Journal article
Sox2Controls Periderm and Rugae Development to Inhibit Oral Adhesions
Journal of dental research, Vol.99(12), pp.1397-1405
11/01/2020
DOI: 10.1177/0022034520939013
PMCID: PMC7580171
PMID: 32674684
Abstract
In humans, ankyloglossia and cleft palate are common congenital craniofacial anomalies, and these are regulated by a complex gene regulatory network. Understanding the genetic underpinnings of ankyloglossia and cleft palate will be an important step toward rational treatment of these complex anomalies. We inactivated the Sry (sex-determining region Y)-box 2 (Sox2) gene in the developing oral epithelium, including the periderm, a transient structure that prevents abnormal oral adhesions during development. This resulted in ankyloglossia and cleft palate with 100% penetrance in embryos examined after embryonic day 14.5. InSox2conditional knockout embryos, the oral epithelium failed to differentiate, as demonstrated by the lack of keratin 6, a marker of the periderm. Further examination revealed that the adhesion of the tongue and mandible expressed the epithelial markersE-CadandP63. The expanded epithelia are Sox9-, Pitx2-, and Tbx1-positive cells, which are markers of the dental epithelium; thus, the dental epithelium contributes to the development of oral adhesions. Furthermore, we found thatSox2is required for palatal shelf extension, as well as for the formation of palatal rugae, which are signaling centers that regulate palatogenesis. In conclusion, the deletion ofSox2in oral epithelium disrupts palatal shelf extension, palatal rugae formation, tooth development, and periderm formation. The periderm is required to inhibit oral adhesions and ankyloglossia, which is regulated bySox2. In addition, oral adhesions occur through an expanded dental epithelial layer that inhibits epithelial invagination and incisor development. This process may contribute to dental anomalies due to ankyloglossia.
Details
- Title: Subtitle
- Sox2Controls Periderm and Rugae Development to Inhibit Oral Adhesions
- Creators
- Y. Y. Sweat - University of IowaM. Sweat - University of IowaW. Yu - University of IowaM. Sanz-Navarro - University of HelsinkiL. Zhang - Binzhou UniversityZ. Sun - University of IowaS. Eliason - University of IowaO. D. Klein - University of California, San FranciscoF. Michon - University of HelsinkiZ. Chen - Wuhan UniversityB. A. Amendt - University of Iowa
- Resource Type
- Journal article
- Publication Details
- Journal of dental research, Vol.99(12), pp.1397-1405
- DOI
- 10.1177/0022034520939013
- PMID
- 32674684
- PMCID
- PMC7580171
- NLM abbreviation
- J Dent Res
- ISSN
- 0022-0345
- eISSN
- 1544-0591
- Publisher
- Sage
- Number of pages
- 9
- Grant note
- College of Dentistry, University of Iowa Carver College of Medicine, University of Iowa R43DE027569; R01DE026433; R03EB025873; R21DE025328; 5T90DE023520-07 / National Institutes of Health; United States Department of Health & Human Services; National Institutes of Health (NIH) - USA
- Language
- English
- Date published
- 11/01/2020
- Academic Unit
- Orthodontics; Anatomy and Cell Biology; Craniofacial Anomalies Research Center; Dental Research; Internal Medicine
- Record Identifier
- 9984284331102771
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