{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T03:30:54Z","timestamp":1790652654064,"version":"4.1.0"},"reference-count":60,"publisher":"Rockefeller University Press","issue":"4","license":[{"start":{"date-parts":[[2025,2,26]],"date-time":"2025-02-26T00:00:00Z","timestamp":1740528000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/rupress.org\/pages\/terms102024"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01 GM133897"],"award-info":[{"award-number":["R01 GM133897"]}],"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":["R01 GM114119"],"award-info":[{"award-number":["R01 GM114119"]}],"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":["R01 CA266199"],"award-info":[{"award-number":["R01 CA266199"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001021","name":"Damon Runyon Cancer Research Foundation","doi-asserted-by":"publisher","award":["DRG-2478-22"],"award-info":[{"award-number":["DRG-2478-22"]}],"id":[{"id":"10.13039\/100001021","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["rupress.org"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2025,4,7]]},"abstract":"<jats:p>Hydrolethalus syndrome (HLS) is a lethal, autosomal recessive ciliopathy caused by the mutation of the conserved centriole protein HYLS1. How HYLS1 controls centriole function is poorly understood. Here, we show that mice harboring the HYLS1 disease mutation die shortly after birth and exhibit developmental defects that recapitulate several manifestations of HLS. These phenotypes arise from a loss of centriole integrity that causes tissue-specific defects in cilia assembly and function. We show that HYLS1 is recruited to the centriole by CEP120 and stabilizes the localization of centriole inner scaffold proteins that ensure the integrity of the centriolar microtubule wall. The HLS disease mutation reduced the centriole localization of HYLS1 and caused degeneration of the centriole distal end. We propose that tissue-specific defects in centriole integrity caused by the HYLS1 mutation prevent ciliogenesis and contribute to HLS phenotypes.<\/jats:p>","DOI":"10.1083\/jcb.202403022","type":"journal-article","created":{"date-parts":[[2025,1,22]],"date-time":"2025-01-22T02:21:04Z","timestamp":1737512464000},"update-policy":"https:\/\/doi.org\/10.1083\/jcb.crossmarkpolicy","source":"Crossref","is-referenced-by-count":8,"title":["Centriole structural integrity defects are a crucial feature of hydrolethalus syndrome"],"prefix":"10.1083","volume":"224","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-5745-1601","authenticated-orcid":true,"given":"Ana","family":"Curinha","sequence":"first","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00za53h95","id-type":"ROR","asserted-by":"publisher"}],"name":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-9247-4349","authenticated-orcid":true,"given":"Zhaoyu","family":"Huang","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00za53h95","id-type":"ROR","asserted-by":"publisher"}],"name":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4451-3046","authenticated-orcid":true,"given":"Taylor","family":"Anglen","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00py81415","id-type":"ROR","asserted-by":"publisher"}],"name":"Duke University 2 Department of Biomedical Engineering, , Durham, NC,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0055-6573","authenticated-orcid":true,"given":"Margaret A.","family":"Strong","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00za53h95","id-type":"ROR","asserted-by":"publisher"}],"name":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5410-1718","authenticated-orcid":true,"given":"Colin R.","family":"Gliech","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00za53h95","id-type":"ROR","asserted-by":"publisher"}],"name":"Johns Hopkins University School of Medicine 3 Department of Rheumatology, , Baltimore, MD,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8406-0814","authenticated-orcid":true,"given":"Cayla