doi-asserted-by":"publisher","first-page":"353","DOI":"10.1038\/ng1539","article-title":"A centrosomal mechanism involving CDK5RAP2 and CENPJ controls brain size.","volume":"37","author":"Bond","year":"2005","journal-title":"Nat. Genet."},{"key":"2021080923294685000_b6","doi-asserted-by":"publisher","first-page":"731","DOI":"10.1083\/jcb.201007107","article-title":"Cep152 acts as a scaffold for recruitment of Plk4 and CPAP to the centrosome.","volume":"191","author":"Cizmecioglu","year":"2010","journal-title":"J. Cell Biol."},{"key":"2021080923294685000_b7","doi-asserted-by":"publisher","first-page":"358","DOI":"10.1016\/j.molmed.2006.06.006","article-title":"What primary microcephaly can tell us about brain growth.","volume":"12","author":"Cox","year":"2006","journal-title":"Trends Mol. 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Cell Biol."},{"key":"2021080923294685000_b11","doi-asserted-by":"publisher","first-page":"2591","DOI":"10.1016\/j.yexcr.2008.05.012","article-title":"Functional characterization of the microtubule-binding and -destabilizing domains of CPAP and d-SAS-4.","volume":"314","author":"Hsu","year":"2008","journal-title":"Exp. Cell Res."},{"key":"2021080923294685000_b12","doi-asserted-by":"publisher","first-page":"7813","DOI":"10.1128\/MCB.20.20.7813--7825.2000","article-title":"Protein 4.1 R-135 interacts with a novel centrosomal protein (CPAP) which is associated with the gamma-tubulin complex.","volume":"20","author":"Hung","year":"2000","journal-title":"Mol. Cell. Biol."},{"key":"2021080923294685000_b13","doi-asserted-by":"publisher","first-page":"517","DOI":"10.1038\/ncb1251","article-title":"Odf2-deficient mother centrioles lack distal\/subdistal appendages and the ability to generate primary cilia.","volume":"7","author":"Ishikawa","year":"2005","journal-title":"Nat. Cell Biol."},{"key":"2021080923294685000_b14","doi-asserted-by":"publisher","first-page":"2406","DOI":"10.1038\/nprot.2006.356","article-title":"Culturing hippocampal neurons.","volume":"1","author":"Kaech","year":"2006","journal-title":"Nat. Protoc."},{"key":"2021080923294685000_b15","doi-asserted-by":"publisher","first-page":"190","DOI":"10.1016\/j.devcel.2007.07.002","article-title":"Plk4-induced centriole biogenesis in human cells.","volume":"13","author":"Kleylein-Sohn","year":"2007","journal-title":"Dev. 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Cell Biol."},{"key":"2021080923294685000_b27","doi-asserted-by":"publisher","first-page":"4790","DOI":"10.1038\/emboj.2011.378","article-title":"The human microcephaly protein STIL interacts with CPAP and is required for procentriole formation.","volume":"30","author":"Tang","year":"2011","journal-title":"EMBO J."}],"container-title":["Biology Open"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/journals.logists.com\/bio\/bio\/article-pdf\/1\/6\/559\/2097130\/bio-01-06-559.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/journals.logists.com\/bio\/bio\/article-pdf\/1\/6\/559\/2097130\/bio-01-06-559.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,8,10]],"date-time":"2021-08-10T00:08:40Z","timestamp":1628554120000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/bio\/article\/1\/6\/559\/885\/CPAP-is-required-for-cilia-formation-in-neuronal"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2012,4,24]]},"references-count":27,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2012,6,15]]}},"URL":"https:\/\/doi.org\/10.1242\/bio.20121388","relation":{},"ISSN":["2046-6390"],"issn-type":[{"value":"2046-6390","type":"electronic"}],"subject":[],"published":{"date-parts":[[2012,4,24]]}}}