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Appendicular skeletal deficits also appear in males before females with DS. In femurs of male Ts65Dn DS model mice, cortical deficits were pronounced throughout development, but trabecular deficits and Dyrk1a overexpression were transitory until postnatal day (P) 30, when there were persistent trabecular and cortical deficits and Dyrk1a was trending toward overexpression. Correction of DS-related skeletal deficits by a purported DYRK1A inhibitor or through genetic means beginning at P21 was not effective at P30, but germline normalization of Dyrk1a improved male bone structure by P36. Trabecular and cortical deficits in female Ts65Dn mice were evident at P30 but subsided by P36, typifying periodic developmental skeletal normalizations that progressed to more prominent bone deficiencies. Sex-dependent differences in skeletal deficits with a delayed impact of trisomic Dyrk1a are important to find temporally specific treatment periods for bone and other phenotypes associated with trisomy 21.<\/jats:p>","DOI":"10.1242\/dmm.050914","type":"journal-article","created":{"date-parts":[[2024,8,13]],"date-time":"2024-08-13T07:59:47Z","timestamp":1723535987000},"source":"Crossref","is-referenced-by-count":10,"title":["Sex-specific trisomic <i>Dyrk1a<\/i>-related skeletal phenotypes during development in a Down syndrome model"],"prefix":"10.1242","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8867-6018","authenticated-orcid":false,"given":"Jonathan M.","family":"LaCombe","sequence":"first","affiliation":[{"name":"Indiana University Indianapolis 1 Department of Biology , , Indianapolis, IN 46202 , USA"},{"name":"Labcorp Early Development Laboratories, Inc. 2 , Greenfield, IN 46140 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-3552-6085","authenticated-orcid":false,"given":"Kourtney","family":"Sloan","sequence":"additional","affiliation":[{"name":"Indiana University Indianapolis 1 Department of Biology , , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jared R.","family":"Thomas","sequence":"additional","affiliation":[{"name":"Indiana University Indianapolis 1 Department of Biology , , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew P.","family":"Blackwell","sequence":"additional","affiliation":[{"name":"Indiana University Indianapolis 1 Department of Biology , , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabella","family":"Crawford","sequence":"additional","affiliation":[{"name":"Indiana University Indianapolis 1 Department of Biology , , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Flannery","family":"Bishop","sequence":"additional","affiliation":[{"name":"Indiana University Indianapolis 1 Department of Biology , , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6077-8058","authenticated-orcid":false,"given":"Joseph M.","family":"Wallace","sequence":"additional","affiliation":[{"name":"Purdue University 3 Department of Biomedical Engineering , , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9860-5037","authenticated-orcid":false,"given":"Randall J.","family":"Roper","sequence":"additional","affiliation":[{"name":"Indiana University Indianapolis 1 Department of Biology , , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,9,23]]},"reference":[{"key":"2024092308413519900_DMM050914C1","doi-asserted-by":"publisher","first-page":"102","DOI":"10.1038\/bonekey.2012.102","article-title":"Aging mechanisms in bone","volume":"1","author":"Almeida","year":"2012","journal-title":"Bonekey Rep."},{"key":"2024092308413519900_DMM050914C2","doi-asserted-by":"publisher","first-page":"648","DOI":"10.1007\/s003300050726","article-title":"Bone mineral density in adults with Down's syndrome","volume":"9","author":"Angelopoulou","year":"1999","journal-title":"Eur. 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