{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T05:04:36Z","timestamp":1778043876543,"version":"3.51.4"},"reference-count":51,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2021,8,9]],"date-time":"2021-08-09T00:00:00Z","timestamp":1628467200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001665","name":"Agence Nationale de la Recherche","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001665","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,12,17]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Fanconi anemia (FA) is a rare human genetic disorder characterized by bone marrow failure, predisposition to cancer and developmental defects including hypogonadism. Reproductive defects leading to germ cell aplasia are the most consistent phenotypes seen in FA mouse models. We examined the role of the nuclear FA core complex gene Fancg in the development of primordial germ cells (PGCs), the embryonic precursors of adult gametes, during fetal development. PGC maintenance was severely impaired in Fancg\u2212\/\u2212 embryos. We observed a defect in the number of PGCs starting at E9.5 and a strong attrition at E11.5 and E13.5. Remarkably, we observed a mosaic pattern reflecting a portion of testicular cords devoid of PGCs in E13.5 fetal gonads. Our in vitro and in vivo data highlight a potential role of Fancg in the proliferation and in the intrinsic cell motility abilities of PGCs. The random migratory process is abnormally activated in Fancg\u2212\/\u2212 PGCs, altering the migration of cells. Increased cell death and PGC attrition observed in E11.5 Fancg\u2212\/\u2212 embryos are features consistent with delayed migration of PGCs along the migratory pathway to the genital ridges. Moreover, we show that an inhibitor of RAC1 mitigates the abnormal migratory pattern observed in Fancg\u2212\/\u2212 PGCs.<\/jats:p>","DOI":"10.1093\/hmg\/ddab222","type":"journal-article","created":{"date-parts":[[2021,8,4]],"date-time":"2021-08-04T11:45:27Z","timestamp":1628077527000},"page":"97-110","source":"Crossref","is-referenced-by-count":11,"title":["Abnormal migration behavior linked to Rac1 signaling contributes to primordial germ cell exhaustion in Fanconi anemia pathway-deficient <i>Fancg<\/i>\u2212\/\u2212 embryos"],"prefix":"10.1093","volume":"31","author":[{"given":"Amandine","family":"Jarysta","sequence":"first","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire des Cellules Souches Germinales, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lydia","family":"Riou","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire des Cellules Souches Germinales, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Virginie","family":"Firlej","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire des Cellules Souches Germinales, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cl\u00e9mentine","family":"Lapoujade","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire des Cellules Souches Germinales, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Thierry","family":"Kortulewski","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, Inserm, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire de RadioPathologie, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vilma","family":"Barroca","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, Inserm, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anne-Sophie","family":"Gille","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire des Cellules Souches Germinales, Fontenay-aux-Roses F-92265, France"},{"name":"D\u00e9partement de G\u00e9n\u00e9tique, D\u00e9veloppement et Cancer. Team From Gametes to Birth, Institut Cochin, INSERM U1016, Paris F-75014, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Florent","family":"Dumont","sequence":"additional","affiliation":[{"name":"Universit\u00e9 Paris Saclay, UMS IPSIT, Ch\u00e2tenay-Malabry F-92296, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"S\u00e9bastien","family":"Jacques","sequence":"additional","affiliation":[{"name":"Plate-Forme S\u00e9quen\u00e7age et G\u00e9nomique, Institut Cochin, Inserm U1016, Universit\u00e9 de Paris, 22 rue M\u00e9chain Paris 75014, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Franck","family":"Letourneur","sequence":"additional","affiliation":[{"name":"Plate-Forme S\u00e9quen\u00e7age et G\u00e9nomique, Institut Cochin, Inserm U1016, Universit\u00e9 de Paris, 22 rue M\u00e9chain Paris 75014, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Filippo","family":"Rosselli","sequence":"additional","affiliation":[{"name":"CNRS-UMR9019, Int\u00e9grit\u00e9 du G\u00e9nome et Cancers, Universit\u00e9 Paris-Saclay, Villejuif 94805, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Isabelle","family":"Allemand","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire des Cellules Souches Germinales, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0891-079X","authenticated-orcid":false,"given":"Pierre","family":"Fouchet","sequence":"additional","affiliation":[{"name":"Universit\u00e9 de Paris and Universit\u00e9 Paris-Saclay, iRCM\/IBFJ CEA, UMR Stabilit\u00e9 G\u00e9n\u00e9tique Cellules Souches et Radiations, Laboratoire des Cellules Souches Germinales, Fontenay-aux-Roses F-92265, France"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2021,8,9]]},"reference":[{"key":"2021121717444961000_ref1","doi-asserted-by":"crossref","first-page":"337","DOI":"10.1038\/nrm.2016.48","article-title":"The Fanconi anaemia pathway: new players and new functions","volume":"17","author":"Ceccaldi","year":"2016","journal-title":"Nat. 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