E.","family":"Jewett","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00za53h95","id-type":"ROR","asserted-by":"publisher"}],"name":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9845-3715","authenticated-orcid":true,"given":"Anoek","family":"Friskes","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01n92vv28","id-type":"ROR","asserted-by":"publisher"}],"name":"Oncode Institute, The Netherlands Cancer Institute 4 Division of Cell Biology, , Amsterdam,","place":["Netherlands"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0389-7127","authenticated-orcid":true,"given":"Thao P.","family":"Phan","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/043mz5j54","id-type":"ROR","asserted-by":"publisher"}],"name":"Cardiovascular Research Institute, University of California, San Francisco 5 Department of Biochemistry and Biophysics, , San Francisco, CA,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7495-4080","authenticated-orcid":true,"given":"Zachary","family":"Nicholas","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00za53h95","id-type":"ROR","asserted-by":"publisher"}],"name":"Johns Hopkins University School of Medicine 6 Department of Neuroscience, , Baltimore, MD,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3728-6367","authenticated-orcid":true,"given":"Andrew J.","family":"Holland","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/00za53h95","id-type":"ROR","asserted-by":"publisher"}],"name":"Johns Hopkins University School of Medicine 1 Department of Molecular Biology and Genetics, , Baltimore, MD,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2025,2,26]]},"reference":[{"key":"2025022614462031700_bib1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.202305009","article-title":"CCDC15 localizes to the centriole inner scaffold and controls centriole length and integrity","volume":"222","author":"Arslanhan","year":"2023","journal-title":"J. Cell Biol."},{"key":"2025022614462031700_bib2","doi-asserted-by":"publisher","first-page":"R816","DOI":"10.1016\/j.cub.2010.08.010","article-title":"Building the centriole","volume":"20","author":"Azimzadeh","year":"2010","journal-title":"Curr. Biol."},{"key":"2025022614462031700_bib3","doi-asserted-by":"publisher","first-page":"1032","DOI":"10.1016\/j.cell.2008.05.039","article-title":"Centrosome amplification can initiate tumorigenesis in flies","volume":"133","author":"Basto","year":"2008","journal-title":"Cell"},{"key":"2025022614462031700_bib4","doi-asserted-by":"publisher","first-page":"691","DOI":"10.1146\/annurev-biochem-013118-111153","article-title":"Mechanism and regulation of centriole and cilium biogenesis","volume":"88","author":"Breslow","year":"2019","journal-title":"Annu. Rev. Biochem."},{"key":"2025022614462031700_bib5","doi-asserted-by":"publisher","first-page":"2501","DOI":"10.1242\/jcs.186338","article-title":"CEP295 interacts with microtubules and is required for centriole elongation","volume":"129","author":"Chang","year":"2016","journal-title":"J. Cell Sci."},{"key":"2025022614462031700_bib6","doi-asserted-by":"publisher","DOI":"10.1126\/sciadv.abe3401","article-title":"Ciliopathy protein HYLS1 coordinates the biogenesis and signaling of primary cilia by activating the ciliary lipid kinase PIPKI\u03b3","volume":"7","author":"Chen","year":"2021","journal-title":"Sci. Adv."},{"key":"2025022614462031700_bib7","doi-asserted-by":"publisher","first-page":"150209","DOI":"10.1098\/rsob.150209","article-title":"Over-expression of Plk4 induces centrosome amplification, loss of primary cilia and associated tissue hyperplasia in the mouse","volume":"5","author":"Coelho","year":"2015","journal-title":"Open Biol."},{"key":"2025022614462031700_bib8","doi-asserted-by":"publisher","first-page":"1360","DOI":"10.1016\/j.cub.2013.06.002","article-title":"CEP120 and SPICE1 cooperate with CPAP in centriole elongation","volume":"23","author":"Comartin","year":"2013","journal-title":"Curr. Biol."