{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T12:24:17Z","timestamp":1790771057001,"version":"4.1.0"},"reference-count":45,"publisher":"The Company of Biologists","license":[{"start":{"date-parts":[[2019,1,1]],"date-time":"2019-01-01T00:00:00Z","timestamp":1546300800000},"content-version":"am","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"funder":[{"DOI":"10.13039\/501100003329","name":"Ministerio de Econom?a y Competitividad","doi-asserted-by":"publisher","award":["BFU2015-68354"],"award-info":[{"award-number":["BFU2015-68354"]}],"id":[{"id":"10.13039\/501100003329","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003329","name":"Ministerio de Econom?a y Competitividad","doi-asserted-by":"publisher","award":["PI13\/00864"],"award-info":[{"award-number":["PI13\/00864"]}],"id":[{"id":"10.13039\/501100003329","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01CA220551"],"award-info":[{"award-number":["R01CA220551"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:p>GEMC1 and MCIDAS are Geminin family proteins that transcriptionally activate E2F4\/5-target genes during multiciliogenesis, including FoxJ1 and Ccno. Male mice lacking Gemc1, Mcidas or Ccno were found to be infertile, but the origin of this defect has remained unclear. Here we show that all three genes are necessary for the generation of functional multiciliated cells in the efferent ducts that are required for spermatozoa to enter the epididymis. In mice mutant for Gemc1, Mcidas or Ccno, we observed a similar spectrum of phenotypes, including thinning of the seminiferous tubule epithelia, dilation of the rete testes, sperm agglutinations in the efferent ducts and lack of spermatozoa in the epididymis (azoospermia). These data suggest that defective efferent duct development is the dominant cause of male infertility in these mouse models and this likely extends to patients with the ciliopathy Reduced Generation of Multiple Motile Cilia with mutations in MCIDAS and CCNO.<\/jats:p>","DOI":"10.1242\/dev.162628","type":"journal-article","created":{"date-parts":[[2019,4,1]],"date-time":"2019-04-01T15:36:16Z","timestamp":1554132976000},"source":"Crossref","is-referenced-by-count":55,"title":["Defects in efferent duct multiciliogenesis underlie male infertility in GEMC1, MCIDAS or CCNO deficient mice"],"prefix":"10.1242","author":[{"given":"Berta","family":"Terr\u00e9","sequence":"first","affiliation":[{"name":"Institute for Research in Biomedicine (IRB Barcelona), The Barcelona Institute of Science and Technology, Barcelona 08028, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Lewis","sequence":"additional","affiliation":[{"name":"Institute for Research in Biomedicine (IRB Barcelona), The Barcelona Institute of Science and Technology, Barcelona 08028, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gabriel","family":"Gil-G\u00f3mez","sequence":"additional","affiliation":[{"name":"Apoptosis Signalling Group, IMIM (Institut Hospital del Mar d'Investigacions M\u00e8diques), Barcelona, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhiyuan","family":"Han","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, New York Institute of Technology College of Osteopathic Medicine, Old Westbury, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lu","family":"Hao","sequence":"additional","affiliation":[{"name":"Institute of Molecular and Cell Biology, Proteos, 61 Biopolis Drive, Singapore 138673, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M\u00f2nica","family":"Aguilera","sequence":"additional","affiliation":[{"name":"Institute for Research in Biomedicine (IRB Barcelona), The Barcelona Institute of Science and Technology, Barcelona 08028, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Neus","family":"Prats","sequence":"additional","affiliation":[{"name":"Institute for Research in Biomedicine (IRB Barcelona), The Barcelona Institute of Science and Technology, Barcelona 08028, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sudipto","family":"Roy","sequence":"additional","affiliation":[{"name":"Institute of Molecular and Cell Biology, Proteos, 61 Biopolis Drive, Singapore 138673, Singapore"},{"name":"Department of Pediatrics, Yong Loo Lin School of Medicine, National University of Singapore, 1E Kent Ridge Road, 119288, Singapore"},{"name":"Department of Biological Sciences, National University of Singapore, 14 Science Drive 4, Singapore 117543, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2315-8452","authenticated-orcid":false,"given":"Haotian","family":"Zhao","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences, New York Institute of Technology College of Osteopathic Medicine, Old Westbury, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8650-2081","authenticated-orcid":false,"given":"Travis H.","family":"Stracker","sequence":"additional","affiliation":[{"name":"Institute for Research in Biomedicine (IRB Barcelona), The Barcelona Institute of Science and Technology, Barcelona 08028, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2019,1,1]]},"reference":[{"key":"2021042512421343200_DEV162628C1","doi-asserted-by":"publisher","first-page":"433","DOI":"10.1679\/aohc.51.433","article-title":"Changes in distribution and staining reactivity of PAS-positive material in the mouse epididymal duct after efferent duct ligation","volume":"51","author":"Abe","year":"1988","journal-title":"Arch. 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