{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T03:40:33Z","timestamp":1788234033268,"version":"build-2803163510"},"reference-count":78,"publisher":"Life Science Alliance, LLC","issue":"6","license":[{"start":{"date-parts":[[2024,4,3]],"date-time":"2024-04-03T00:00:00Z","timestamp":1712102400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["APP1120356"],"award-info":[{"award-number":["APP1120356"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["life-science-alliance.org"],"crossmark-restriction":true},"short-container-title":["Life Sci. Alliance"],"accepted":{"date-parts":[[2024,3,26]]},"published-print":{"date-parts":[[2024,6]]},"abstract":"<jats:p>The transition zone is a specialised gate at the base of cilia\/flagella, which separates the ciliary compartment from the cytoplasm and strictly regulates protein entry. We identified a potential new regulator of the male germ cell transition zone, CEP76. We demonstrated that CEP76 was involved in the selective entry and incorporation of key proteins required for sperm function and fertility into the ciliary compartment and ultimately the sperm tail. In the mutant, sperm tails were shorter and immotile as a consequence of deficits in essential sperm motility proteins including DNAH2 and AKAP4, which accumulated at the sperm neck in the mutant. Severe annulus, fibrous sheath, and outer dense fibre abnormalities were also detected in sperm lacking CEP76. Finally, we identified that CEP76 dictates annulus positioning and structure. This study suggests CEP76 as a male germ cell transition zone protein and adds further evidence to the hypothesis that the spermatid transition zone and annulus are part of the same functional structure.<\/jats:p>","DOI":"10.26508\/lsa.202302452","type":"journal-article","created":{"date-parts":[[2024,4,3]],"date-time":"2024-04-03T14:30:21Z","timestamp":1712154621000},"page":"e202302452","update-policy":"https:\/\/doi.org\/10.26508\/lsa.crossmarkpolicy","source":"Crossref","is-referenced-by-count":11,"title":["Genetic mutation of\n                    <i>Cep76<\/i>\n                    results in male infertility due to abnormal sperm tail composition"],"prefix":"10.26508","volume":"7","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1078-756X","authenticated-orcid":true,"given":"Brendan J","family":"Houston","sequence":"first","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01ej9dk98","id-type":"ROR","asserted-by":"publisher"}],"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne","place":[", Parkville, Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D Jo","family":"Merriner","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01ej9dk98","id-type":"ROR","asserted-by":"publisher"}],"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne","place":[", Parkville, Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G Gemma","family":"Stathatos","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01ej9dk98","id-type":"ROR","asserted-by":"publisher"}],"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne","place":[", Parkville, Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph H","family":"Nguyen","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01ej9dk98","id-type":"ROR","asserted-by":"publisher"}],"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne","place":[", Parkville, Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne E","family":"O\u2019Connor","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01ej9dk98","id-type":"ROR","asserted-by":"publisher"}],"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne","place":[", Parkville, Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandra M","family":"Lopes","sequence":"additional","affiliation":[{"name":"Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal"},{"name":"Institute of Molecular Pathology & Immunology, University of Porto, Porto, Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donald F","family":"Conrad","sequence":"additional","affiliation":[{"name":"Oregon National Primate Research Center, Oregon Health and Science University, Beaverton, OR, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark","family":"Baker","sequence":"additional","affiliation":[{"name":"School of Environmental and Life Sciences, The University of Newcastle, Callaghan, Australia"},{"name":"School of Biomedical Sciences and Pharmacy, College of Health, Medicine and Wellbeing, The University of Newcastle, Callaghan, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica EM","family":"Dunleavy","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01ej9dk98","id-type":"ROR","asserted-by":"publisher"}],"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne","place":[", Parkville, Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7298-4940","authenticated-orcid":true,"given":"Moira