},{"key":"2025022614462031700_bib9","doi-asserted-by":"publisher","first-page":"2245","DOI":"10.1016\/j.cub.2013.09.037","article-title":"Regulation of autophosphorylation controls PLK4 self-destruction and centriole number","volume":"23","author":"Cunha-Ferreira","year":"2013","journal-title":"Curr. Biol."},{"key":"2025022614462031700_bib10","doi-asserted-by":"publisher","first-page":"2046","DOI":"10.1101\/gad.1810409","article-title":"The hydrolethalus syndrome protein HYLS-1 links core centriole structure to cilia formation","volume":"23","author":"Dammermann","year":"2009","journal-title":"Genes Dev."},{"key":"2025022614462031700_bib11","doi-asserted-by":"publisher","first-page":"19","DOI":"10.1016\/bs.mcb.2015.01.004","article-title":"Ependymal cell differentiation, from monociliated to multiciliated cells","volume":"127","author":"Delgehyr","year":"2015","journal-title":"Methods Cell Biol."},{"key":"2025022614462031700_bib12","doi-asserted-by":"publisher","DOI":"10.15252\/embj.2020105106","article-title":"ANKRD26 recruits PIDD1 to centriolar distal appendages to activate the PIDDosome following centrosome amplification","volume":"40","author":"Evans","year":"2021","journal-title":"EMBO J."},{"key":"2025022614462031700_bib60","doi-asserted-by":"publisher","first-page":"71","DOI":"10.1038\/s41592-018-0238-1","article-title":"Imaging cellular ultrastructures using expansion microscopy (U-ExM)","volume":"16","author":"Gambarotto","year":"2019","journal-title":"Nat. Methods"},{"key":"2025022614462031700_bib13","doi-asserted-by":"publisher","first-page":"57","DOI":"10.1016\/bs.mcb.2020.05.006","article-title":"Ultrastructure expansion microscopy (U-ExM)","volume":"161","author":"Gambarotto","year":"2021","journal-title":"Methods Cell Biol."},{"key":"2025022614462031700_bib14","doi-asserted-by":"publisher","first-page":"100407","DOI":"10.1016\/j.xpro.2021.100407","article-title":"CRISPR\/Cas9 ribonucleoprotein-mediated knockin generation in hTERT-RPE1 cells","volume":"2","author":"Ghetti","year":"2021","journal-title":"STAR Protoc."},{"key":"2025022614462031700_bib15","doi-asserted-by":"publisher","first-page":"666","DOI":"10.1038\/s44318-024-00031-6","article-title":"Weakened APC\/C activity at mitotic exit drives cancer vulnerability to KIF18A inhibition","volume":"43","author":"Gliech","year":"2024","journal-title":"EMBO J."},{"key":"2025022614462031700_bib16","doi-asserted-by":"publisher","first-page":"425","DOI":"10.1038\/nrm3373","article-title":"Towards a molecular architecture of centriole assembly","volume":"13","author":"G\u00f6nczy","year":"2012","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2025022614462031700_bib17","doi-asserted-by":"publisher","first-page":"2163","DOI":"10.1242\/jcs.068502","article-title":"Plk4 trans-autophosphorylation regulates centriole number by controlling betaTrCP-mediated degradation","volume":"123","author":"Guderian","year":"2010","journal-title":"J. Cell Sci."},{"key":"2025022614462031700_bib18","doi-asserted-by":"publisher","first-page":"1565","DOI":"10.1038\/emboj.2010.45","article-title":"Procentriole assembly revealed by cryo-electron tomography","volume":"29","author":"Guichard","year":"2010","journal-title":"EMBO J."},{"key":"2025022614462031700_bib19","doi-asserted-by":"publisher","first-page":"1620","DOI":"10.1016\/j.cub.2013.06.061","article-title":"Native architecture of the centriole proximal region reveals features underlying its 9-fold radial symmetry","volume":"23","author":"Guichard","year":"2013","journal-title":"Curr. Biol."},{"key":"2025022614462031700_bib20","doi-asserted-by":"publisher","first-page":"2731","DOI":"10.4161\/cc.9.14.12184","article-title":"Centriole duplication: A lesson in self-control","volume":"9","author":"Holland","year":"2010","journal-title":"Cell Cycle"},{"key":"2025022614462031700_bib21","doi-asserted-by":"publisher","first-page":"2","DOI":"10.1186\/1755-8417-2-2","article-title":"Unraveling the disease pathogenesis behind lethal hydrolethalus syndrome revealed multiple changes in molecular and cellular level","volume":"2","author":"Honkala","year":"2009","journal-title":"PathoGenetics"},{"key":"2025022614462031700_bib22","doi-asserted-by":"publisher","first-page":"301","DOI":"10.3389\/fcell.2020.00301","article-title":"Functional analysis of hydrolethalus syndrome protein HYLS1 in ciliogenesis and spermatogenesis in Drosophila","volume":"8","author":"Hou","year":"2020","journal-title":"Front. Cell Dev. Biol."