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作 者:Elizabeth Guirado Cassandra Villani Adrienn Petho Yinghua Chen Mark Maienschein-Cline Zhengdeng Lei Nina Los Anne George
机构地区:[1]Department of Oral Biology,University of Illinois Chicago,Chicago,IL,USA [2]Research Informatics Core,University of Illinois at Chicago,Chicago,IL,USA [3]Bioinformatics Scientist Ⅲ,Ambry Genetics,Aliso,CA,USA [4]Genome Research Core,University of Illinois at Chicago,Chicago,IL,USA
出 处:《International Journal of Oral Science》2023年第1期127-135,共9页国际口腔科学杂志(英文版)
基 金:our funding sources U.S.Department of Health&Human Services,NIH,NIDCR T32 DE018381[Multidisciplinary Oral Science Training Program];DE028193[E.G.];R01 DE031737 and DE 028531[A.G.];the Brodie Endowment Fund。
摘 要:X-linked hypophosphatemia(XLH)represents the most common form of familial hypophosphatemia.Although significant advances have been made in the treatment of bone pathology,patients undergoing therapy continue to experience significantly decreased oral health-related quality of life.The following study addresses this persistent oral disease by further investigating the effect of DMP1 expression on the differentiation of XLH dental pulp cells.Dental pulp cells were isolated from the third molars of XLH and healthy controls and stable transduction of full-length human DMP1 were achieved.RNA sequencing was performed to evaluate the genetic changes following the induction of odontogenic differentiation.RNAseq data shows the upregulation of inhibitors of the canonical Wnt pathway in XLH cells,while constitutive expression of full-length DMP1 in XLH cells reversed this effect during odontogenic differentiation.These results imply that inhibition of the canonical Wnt pathway may contribute to the pathophysiology of XLH and suggest a new therapeutic strategy for the management of oral disease.
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