{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,12]],"date-time":"2026-03-12T00:41:44Z","timestamp":1773276104810,"version":"3.50.1"},"reference-count":62,"publisher":"Wiley","issue":"22","license":[{"start":{"date-parts":[[2024,11,21]],"date-time":"2024-11-21T00:00:00Z","timestamp":1732147200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100003725","name":"National Research Foundation of Korea","doi-asserted-by":"publisher","award":["RS\u20102024\u201000344272"],"award-info":[{"award-number":["RS\u20102024\u201000344272"]}],"id":[{"id":"10.13039\/501100003725","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003725","name":"National Research Foundation of Korea","doi-asserted-by":"publisher","award":["2014M3A9D5A01075128"],"award-info":[{"award-number":["2014M3A9D5A01075128"]}],"id":[{"id":"10.13039\/501100003725","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003710","name":"Korea Health Industry Development Institute","doi-asserted-by":"publisher","award":["HI21C169202"],"award-info":[{"award-number":["HI21C169202"]}],"id":[{"id":"10.13039\/501100003710","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100005006","name":"Asan Institute for Life Sciences, Asan Medical Center","doi-asserted-by":"publisher","award":["2021IP0050"],"award-info":[{"award-number":["2021IP0050"]}],"id":[{"id":"10.13039\/501100005006","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["faseb.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The FASEB Journal"],"published-print":{"date-parts":[[2024,11,30]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>\n                    Cep215\/Cdk5rap2 is a centrosome protein crucial for directing microtubule organization during cell division and morphology.\n                    <jats:italic>Cep215<\/jats:italic>\n                    is a causal gene of autosomal recessive primary microcephaly type 3, characterized by a small brain size and a thin cerebral cortex. Despite previous attempts with\n                    <jats:italic>Cep215<\/jats:italic>\n                    knockout (KO) mice to elucidate its developmental roles, interpreting their phenotypes remained challenging due to potential interference from alternative variants. Here, we generated KO mice completely lacking the\n                    <jats:italic>Cep215<\/jats:italic>\n                    gene and investigated its specific contributions to male germ cell development. In the absence of Cep215, testis size decreased significantly, accompanied by a reduction in male germ cell numbers. Histological analyses unveiled the arrested development of male germ cells around the zygotene stage of meiosis. Concurrently, the formation of the blood\u2010testis barrier (BTB) was impaired in\n                    <jats:italic>Cep215<\/jats:italic>\n                    KO testes. These findings suggest that BTB failure contributes, at least partially, to male germ cell defects observed in\n                    <jats:italic>Cep215<\/jats:italic>\n                    KO mice. We propose that the deletion of\n                    <jats:italic>Cep215<\/jats:italic>\n                    may disrupt microtubule organization in Sertoli cells with a delay in spermatogonial stem cell mitosis, thereby impeding proper BTB formation.\n                  <\/jats:p>","DOI":"10.1096\/fj.202401541r","type":"journal-article","created":{"date-parts":[[2024,11,21]],"date-time":"2024-11-21T08:45:26Z","timestamp":1732178726000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Roles of Cep215\/Cdk5rap2 in establishing testicular architecture for mouse male germ cell development"],"prefix":"10.1096","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-2326-3700","authenticated-orcid":false,"given":"Donghee","family":"Kang","sequence":"first","affiliation":[{"name":"Department of Biological Sciences Seoul National University  Seoul Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6287-5991","authenticated-orcid":false,"given":"Byungho","family":"Shin","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences Seoul National University  Seoul Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gyeong\u2010Nam","family":"Kim","sequence":"additional","affiliation":[{"name":"Department of Cell and Genetic Engineering Asan Medical Center, University of Ulsan College of Medicine  Seoul Korea"},{"name":"Asan Institute for Life Sciences, ConveRgence mEDIcine research cenTer (CREDIT) Asan Medical Center  Seoul Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0003-0426-2593","authenticated-orcid":false,"given":"Ji Hwa","family":"Hea","sequence":"additional","affiliation":[{"name":"Department of Cell and Genetic Engineering Asan Medical Center, University of Ulsan College of Medicine  Seoul Korea"},{"name":"Asan Institute for Life Sciences, ConveRgence mEDIcine research cenTer (CREDIT) Asan Medical Center  Seoul Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1508-9883","authenticated-orcid":false,"given":"Young Hoon","family":"Sung","sequence":"additional","affiliation":[{"name":"Department of Cell and Genetic Engineering Asan Medical Center, University of Ulsan College of Medicine  Seoul Korea"},{"name":"Asan Institute for Life Sciences, ConveRgence mEDIcine research cenTer (CREDIT) Asan Medical Center  Seoul Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5051-876X","authenticated-orcid":false,"given":"Kunsoo","family":"Rhee","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences Seoul National University  Seoul Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2024,11,21]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tcb.2011.04.007"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.e07-05-0474"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.e07-04-0371"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201007030"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.4161\/cc.9.4.10667"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.200912163"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0087016"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M110.105965"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng1539"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/boc.201900090"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2350-8-58"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/ahg.12232"},{"key":"e_1_2_10_14_1","first-page":"84","article-title":"Whole