},{"key":"2025022614462031700_bib23","doi-asserted-by":"publisher","first-page":"1012","DOI":"10.1016\/j.cub.2009.05.018","article-title":"Overly long centrioles and defective cell division upon excess of the SAS-4-related protein CPAP","volume":"19","author":"Kohlmaier","year":"2009","journal-title":"Curr. Biol."},{"key":"2025022614462031700_bib24","doi-asserted-by":"publisher","first-page":"58","DOI":"10.1016\/j.sbi.2020.09.015","article-title":"Overview of the centriole architecture","volume":"66","author":"LeGuennec","year":"2021","journal-title":"Curr. Opin. Struct. Biol."},{"key":"2025022614462031700_bib25","doi-asserted-by":"publisher","first-page":"313","DOI":"10.1016\/j.devcel.2016.12.022","article-title":"Centrosome amplification is sufficient to promote spontaneous tumorigenesis in mammals","volume":"40","author":"Levine","year":"2017","journal-title":"Dev. Cell"},{"key":"2025022614462031700_bib26","doi-asserted-by":"publisher","first-page":"620","DOI":"10.1101\/gad.314351.118","article-title":"The impact of mitotic errors on cell proliferation and tumorigenesis","volume":"32","author":"Levine","year":"2018","journal-title":"Genes Dev."},{"key":"2025022614462031700_bib27","doi-asserted-by":"publisher","first-page":"552","DOI":"10.1038\/emboj.2011.460","article-title":"Three-dimensional structure of basal body triplet revealed by electron cryo-tomography","volume":"31","author":"Li","year":"2012","journal-title":"EMBO J."},{"key":"2025022614462031700_bib28","doi-asserted-by":"publisher","first-page":"211","DOI":"10.1083\/jcb.201212060","article-title":"CEP120 interacts with CPAP and positively regulates centriole elongation","volume":"202","author":"Lin","year":"2013","journal-title":"J. Cell Biol."},{"key":"2025022614462031700_bib29","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.80643","article-title":"PLK4 drives centriole amplification and apical surface area expansion in multiciliated cells","volume":"11","author":"LoMastro","year":"2022","journal-title":"Elife"},{"key":"2025022614462031700_bib30","doi-asserted-by":"publisher","first-page":"1475","DOI":"10.1093\/hmg\/ddi157","article-title":"Hydrolethalus syndrome is caused by a missense mutation in a novel gene HYLS1","volume":"14","author":"Mee","year":"2005","journal-title":"Hum. Mol. Genet."},{"key":"2025022614462031700_bib59","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.46054","article-title":"PLK4 promotes centriole duplication by phosphorylating STIL to link the procentriole cartwheel to the microtubule wall","volume":"8","author":"Moyer","year":"2019","journal-title":"Elife"},{"key":"2025022614462031700_bib31","doi-asserted-by":"publisher","first-page":"297","DOI":"10.1038\/nrm.2017.127","article-title":"Once and only once: Mechanisms of centriole duplication and their deregulation in disease","volume":"19","author":"Nigg","year":"2018","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2025022614462031700_bib32","doi-asserted-by":"publisher","first-page":"663","DOI":"10.1016\/j.cell.2009.10.036","article-title":"Centrioles, centrosomes, and cilia in health and disease","volume":"139","author":"Nigg","year":"2009","journal-title":"Cell"},{"key":"2025022614462031700_bib33","doi-asserted-by":"publisher","first-page":"1154","DOI":"10.1038\/ncb2345","article-title":"The centrosome cycle: Centriole biogenesis, duplication and inherent asymmetries","volume":"13","author":"Nigg","year":"2011","journal-title":"Nat. Cell Biol."