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We report the generation, molecular validation and phenotypic characterization of a novel rat model with a truncating Magel2 mutation modeling variants associated with Schaaf-Yang syndrome-causing mutations. Within the hypothalamus, a brain region in which human MAGEL2 is paternally expressed, we demonstrated, at the level of transcript and peptide detection, that rat Magel2 exhibits a paternal, parent-of-origin effect. In evaluations of behavioral features across several domains, juvenile Magel2 mutant rats displayed alterations in anxiety-like behavior and sociability measures. Moreover, the analysis of peripheral organ systems detected alterations in body composition, cardiac structure and function, and breathing irregularities in Magel2 mutant rats. Several of these findings are concordant with reported mouse phenotypes, indicating the conservation of MAGEL2 function across rodent species. Our comprehensive analysis revealing impairments across multiple domains demonstrates the tractability of this model system for the study of truncating MAGEL2 mutations.<\/jats:p>","DOI":"10.1242\/dmm.049829","type":"journal-article","created":{"date-parts":[[2023,1,13]],"date-time":"2023-01-13T14:53:29Z","timestamp":1673621609000},"source":"Crossref","is-referenced-by-count":9,"title":["<i>Magel2<\/i> truncation alters select behavioral and physiological outcomes in a rat model of Schaaf-Yang syndrome"],"prefix":"10.1242","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7788-9790","authenticated-orcid":false,"given":"Derek L.","family":"Reznik","sequence":"first","affiliation":[{"name":"Baylor College of Medicine 1 , Department of Molecular and Human Genetics, Houston, TX 77030 , USA"},{"name":"Texas Children's Hospital, Jan and Dan Duncan Neurological Research Institute 2 , Houston, TX 77030 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3669-8207","authenticated-orcid":false,"given":"Mingxiao V.","family":"Yang","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 1 , Department of Molecular and Human Genetics, Houston, TX 77030 , USA"},{"name":"Texas Children's Hospital, Jan and Dan Duncan Neurological Research Institute 2 , Houston, TX 77030 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0891-9295","authenticated-orcid":false,"given":"Pedro","family":"Albelda de la Haza","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine 1 , Department of Molecular and Human Genetics, Houston, TX 77030 , USA"},{"name":"Texas Children's Hospital, Jan and Dan Duncan Neurological Research Institute 2 , Houston, TX 77030 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2815-3645","authenticated-orcid":false,"given":"Antrix","family":"Jain","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine, Mass Spectrometry Proteomics Core 3 , Houston, TX 77030 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Melanie","family":"Spanjaard","sequence":"additional","affiliation":[{"name":"Heidelberg University, Institute of Human Genetics 4 , Im Neuenheimer Feld 366, 69120 Heidelberg , Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Susanne","family":"Theiss","sequence":"additional","affiliation":[{"name":"Heidelberg University, Institute of Human Genetics 4 , Im Neuenheimer Feld 366, 69120 Heidelberg , Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2148-7490","authenticated-orcid":false,"given":"Christian P.","family":"Schaaf","sequence":"additional","affiliation":[{"name":"Heidelberg University, Institute of Human Genetics 4 , Im Neuenheimer Feld 366, 69120 Heidelberg , Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2953-6485","authenticated-orcid":false,"given":"Anna","family":"Malovannaya","sequence":"additional","affiliation":[{"name":"Baylor College of Medicine, Mass Spectrometry Proteomics Core 3 , Houston, TX 77030 , USA"},{"name":"Baylor College of Medicine 5 , Verna and Marrs McLean Departments of Biochemistry and Molecular Biology, and Molecular and Cellular Biology, Houston, TX 77030 , USA"},{"name":"Baylor College of Medicine, Dan L. 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