exome sequencing identifies a novel mutation in ASPM and ultra\u2010rare mutation in CDK5RAP2 causing primary microcephaly in consanguineous Pakistani families","volume":"38","author":"Makhdoom EUH","year":"2022","journal-title":"Pak J Med Sci"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF01741984"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.exphem.2005.06.021"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.040410"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.stemcr.2017.01.002"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbd.2019.05.008"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/ahg.12343"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2010.05.017"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.15252\/embj.2021108739"},{"key":"e_1_2_10_23_1","unstructured":"Mouse genome informatics. Accessed June 14 2024.https:\/\/www.informatics.jax.org\/"},{"key":"e_1_2_10_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2020.03.077"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1093\/biolre\/ioy027"},{"key":"e_1_2_10_26_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1219969"},{"key":"e_1_2_10_27_1","doi-asserted-by":"publisher","DOI":"10.1038\/nmeth.2857"},{"key":"e_1_2_10_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbrc.2020.12.072"},{"key":"e_1_2_10_29_1","doi-asserted-by":"publisher","DOI":"10.1038\/nbt.3614"},{"key":"e_1_2_10_30_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0142755"},{"key":"e_1_2_10_31_1","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.078329"},{"key":"e_1_2_10_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2017.06.033"},{"key":"e_1_2_10_33_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12031-018-1062-x"},{"key":"e_1_2_10_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2017.08.031"},{"key":"e_1_2_10_35_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0136684"},{"key":"e_1_2_10_36_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.134.3482.832"},{"key":"e_1_2_10_37_1","doi-asserted-by":"publisher","DOI":"10.1095\/biolreprod.103.017020"},{"key":"e_1_2_10_38_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-019-50855-0"},{"key":"e_1_2_10_39_1","doi-asserted-by":"publisher","DOI":"10.1111\/febs.17212"},{"key":"e_1_2_10_40_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1400568111"},{"key":"e_1_2_10_41_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10059-009-0097-9"},{"key":"e_1_2_10_42_1","doi-asserted-by":"publisher","DOI":"10.15252\/embr.202051030"},{"key":"e_1_2_10_43_1","doi-asserted-by":"publisher","DOI":"10.15252\/embr.202051023"},{"key":"e_1_2_10_44_1","doi-asserted-by":"publisher","DOI":"10.1080\/15384101.2021.1950386"},{"key":"e_1_2_10_45_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.semcdb.2021.06.016"},{"key":"e_1_2_10_46_1","doi-asserted-by":"publisher","DOI":"10.1210\/er.2014-1101"},{"key":"e_1_2_10_47_1","doi-asserted-by":"publisher","DOI":"10.1002\/ar.20417"},{"key":"e_1_2_10_48_1","doi-asserted-by":"publisher","DOI":"10.3389\/fcell.2023.1114769"},{"key":"e_1_2_10_49_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0198100"},{"key":"e_1_2_10_50_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.348866.121"},{"key":"e_1_2_10_51_1","doi-asserted-by":"publisher","DOI":"10.15252\/embj.2020106118"},{"key":"e_1_2_10_52_1","doi-asserted-by":"publisher","DOI":"10.1096\/fj.202100960R"},{"key":"e_1_2_10_53_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajpath.2015.10.007"},{"key":"e_1_2_10_54_1","doi-asserted-by":"publisher","DOI":"10.1534\/genetics.113.150789"},{"key":"e_1_2_10_55_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ceb.2016.09.003"},{"key":"e_1_2_10_56_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00418-021-01999-6"},{"key":"e_1_2_10_57_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.celrep.2021.109885"},{"key":"e_1_2_10_58_1","first-page":"165","article-title":"The perinuclear centriole\u2010containing centrosome is not the major microtubule organizing center in Sertoli cells","volume":"66","author":"Vogl AW","year":"1995","journal-title":"Eur J Cell Biol"},{"key":"e_1_2_10_59_1","doi-asserted-by":"publisher","DOI":"10.1095\/biolreprod.102.011791"},{"key":"e_1_2_10_60_1","doi-asserted-by":"publisher","DOI":"10.1096\/fj.201700681R"},{"key":"e_1_2_10_61_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.add9528"},{"key":"e_1_2_10_62_1","doi-asserted-by":"publisher","DOI":"10.15252\/embr.202051127"},{"key":"e_1_2_10_63_1","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.94694"}],"container-title":["The FASEB Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/faseb.onlinelibrary.wiley.com\/doi\/pdf\/10.1096\/fj.202401541R","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,28]],"date-time":"2025-10-28T17:04:47Z","timestamp":1761671087000},"score":1,"resource":{"primary":{"URL":"https:\/\/faseb.onlinelibrary.wiley.com\/doi\/10.1096\/fj.202401541R"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,11,21]]},"references-count":62,"journal-issue":{"issue":"22","published-print":{"date-parts":[[2024,11,30]]}},"alternative-id":["10.1096\/fj.202401541R"],"URL":"https:\/\/doi.org\/10.1096\/fj.202401541r","archive":["Portico"],"relation":{},"ISSN":["0892-6638","1530-6860"],"issn-type":[{"value":"0892-6638","type":"print"},{"value":"1530-6860","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,11,21]]},"assertion":[{"value":"2024-07-06","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-11-05","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-11-21","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70188"}}