},{"key":"2025022614462031700_bib34","doi-asserted-by":"publisher","first-page":"619","DOI":"10.1038\/nature05318","article-title":"Centriole assembly in Caenorhabditis elegans","volume":"444","author":"Pelletier","year":"2006","journal-title":"Nature"},{"key":"2025022614462031700_bib35","doi-asserted-by":"publisher","first-page":"718","DOI":"10.1101\/gad.349604.122","article-title":"Upstream open reading frames control PLK4 translation and centriole duplication in primordial germ cells","volume":"36","author":"Phan","year":"2022","journal-title":"Genes Dev."},{"key":"2025022614462031700_bib36","doi-asserted-by":"publisher","first-page":"533","DOI":"10.1038\/nrm.2017.60","article-title":"Genes and molecular pathways underpinning ciliopathies","volume":"18","author":"Reiter","year":"2017","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2025022614462031700_bib58","doi-asserted-by":"publisher","first-page":"113","DOI":"10.1007\/978-1-62703-989-5_9","article-title":"Whole-mount skeletal staining","volume":"1130","author":"Rigueur","year":"2014","journal-title":"Methods Mol. Biol."},{"key":"2025022614462031700_bib37","doi-asserted-by":"publisher","first-page":"282","DOI":"10.1074\/mcp.M700342-MCP200","article-title":"A versatile nanotrap for biochemical and functional studies with fluorescent fusion proteins","volume":"7","author":"Rothbauer","year":"2008","journal-title":"Mol. Cell. Proteomics"},{"key":"2025022614462031700_bib38","doi-asserted-by":"publisher","first-page":"676","DOI":"10.1038\/nmeth.2019","article-title":"Fiji: An open-source platform for biological-image analysis","volume":"9","author":"Schindelin","year":"2012","journal-title":"Nat. Methods"},{"key":"2025022614462031700_bib39","doi-asserted-by":"publisher","first-page":"1005","DOI":"10.1016\/j.cub.2009.05.016","article-title":"Control of centriole length by CPAP and CP110","volume":"19","author":"Schmidt","year":"2009","journal-title":"Curr. Biol."},{"key":"2025022614462031700_bib40","doi-asserted-by":"publisher","first-page":"6042","DOI":"10.1038\/s41467-021-26252-5","article-title":"Sub-centrosomal mapping identifies augmin-\u03b3TuRC as part of a centriole-stabilizing scaffold","volume":"12","author":"Schweizer","year":"2021","journal-title":"Nat. Commun."},{"key":"2025022614462031700_bib41","doi-asserted-by":"publisher","first-page":"100","DOI":"10.1038\/ncb3270","article-title":"Transient PLK4 overexpression accelerates tumorigenesis in p53-deficient epidermis","volume":"18","author":"Ser\u00e7in","year":"2016","journal-title":"Nat. Cell Biol."},{"key":"2025022614462031700_bib42","doi-asserted-by":"publisher","first-page":"1659","DOI":"10.1083\/jcb.201610070","article-title":"Centrioles initiate cilia assembly but are dispensable for maturation and maintenance in C. elegans","volume":"216","author":"Serwas","year":"2017","journal-title":"J. Cell Biol."},{"key":"2025022614462031700_bib43","doi-asserted-by":"publisher","first-page":"362","DOI":"10.1016\/j.devcel.2016.04.024","article-title":"Centriolar CPAP\/SAS-4 imparts slow processive microtubule growth","volume":"37","author":"Sharma","year":"2016","journal-title":"Dev. Cell"},{"key":"2025022614462031700_bib44","doi-asserted-by":"publisher","first-page":"62","DOI":"10.1016\/j.jsb.2018.01.009","article-title":"Cep120 promotes microtubule formation through a unique tubulin binding C2 domain","volume":"203","author":"Sharma","year":"2018","journal-title":"J. Struct. Biol."},{"key":"2025022614462031700_bib45","doi-asserted-by":"publisher","first-page":"89","DOI":"10.1016\/j.sbi.2020.10.011","article-title":"Centriole length control","volume":"66","author":"Sharma","year":"2021","journal-title":"Curr. Opin. Struct. Biol."},{"key":"2025022614462031700_bib46","doi-asserted-by":"publisher","first-page":"843","DOI":"10.1101\/gad.349727.122","article-title":"Centriole signaling restricts hepatocyte ploidy to maintain liver integrity","volume":"36","author":"Sladky","year":"2022","journal-title":"Genes Dev."},{"key":"2025022614462031700_bib47","doi-asserted-by":"publisher","DOI":"10.15252\/embr.202050893","article-title":"PIDDosome-induced p53-dependent ploidy restriction facilitates hepatocarcinogenesis","volume":"21","author":"Sladky","year":"2020","journal-title":"EMBO Rep."