Sox2 regulates palate development by promoting basal cell differentiation

With a frequency of 1:500–2,500, cleft lip with or without cleft palate (CL/P) is the most common congenital craniofacial defect. In mice and humans, a complex gene regulatory network (GRN) controls the different processes required for palatogenesis. Understanding how transcription factors act as the modulators of gene expression and function in palatogenesis will lead to better genetic counseling and genetic therapies for CL/P. Our group has shown that conditionally inactivating Sox2 , one of the earliest transcription factors required during embryogenesis, results in clefting of the palate and fusion of the tongue to the mandible (ankylglossia). Objective To examine the role of Sox2 , a stem cell factor, in palatogenesis. Methods Using Pitx2 cre , Sox2 was conditionally inactivated in the oral epithelial tissue of developing murine embryos. Palate development was studied during the stages of embryogenesis. Results The conditional ablation of Sox2 was sufficient to generate clefting in mice. Further examination revealed that Sox2 is expressed in the P63 + basal cell population of the oral epithelium as well as the periderm, which acts to prevent oral adhesions during development. Inactivating Sox2 resulted in defective periderm formation. The loss of an intact periderm resulted in intraoral adhesions that prevented complete palate shelf extension and ankylglossia. Conclusions Sox2 is required for periderm differentiation. Inactivating Sox2 results in cleft palate and ankylglossia. Support or Funding Information University of Iowa College of Dentistry NIH, NIDCR This abstract is from the Experimental Biology 2019 Meeting. There is no full text article associated with this abstract published in The FASEB Journal .