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To understand this association, we studied the role of the microcephaly gene\n                    <jats:italic>Mcph1<\/jats:italic>\n                    in hematological development. Our research revealed that\n                    <jats:italic>Mcph1<\/jats:italic>\n                    -knockout mice exhibited congenital macrocytic anemia due to impaired terminal erythroid differentiation during fetal development. Anemia\u2019s cause is a failure to complete cell division, evident from tetraploid erythroid progenitors with DNA content exceeding 4n. Gene expression profiling demonstrated activation of the p53 pathway in\n                    <jats:italic>Mcph1<\/jats:italic>\n                    -deficient erythroid precursors, leading to overexpression of\n                    <jats:italic>Cdkn1a\/p21<\/jats:italic>\n                    , a major mediator of p53-dependent cell cycle arrest. Surprisingly, fetal brain analysis revealed hypertrophied binucleated neuroprogenitors overexpressing p21 in\n                    <jats:italic>Mcph1<\/jats:italic>\n                    -knockout mice, indicating a shared pathophysiological mechanism underlying both erythroid and neurological defects. However, inactivating p53 in\n                    <jats:italic>Mcph1<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice failed to reverse anemia and microcephaly, suggesting that p53 activation in\n                    <jats:italic>Mcph1<\/jats:italic>\n                    -deficient cells resulted from their proliferation defect rather than causing it. These findings shed new light on Mcph1\u2019s function in fetal hematopoietic development, emphasizing the impact of disrupted cell division on neurogenesis and erythropoiesis\u00a0\u2014\u00a0a common limiting pathway.\n                  <\/jats:p>","DOI":"10.1038\/s44319-024-00123-8","type":"journal-article","created":{"date-parts":[[2024,4,11]],"date-time":"2024-04-11T11:01:56Z","timestamp":1712833316000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Mcph1, mutated in primary microcephaly, is also crucial for erythropoiesis"],"prefix":"10.1038","volume":"25","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1437-0427","authenticated-orcid":false,"given":"Yoann","family":"Vial","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0779-8863","authenticated-orcid":false,"given":"Jeannette","family":"Nardelli","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adeline A","family":"Bonnard","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-7051-6998","authenticated-orcid":false,"given":"Justine","family":"Rousselot","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mich\u00e8le","family":"Souyri","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0909-4221","authenticated-orcid":false,"given":"Pierre","family":"Gressens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2840-1511","authenticated-orcid":false,"given":"H\u00e9l\u00e8ne","family":"Cav\u00e9","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6777-4639","authenticated-orcid":false,"given":"S\u00e9verine","family":"Drunat","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,4,11]]},"reference":[{"key":"123_CR1","doi-asserted-by":"publisher","first-page":"725","DOI":"10.1038\/ncb1431","volume":"8","author":"GK Alderton","year":"2006","unstructured":"Alderton GK, Galbiati L, Griffith E, Surinya KH, Neitzel H, Jackson AP, Jeggo PA, O\u2019Driscoll M (2006) Regulation of mitotic entry by microcephalin and its overlap with ATR signalling. 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