},{"key":"2025022614462031700_bib48","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.57205","article-title":"WDR90 is a centriolar microtubule wall protein important for centriole architecture integrity","volume":"9","author":"Steib","year":"2020","journal-title":"Elife"},{"key":"2025022614462031700_bib49","doi-asserted-by":"publisher","first-page":"2216","DOI":"10.1038\/s41467-024-46454-x","article-title":"Molecular basis promoting centriole triplet microtubule assembly","volume":"15","author":"Takeda","year":"2024","journal-title":"Nat. Commun."},{"key":"2025022614462031700_bib50","doi-asserted-by":"publisher","first-page":"825","DOI":"10.1038\/ncb1889","article-title":"CPAP is a cell-cycle regulated protein that controls centriole length","volume":"11","author":"Tang","year":"2009","journal-title":"Nat. Cell Biol."},{"key":"2025022614462031700_bib51","doi-asserted-by":"publisher","first-page":"163","DOI":"10.1101\/gad.207043.112","article-title":"Centriole distal appendages promote membrane docking, leading to cilia initiation","volume":"27","author":"Tanos","year":"2013","journal-title":"Genes Dev."},{"key":"2025022614462031700_bib52","doi-asserted-by":"publisher","first-page":"6037","DOI":"10.1038\/s41598-019-42577-0","article-title":"CEP120 interacts with C2CD3 and Talpid3 and is required for centriole appendage assembly and ciliogenesis","volume":"9","author":"Tsai","year":"2019","journal-title":"Sci. Rep."},{"key":"2025022614462031700_bib53","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.202108018","article-title":"CPAP insufficiency leads to incomplete centrioles that duplicate but fragment","volume":"221","author":"V\u00e1squez-Limeta","year":"2022","journal-title":"J. Cell Biol."},{"key":"2025022614462031700_bib54","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.29061","article-title":"Centriole triplet microtubules are required for stable centriole formation and inheritance in human cells","volume":"6","author":"Wang","year":"2017","journal-title":"Elife"},{"key":"2025022614462031700_bib55","doi-asserted-by":"publisher","first-page":"3938","DOI":"10.1038\/s41467-018-06286-y","article-title":"A distal centriolar protein network controls organelle maturation and asymmetry","volume":"9","author":"Wang","year":"2018","journal-title":"Nat. Commun."},{"key":"2025022614462031700_bib56","doi-asserted-by":"publisher","first-page":"12437","DOI":"10.1038\/ncomms12437","article-title":"The hydrolethalus syndrome protein HYLS-1 regulates formation of the ciliary gate","volume":"7","author":"Wei","year":"2016","journal-title":"Nat. Commun."},{"key":"2025022614462031700_bib57","doi-asserted-by":"publisher","first-page":"11874","DOI":"10.1038\/ncomms11874","article-title":"Molecular basis for CPAP-tubulin interaction in controlling centriolar and ciliary length","volume":"7","author":"Zheng","year":"2016","journal-title":"Nat. Commun."}],"container-title":["Journal of Cell Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/rupress.org\/jcb\/article-pdf\/doi\/10.1083\/jcb.202403022\/1939488\/jcb_202403022.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/rupress.org\/jcb\/article-pdf\/doi\/10.1083\/jcb.202403022\/1939488\/jcb_202403022.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,2,26]],"date-time":"2025-02-26T09:49:24Z","timestamp":1740563364000},"score":1,"resource":{"primary":{"URL":"https:\/\/rupress.org\/jcb\/article\/224\/4\/e202403022\/277290\/Centriole-structural-integrity-defects-are-a"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,2,26]]},"references-count":60,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2025,4,7]]}},"URL":"https:\/\/doi.org\/10.1083\/jcb.202403022","relation":{"has-preprint":[{"id-type":"doi","id":"10.1101\/2024.03.06.583733","asserted-by":"object"}]},"ISSN":["0021-9525","1540-8140"],"issn-type":[{"value":"0021-9525","type":"print"},{"value":"1540-8140","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,2,26]]},"article-number":"e202403022"}}