{"data":{"id":"10.25384/sage.c.4125929.v1","type":"dois","attributes":{"doi":"10.25384/sage.c.4125929.v1","prefix":"10.25384","suffix":"sage.c.4125929.v1","identifiers":[],"alternateIdentifiers":[],"creators":[{"name":"J. Zheng","affiliation":[],"nameIdentifiers":[]},{"name":"X. Nie","affiliation":[],"nameIdentifiers":[]},{"name":"L. He","affiliation":[],"nameIdentifiers":[]},{"name":"A. J. Yoon","affiliation":[],"nameIdentifiers":[]},{"name":"L. Wu","affiliation":[],"nameIdentifiers":[]},{"name":"X. Zhang","affiliation":[],"nameIdentifiers":[]},{"name":"M. Vats","affiliation":[],"nameIdentifiers":[]},{"name":"M.D. Schiff","affiliation":[],"nameIdentifiers":[]},{"name":"L. Xiang","affiliation":[],"nameIdentifiers":[]},{"name":"Z. Tian","affiliation":[],"nameIdentifiers":[]},{"name":"J. Ling","affiliation":[],"nameIdentifiers":[]},{"name":"J.J. Mao","affiliation":[],"nameIdentifiers":[]}],"titles":[{"title":"Epithelial \u003ci\u003eCdc42\u003c/i\u003e Deletion Induced Enamel Organ Defects and Cystogenesis"}],"publisher":"Figshare","container":{},"publicationYear":2018,"subjects":[{"subject":"110599 Dentistry not elsewhere classified","subjectScheme":"FOR"},{"subject":"FOS: Clinical medicine","schemeUri":"http://www.oecd.org/science/inno/38235147.pdf","subjectScheme":"Fields of Science and Technology (FOS)"},{"subject":"91299 Materials Engineering not elsewhere classified","subjectScheme":"FOR"},{"subject":"FOS: Materials engineering","schemeUri":"http://www.oecd.org/science/inno/38235147.pdf","subjectScheme":"Fields of Science and Technology (FOS)"}],"contributors":[],"dates":[{"date":"2018-06-07","dateType":"Created"},{"date":"2018-06-07","dateType":"Updated"},{"date":"2018","dateType":"Issued"}],"language":null,"types":{"ris":"GEN","bibtex":"misc","citeproc":"article","schemaOrg":"Collection","resourceType":"Collection","resourceTypeGeneral":"Collection"},"relatedIdentifiers":[{"relationType":"IsSupplementTo","relatedIdentifier":"10.1177/0022034518779546","relatedIdentifierType":"DOI"},{"relationType":"IsIdenticalTo","relatedIdentifier":"10.25384/sage.c.4125929","relatedIdentifierType":"DOI"}],"relatedItems":[],"sizes":[],"formats":[],"version":null,"rightsList":[{"rights":"CC BY 4.0","rightsUri":"https://creativecommons.org/licenses/by/4.0"}],"descriptions":[{"description":"Cdc42, a Rho family small GTPase, regulates cytoskeleton organization, vesicle trafficking, and other cellular processes in development and homeostasis. However, Cdc42’s roles in prenatal tooth development remain elusive. Here, we investigated Cdc42 functions in mouse enamel organ. Cdc42 showed highly dynamic temporospatial patterns in the developing enamel organ, with robust expression in the outer enamel epithelium, stellate reticulum (SR), and stratum intermedium layers. Strikingly, epithelium-specific \u003ci\u003eCdc42\u003c/i\u003e deletion resulted in cystic lesions in the enamel organ. Cystic lesions were first noted at embryonic day 15.5 and progressively enlarged during gestation. At birth, cystic lesions occupied the bulk of the entire enamel organ, with intracystic erythrocyte accumulation. Ameloblast differentiation was retarded upon epithelial \u003ci\u003eCdc42\u003c/i\u003e deletion. Apoptosis occurred in the \u003ci\u003eCdc42\u003c/i\u003e mutant enamel organ prior to and synchronously with cystogenesis. Transmission electron microscopy examination showed disrupted actin assemblies, aberrant desmosomes, and significantly fewer cell junctions in the SR cells of \u003ci\u003eCdc42\u003c/i\u003e mutants than littermate controls. Autophagosomes were present in the SR cells of \u003ci\u003eCdc42\u003c/i\u003e mutants relative to the virtual absence of autophagosome in the SR cells of littermate controls. Epithelium-specific \u003ci\u003eCdc42\u003c/i\u003e deletion attenuated Wnt/β-catenin and Shh signaling in dental epithelium and induced aberrant Sox2 expression in the secondary enamel knot. These findings suggest that excessive cell death and disrupted cell-cell connections may be among multiple factors responsible for the observed cystic lesions in \u003ci\u003eCdc42\u003c/i\u003e mutant enamel organs. Taken together, \u003ci\u003eCdc42\u003c/i\u003e exerts multidimensional and pivotal roles in enamel organ development and is particularly required for cell survival and tooth morphogenesis.","descriptionType":"Abstract"}],"geoLocations":[],"fundingReferences":[],"xml":"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