{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T20:31:14Z","timestamp":1790713874193,"version":"4.1.0"},"reference-count":54,"publisher":"MDPI AG","issue":"11","license":[{"start":{"date-parts":[[2021,11,16]],"date-time":"2021-11-16T00:00:00Z","timestamp":1637020800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Genes"],"abstract":"<jats:p>Down syndrome (DS) is the most common chromosomal disorder. It is responsible for intellectual disability (ID) and several medical conditions. Although men with DS are thought to be infertile, some spontaneous paternities have been reported. The few studies of the mechanism of infertility in men with DS are now dated. Recent research in zebrafish has indicated that overexpression of DYRK1A (the protein primarily responsible for ID in DS) impairs gonadogenesis at the embryonic stage. To better ascertain DYRK1A\u2019s role in infertility in DS, we investigated the effect of DYRK1A overexpression in a transgenic mouse model. We found that overexpression of DYRK1A impairs fertility in transgenic male mice. Interestingly, the mechanism in mice differs slightly from that observed in zebrafish but, with disruption of the early stages of spermatogenesis, is similar to that seen in humans. Unexpectedly, we observed hypogonadotropic hypogonadism in the transgenic mice.<\/jats:p>","DOI":"10.3390\/genes12111800","type":"journal-article","created":{"date-parts":[[2021,11,16]],"date-time":"2021-11-16T11:32:03Z","timestamp":1637062323000},"page":"1800","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["DYRK1A Overexpression in Mice Downregulates the Gonadotropic Axis and Disturbs Early Stages of Spermatogenesis"],"prefix":"10.3390","volume":"12","author":[{"given":"Rodolphe","family":"Dard","sequence":"first","affiliation":[{"name":"Laboratoire Processus D\u00e9g\u00e9n\u00e9ratifs, Stress et Vieillissement, Unit\u00e9 de Biologie Fonctionnelle et Adaptative (BFA), UMR 8251 CNRS, Universit\u00e9 de Paris, 75205 Paris, France"},{"name":"Universit\u00e9 Paris-Saclay, UVSQ, INRAE, ENVA, BREED, 78350 Jouy-en-Josas, France"},{"name":"D\u00e9partement de G\u00e9n\u00e9tique, CHI de Poissy St Germain en Laye, 78300 Poissy, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manon","family":"Moreau","sequence":"additional","affiliation":[{"name":"Laboratoire Processus D\u00e9g\u00e9n\u00e9ratifs, Stress et Vieillissement, Unit\u00e9 de Biologie Fonctionnelle et Adaptative (BFA), UMR 8251 CNRS, Universit\u00e9 de Paris, 75205 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Estelle","family":"Parizot","sequence":"additional","affiliation":[{"name":"Laboratoire Processus D\u00e9g\u00e9n\u00e9ratifs, Stress et Vieillissement, Unit\u00e9 de Biologie Fonctionnelle et Adaptative (BFA), UMR 8251 CNRS, Universit\u00e9 de Paris, 75205 Paris, France"},{"name":"Universit\u00e9 Paris-Saclay, UVSQ, INRAE, ENVA, BREED, 78350 Jouy-en-Josas, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Farah","family":"Ghieh","sequence":"additional","affiliation":[{"name":"Universit\u00e9 Paris-Saclay, UVSQ, INRAE, ENVA, BREED, 78350 Jouy-en-Josas, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leslie","family":"Brehier","sequence":"additional","affiliation":[{"name":"Universit\u00e9 Paris-Saclay, UVSQ, INRAE, ENVA, BREED, 78350 Jouy-en-Josas, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nadim","family":"Kassis","sequence":"additional","affiliation":[{"name":"Laboratoire Processus D\u00e9g\u00e9n\u00e9ratifs, Stress et Vieillissement, Unit\u00e9 de Biologie Fonctionnelle et Adaptative (BFA), UMR 8251 CNRS, Universit\u00e9 de Paris, 75205 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Val\u00e9rie","family":"Serazin","sequence":"additional","affiliation":[{"name":"D\u00e9partement de G\u00e9n\u00e9tique, CHI de Poissy St Germain en Laye, 78300 Poissy, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonin","family":"Lamaziere","sequence":"additional","affiliation":[{"name":"Centre de Recherche Saint-Antoine (CRSA), Sorbonne Universit\u00e9-INSERM, 75012 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chryst\u00e8le","family":"Racine","sequence":"additional","affiliation":[{"name":"Centre de Recherche Saint-Antoine (CRSA), Sorbonne Universit\u00e9-INSERM, 75012 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie","family":"di Clemente","sequence":"additional","affiliation":[{"name":"Centre de Recherche Saint-Antoine (CRSA), Sorbonne Universit\u00e9-INSERM, 75012 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5774-2756","authenticated-orcid":false,"given":"Fran\u00e7ois","family":"Vialard","sequence":"additional","affiliation":[{"name":"Universit\u00e9 Paris-Saclay, UVSQ, INRAE, ENVA, BREED, 78350 Jouy-en-Josas, France"},{"name":"D\u00e9partement de G\u00e9n\u00e9tique, CHI de Poissy St Germain en Laye, 78300 Poissy, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie","family":"Janel","sequence":"additional","affiliation":[{"name":"Laboratoire Processus D\u00e9g\u00e9n\u00e9ratifs, Stress et Vieillissement, Unit\u00e9 de Biologie Fonctionnelle et Adaptative (BFA), UMR 8251 CNRS, Universit\u00e9 de Paris, 75205 Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2021,11,16]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1163","DOI":"10.1016\/j.jpeds.2013.06.013","article-title":"Current Estimate of Down Syndrome Population Prevalence in the United States","volume":"163","author":"Presson","year":"2013","journal-title":"J. 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