Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph^C41*/C41* mice.

ABSTRACT: Mutations of Odontogenesis-Associated Phosphoprotein (ODAPH, OMIM *614829) cause autosomal recessive amelogenesis imperfecta, however, the function of ODAPH during amelogenesis is unknown. Here we characterized normal Odaph expression by in situ hybridization, generated Odaph truncation mice using CRISPR/Cas9 to replace the TGC codon encoding Cys41 into a TGA translation termination codon, and characterized and compared molar and incisor tooth formation in Odaph^+/+, Odaph^+/C41*, and Odaph^C41*/C41* mice. We also searched genomes to determine when Odaph first appeared phylogenetically. We determined that tooth development in Odaph^+/+ and Odaph^+/C41* mice was indistinguishable in all respects, so the condition in mice is inherited in a re

SUBMITTER: Liang T 

PROVIDER: S-EPMC7807025 | biostudies-literature | 2021 Jan

REPOSITORIES: biostudies-literature