{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T07:00:57Z","timestamp":1772953257447,"version":"3.50.1"},"reference-count":33,"publisher":"MDPI AG","issue":"17","license":[{"start":{"date-parts":[[2022,8,31]],"date-time":"2022-08-31T00:00:00Z","timestamp":1661904000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"DFG","award":["WA2627\/2-2"],"award-info":[{"award-number":["WA2627\/2-2"]}]},{"name":"DFG","award":["KF2020005"],"award-info":[{"award-number":["KF2020005"]}]},{"name":"DFG","award":["KF2021008"],"award-info":[{"award-number":["KF2021008"]}]},{"name":"DFG","award":["31770871"],"award-info":[{"award-number":["31770871"]}]},{"name":"NHC Key Laboratory of Birth Defect for Research and Prevention (Hunan Provincial Maternal","award":["WA2627\/2-2"],"award-info":[{"award-number":["WA2627\/2-2"]}]},{"name":"NHC Key Laboratory of Birth Defect for Research and Prevention (Hunan Provincial Maternal","award":["KF2020005"],"award-info":[{"award-number":["KF2020005"]}]},{"name":"NHC Key Laboratory of Birth Defect for Research and Prevention (Hunan Provincial Maternal","award":["KF2021008"],"award-info":[{"award-number":["KF2021008"]}]},{"name":"NHC Key Laboratory of Birth Defect for Research and Prevention (Hunan Provincial Maternal","award":["31770871"],"award-info":[{"award-number":["31770871"]}]},{"name":"National Natural Science Foundation of China","award":["WA2627\/2-2"],"award-info":[{"award-number":["WA2627\/2-2"]}]},{"name":"National Natural Science Foundation of China","award":["KF2020005"],"award-info":[{"award-number":["KF2020005"]}]},{"name":"National Natural Science Foundation of China","award":["KF2021008"],"award-info":[{"award-number":["KF2021008"]}]},{"name":"National Natural Science Foundation of China","award":["31770871"],"award-info":[{"award-number":["31770871"]}]},{"name":"Qilu Youth Scholar Startup Funding of Shandong University","award":["WA2627\/2-2"],"award-info":[{"award-number":["WA2627\/2-2"]}]},{"name":"Qilu Youth Scholar Startup Funding of Shandong University","award":["KF2020005"],"award-info":[{"award-number":["KF2020005"]}]},{"name":"Qilu Youth Scholar Startup Funding of Shandong University","award":["KF2021008"],"award-info":[{"award-number":["KF2021008"]}]},{"name":"Qilu Youth Scholar Startup Funding of Shandong University","award":["31770871"],"award-info":[{"award-number":["31770871"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cells"],"abstract":"<jats:p>MCPH1 is the first gene identified to be responsible for the human autosomal recessive disorder primary microcephaly (MCPH). Mutations in the N-terminal and central domains of MCPH1 are strongly associated with microcephaly in human patients. A recent study showed that the central domain of MCPH1, which is mainly encoded by exon 8, interacts with E3 ligase \u03b2TrCP2 and regulates the G2\/M transition of the cell cycle. In order to investigate the biological functions of MCPH1\u2019s central domain, we constructed a mouse model that lacked the central domain of MCPH1 by deleting its exon 8 (designated as Mcph1-\u0394e8). Mcph1-\u0394e8 mice exhibited a reduced brain size and thinner cortex, likely caused by a compromised self-renewal capacity and premature differentiation of Mcph1-\u0394e8 neuroprogenitors during corticogenesis. Furthermore, Mcph1-\u0394e8 mice were sterile because of a loss of germ cells in the testis and ovary. The embryonic fibroblasts of Mcph1-\u0394e8 mice exhibited premature chromosome condensation (PCC). All of these findings indicate that Mcph1-\u0394e8 mice are reminiscent of MCPH1 complete knockout mice and Mcph1-\u0394BR1 mice. Our study demonstrates that the central domain of MCPH1 represses microcephaly, and is essential for gonad development in mammals.<\/jats:p>","DOI":"10.3390\/cells11172715","type":"journal-article","created":{"date-parts":[[2022,8,31]],"date-time":"2022-08-31T23:53:21Z","timestamp":1661990001000},"page":"2715","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["The Central Domain of MCPH1 Controls Development of the Cerebral Cortex and Gonads in Mice"],"prefix":"10.3390","volume":"11","author":[{"given":"Yaru","family":"Wang","sequence":"first","affiliation":[{"name":"State Key Laboratory of Microbial Technology, Shandong University, Qingdao 250100, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7800-128X","authenticated-orcid":false,"given":"Wen","family":"Zong","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Microbial Technology, Shandong University, Qingdao 250100, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wenli","family":"Sun","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Microbial Technology, Shandong University, Qingdao 250100, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chengyan","family":"Chen","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Microbial Technology, Shandong University, Qingdao 250100, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8336-3485","authenticated-orcid":false,"given":"Zhao-Qi","family":"Wang","sequence":"additional","affiliation":[{"name":"Leibniz Institute on Aging\u2014Fritz Lipmann Institute (FLI), 07745 Jena, Germany"},{"name":"Faculty of Biological Sciences, Friedrich-Schiller University of Jena, 07743 Jena, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0671-9166","authenticated-orcid":false,"given":"Tangliang","family":"Li","sequence":"additional","affiliation":[{"name":"State Key Laboratory of Microbial Technology, Shandong University, Qingdao 250100, China"},{"name":"Department of Pathology and Pathophysiology, School of Basic Medical Sciences, Hangzhou Normal University, Hangzhou 311121, China"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2022,8,31]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"136","DOI":"10.1086\/341283","article-title":"Identification of microcephalin, a protein implicated in determining the size of the human brain","volume":"71","author":"Jackson","year":"2002","journal-title":"Am. 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