K","family":"O\u2019Bryan","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01ej9dk98","id-type":"ROR","asserted-by":"publisher"}],"name":"School of BioSciences and Bio21 Molecular Sciences and Biotechnology Institute, The University of Melbourne","place":[", Parkville, Australia"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"12380","published-online":{"date-parts":[[2024,4,3]]},"reference":[{"key":"2024040311301473000_7.6.e202302452.1","doi-asserted-by":"publisher","DOI":"10.1093\/molehr\/gar034"},{"key":"2024040311301473000_7.6.e202302452.2","doi-asserted-by":"publisher","DOI":"10.1038\/ng.891"},{"key":"2024040311301473000_7.6.e202302452.3","doi-asserted-by":"publisher","DOI":"10.1126\/science.1191054"},{"key":"2024040311301473000_7.6.e202302452.4","doi-asserted-by":"publisher","DOI":"10.1038\/ncb2410"},{"key":"2024040311301473000_7.6.e202302452.5","doi-asserted-by":"publisher","DOI":"10.14348\/molcells.2017.0054"},{"key":"2024040311301473000_7.6.e202302452.6","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.cellbio.042308.113337"},{"key":"2024040311301473000_7.6.e202302452.7","doi-asserted-by":"publisher","DOI":"10.1016\/s0022-5320(84)80021-0"},{"key":"2024040311301473000_7.6.e202302452.8","doi-asserted-by":"publisher","DOI":"10.1038\/embor.2012.73"},{"key":"2024040311301473000_7.6.e202302452.9","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1604258113"},{"key":"2024040311301473000_7.6.e202302452.10","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2008.10.002"},{"key":"2024040311301473000_7.6.e202302452.11","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.1002416"},{"key":"2024040311301473000_7.6.e202302452.12","doi-asserted-by":"publisher","DOI":"10.1038\/ncb3273"},{"key":"2024040311301473000_7.6.e202302452.13","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2015.11.001"},{"key":"2024040311301473000_7.6.e202302452.14","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-cellbio-120219-034238"},{"key":"2024040311301473000_7.6.e202302452.15","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms3750"},{"key":"2024040311301473000_7.6.e202302452.16","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-022-31751-0"},{"key":"2024040311301473000_7.6.e202302452.17","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2014.09.047"},{"key":"2024040311301473000_7.6.e202302452.18","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201603086"},{"key":"2024040311301473000_7.6.e202302452.19","doi-asserted-by":"publisher","DOI":"10.1038\/nrm.2017.60"},{"key":"2024040311301473000_7.6.e202302452.20","doi-asserted-by":"publisher","DOI":"10.1042\/bse0350103"},{"key":"2024040311301473000_7.6.e202302452.21","doi-asserted-by":"publisher","DOI":"10.15252\/embj.2020107410"},{"key":"2024040311301473000_7.6.e202302452.22","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.2002.0728"},{"key":"2024040311301473000_7.6.e202302452.23","doi-asserted-by":"publisher","DOI":"10.1002\/jemt.10320"},{"key":"2024040311301473000_7.6.e202302452.24","doi-asserted-by":"publisher","DOI":"10.1095\/biolreprod.102.013466"},{"key":"2024040311301473000_7.6.e202302452.25","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.2106673118"},{"key":"2024040311301473000_7.6.e202302452.26","doi-asserted-by":"publisher","DOI":"10.1093\/humupd\/dmaa004"},{"key":"2024040311301473000_7.6.e202302452.27","doi-asserted-by":"publisher","DOI":"10.1186\/s12958-019-0510-y"},{"key":"2024040311301473000_7.6.e202302452.28","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-7580.2005.00437.x"},{"key":"2024040311301473000_7.6.e202302452.29","doi-asserted-by":"publisher","DOI":"10.1016\/0012-1606(75)90411-X"},{"key":"2024040311301473000_7.6.e202302452.30","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2005.01.021"},{"key":"2024040311301473000_7.6.e202302452.31","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a028142"},{"key":"2024040311301473000_7.6.e202302452.32","doi-asserted-by":"publisher","DOI":"10.1095\/biolreprod.109.079566"},{"key":"2024040311301473000_7.6.e202302452.33","doi-asserted-by":"publisher","DOI":"10.1002\/humu.22028"},{"key":"2024040311301473000_7.6.e202302452.34","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1006631"},{"key":"2024040311301473000_7.6.e202302452.35","doi-asserted-by":"publisher","DOI":"10.1002\/jemt.20457"},{"key":"2024040311301473000_7.6.e202302452.36","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-022-35661-z"},{"key":"2024040311301473000_7.6.e202302452.37","doi-asserted-by":"publisher","DOI":"10.1093\/humupd\/dmab030"},{"key":"2024040311301473000_7.6.e202302452.38","doi-asserted-by":"publisher","DOI":"10.1038\/onc.2016.74"},{"key":"2024040311301473000_7.6.e202302452.39","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2009.03.004"},{"key":"2024040311301473000_7.6.e202302452.40","doi-asserted-by":"publisher","DOI":"10.1074\/mcp.M113.035600"},{"key":"2024040311301473000_7.6.e202302452.41","doi-asserted-by":"publisher","DOI":"10.1530