Variations in maternal vitamin A intake modifies phenotypes in a mouse model of 22q11.2 deletion syndrome

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dc.contributor.authorYitsege, Gelilaen
dc.contributor.authorStokes, Bethany A.en
dc.contributor.authorSabatino, Julia A.en
dc.contributor.authorSugrue, Kelsey F.en
dc.contributor.authorBanyai, Gaboren
dc.contributor.authorParonett, Elizabeth M.en
dc.contributor.authorKarpinski, Beverly A.en
dc.contributor.authorMaynard, Thomas M.en
dc.contributor.authorLaMantia, Anthony-Samuelen
dc.contributor.authorZohn, Irene E.en
dc.contributor.departmentFralin Biomedical Research Instituteen
dc.contributor.departmentBiological Sciencesen
dc.date.accessioned2020-12-23T18:08:33Zen
dc.date.available2020-12-23T18:08:33Zen
dc.date.issued2020-10en
dc.description.abstractBackground Vitamin A regulates patterning of the pharyngeal arches, cranial nerves, and hindbrain that are essential for feeding and swallowing. In the LgDel mouse model of 22q11.2 deletion syndrome (22q11DS), morphogenesis of multiple structures involved in feeding and swallowing are dysmorphic. We asked whether changes in maternal dietary Vitamin A intake can modify cranial nerve, hindbrain and pharyngeal arch artery development in the embryo as well as lung pathology that can be a sign of aspiration dysphagia in LgDel pups. Methods Three defined amounts of vitamin A (4, 10, and 16 IU/g) were provided in the maternal diet. Cranial nerve, hindbrain and pharyngeal arch artery development was evaluated in embryos and inflammation in the lungs of pups to determine the impact of altering maternal diet on these phenotypes. Results Reduced maternal vitamin A intake improved whereas increased intake exacerbated lung inflammation in LgDel pups. These changes were accompanied by increased incidence and/or severity of pharyngeal arch artery and cranial nerve V (CN V) abnormalities in LgDel embryos as well as altered expression of Cyp26b1 in the hindbrain. Conclusions Our studies demonstrate that variations in maternal vitamin A intake can influence the incidence and severity of phenotypes in a mouse model 22q11.2 deletion syndrome.en
dc.description.notesEunice Kennedy Shriver National Institute of Child Health and Human Development, Grant/Award Numbers: P01 HD083157, R21 HD090623, U54 HD090257en
dc.description.sponsorshipEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentUnited States Department of Health & Human ServicesNational Institutes of Health (NIH) - USANIH Eunice Kennedy Shriver National Institute of Child Health & Human Development (NICHD) [P01 HD083157, R21 HD090623, U54 HD090257]en
dc.format.mimetypeapplication/pdfen
dc.identifier.doihttps://doi.org/10.1002/bdr2.1709en
dc.identifier.issn2472-1727en
dc.identifier.issue16en
dc.identifier.pmid32431076en
dc.identifier.urihttp://hdl.handle.net/10919/101620en
dc.identifier.volume112en
dc.language.isoenen
dc.rightsCreative Commons Attribution 4.0 Internationalen
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/en
dc.subject22q11en
dc.subject2 deletion syndromeen
dc.subjectRetinoid aciden
dc.subjectvitamin Aen
dc.subjectDysphagiaen
dc.subjectGene-environment interactionen
dc.titleVariations in maternal vitamin A intake modifies phenotypes in a mouse model of 22q11.2 deletion syndromeen
dc.title.serialBirth Defects Researchen
dc.typeArticle - Refereeden
dc.type.dcmitypeTexten
dc.type.dcmitypeStillImageen

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