{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,12]],"date-time":"2026-07-12T02:29:31Z","timestamp":1783823371582,"version":"3.55.0"},"reference-count":26,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2009,9,16]],"date-time":"2009-09-16T00:00:00Z","timestamp":1253059200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2009,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Ts65Dn mice have segmental trisomy for orthologs of about half of the genes on human chromosome 21, including <jats:italic>Ets2<\/jats:italic>. These mice develop anomalies of the cranial skeleton and thymus that parallel those in Down syndrome. Overexpression of the <jats:italic>Ets2<\/jats:italic> transcription factor gene was posited to be sufficient to produce these craniofacial and thymus deficits in transgenic mice that constitutively overexpress a processed <jats:italic>Ets2<\/jats:italic> transcript under a promiscuous promoter [Sumarsono et al. (1996); Nature 379:534\u2013537; Wolvetang et al. (2003); Hum Mol Genet 12:247\u2013255]. Evaluation of trisomic mice with varying copy numbers of a properly regulated <jats:italic>Ets2<\/jats:italic> gene indicated increased dosage of <jats:italic>Ets2<\/jats:italic> was not sufficient to produce effects on thymus and most of the cranial anomalies seen in Ts65Dn mice. However, mesoderm\u2010derived cranial skeletal elements are significantly more affected in Ts65Dn, <jats:italic>Ets2<\/jats:italic><jats:sup>+\/\u2212<\/jats:sup> mice compared to Ts65Dn littermates suggesting a differential interaction of <jats:italic>Ets2<\/jats:italic>\u2010related processes with mesoderm\u2010derived and neural crest\u2010derived formative tissues. Our results support the growing evidence for interactions among multiple genes contributing to developmental perturbations resulting in variation in complex Down syndrome phenotypes. \u00a9 2009 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/ajmg.a.33012","type":"journal-article","created":{"date-parts":[[2009,9,17]],"date-time":"2009-09-17T01:37:37Z","timestamp":1253151457000},"page":"2158-2165","source":"Crossref","is-referenced-by-count":28,"title":["Complex contributions of <i>Ets2<\/i> to craniofacial and thymus phenotypes of trisomic \u201cDown syndrome\u201d mice"],"prefix":"10.1002","volume":"149A","author":[{"given":"Cheryl A.","family":"Hill","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas E.","family":"Sussan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roger H.","family":"Reeves","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joan T.","family":"Richtsmeier","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2009,9,24]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/9.2.195"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/bies.200800133"},{"key":"e_1_2_6_4_1","first-page":"347","article-title":"Measuring precision of three\u2010dimensional landmark data","volume":"3","author":"Corner BD","year":"1992","journal-title":"J Quant Anthropol"},{"key":"e_1_2_6_5_1","first-page":"117","article-title":"Segmental trisomy as a mouse model for Down syndrome","volume":"384","author":"Davisson MT","year":"1993","journal-title":"Prog Clin Biol Res"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1159\/000472398"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.02277"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.2001.0487"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1101\/gr.1951304"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.91.11.4997"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1201\/9781420036176"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-7580.2005.00473.x"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1098992"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddm022"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320370751"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.onc.1204037"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569(1995)032<0217:PRAVOT>2.3.CO;2"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-0177(200002)217:2<137::AID-DVDY1>3.0.CO;2-N"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/jez.b.21092"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0925-4773(02)00126-0"},{"key":"e_1_2_6_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejca.2005.08.013"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1038\/379534a0"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature06446"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pedhc.2005.10.004"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pedhc.2006.02.006"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2009-03-211391"},{"key":"e_1_2_6_27_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddg015"}],"container-title":["American Journal of Medical Genetics Part A"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.a.33012","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.a.33012","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,11]],"date-time":"2023-10-11T20:20:16Z","timestamp":1697055616000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.a.33012"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2009,9,24]]},"references-count":26,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2009,10]]}},"alternative-id":["10.1002\/ajmg.a.33012"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.a.33012","archive":["Portico"],"relation":{},"ISSN":["1552-4825","1552-4833"],"issn-type":[{"value":"1552-4825","type":"print"},{"value":"1552-4833","type":"electronic"}],"subject":[],"published":{"date-parts":[[2009,9,24]]}}}