\/REP-18-0350"},{"key":"2024040311301473000_7.6.e202302452.42","doi-asserted-by":"publisher","DOI":"10.4161\/cc.22068"},{"key":"2024040311301473000_7.6.e202302452.43","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.06158-11"},{"key":"2024040311301473000_7.6.e202302452.44","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.28199"},{"key":"2024040311301473000_7.6.e202302452.45","doi-asserted-by":"publisher","DOI":"10.3389\/fcell.2021.662903"},{"key":"2024040311301473000_7.6.e202302452.46","doi-asserted-by":"publisher","DOI":"10.1111\/cge.13525"},{"key":"2024040311301473000_7.6.e202302452.47","doi-asserted-by":"publisher","DOI":"10.1530\/REP-15-0310"},{"key":"2024040311301473000_7.6.e202302452.48","doi-asserted-by":"publisher","DOI":"10.1078\/0171-9335-00097"},{"key":"2024040311301473000_7.6.e202302452.49","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.128587"},{"key":"2024040311301473000_7.6.e202302452.50","doi-asserted-by":"publisher","DOI":"10.1186\/1471-213X-9-23"},{"key":"2024040311301473000_7.6.e202302452.51","doi-asserted-by":"publisher","DOI":"10.1242\/dev.181057"},{"key":"2024040311301473000_7.6.e202302452.52","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201012116"},{"key":"2024040311301473000_7.6.e202302452.53","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a028134"},{"key":"2024040311301473000_7.6.e202302452.54","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.3001034"},{"key":"2024040311301473000_7.6.e202302452.55","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddv123"},{"key":"2024040311301473000_7.6.e202302452.56","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.E15-08-0578"},{"key":"2024040311301473000_7.6.e202302452.57","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra1010172"},{"key":"2024040311301473000_7.6.e202302452.58","doi-asserted-by":"publisher","DOI":"10.1111\/j.1749-6632.1991.tb27312.x"},{"key":"2024040311301473000_7.6.e202302452.59","doi-asserted-by":"publisher","DOI":"10.1095\/biolreprod61.5.1184"},{"key":"2024040311301473000_7.6.e202302452.60","doi-asserted-by":"publisher","DOI":"10.1002\/ar.1092220106"},{"key":"2024040311301473000_7.6.e202302452.61","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2004.12.005"},{"key":"2024040311301473000_7.6.e202302452.62","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.2018355118"},{"key":"2024040311301473000_7.6.e202302452.63","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M112.366690"},{"key":"2024040311301473000_7.6.e202302452.64","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2019-106479"},{"key":"2024040311301473000_7.6.e202302452.65","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.49307"},{"key":"2024040311301473000_7.6.e202302452.66","doi-asserted-by":"publisher","DOI":"10.1093\/nar\/gkg509"},{"key":"2024040311301473000_7.6.e202302452.67","doi-asserted-by":"publisher","DOI":"10.1038\/nmeth0410-248"},{"key":"2024040311301473000_7.6.e202302452.68","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.299"},{"key":"2024040311301473000_7.6.e202302452.69","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-019-09182-1"},{"key":"2024040311301473000_7.6.e202302452.70","doi-asserted-by":"publisher","DOI":"10.1007\/s00439-020-02159-x"},{"key":"2024040311301473000_7.6.e202302452.71","doi-asserted-by":"publisher","DOI":"10.1242\/dev.199922"},{"key":"2024040311301473000_7.6.e202302452.72","doi-asserted-by":"publisher","DOI":"10.1002\/aja.1000990307"},{"key":"2024040311301473000_7.6.e202302452.73","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1015935108"},{"key":"2024040311301473000_7.6.e202302452.74","doi-asserted-by":"publisher","DOI":"10.1093\/biolre\/ioz013"},{"key":"2024040311301473000_7.6.e202302452.75","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1007078"},{"key":"2024040311301473000_7.6.e202302452.76","doi-asserted-by":"publisher","DOI":"10.3389\/fcell.2021.672592"},{"key":"2024040311301473000_7.6.e202302452.77","doi-asserted-by":"publisher","DOI":"10.1002\/pmic.201900205"},{"key":"2024040311301473000_7.6.e202302452.78","doi-asserted-by":"publisher","DOI":"10.1093\/molehr\/gaz056"}],"container-title":["Life Science Alliance"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.26508\/lsa.202302452","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,4,3]],"date-time":"2024-04-03T14:30:29Z","timestamp":1712154629000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.life-science-alliance.org\/lookup\/doi\/10.26508\/lsa.202302452"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,4,3]]},"references-count":78,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2024,4,3]]},"published-print":{"date-parts":[[2024,6]]}},"alternative-id":["10.26508\/lsa.202302452"],"URL":"https:\/\/doi.org\/10.26508\/lsa.202302452","relation":{},"ISSN":["2575-1077"],"issn-type":[{"value":"2575-1077","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,4,3]]}}}