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

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 C...

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Autores principales: Liang, Tian, Hu, Yuanyuan, Kawasaki, Kazuhiko, Zhang, Hong, Zhang, Chuhua, Saunders, Thomas L., Simmer, James P., Hu, Jan C.-C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7807025/
https://www.ncbi.nlm.nih.gov/pubmed/33441959
http://dx.doi.org/10.1038/s41598-020-80912-y
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author Liang, Tian
Hu, Yuanyuan
Kawasaki, Kazuhiko
Zhang, Hong
Zhang, Chuhua
Saunders, Thomas L.
Simmer, James P.
Hu, Jan C.-C.
author_facet Liang, Tian
Hu, Yuanyuan
Kawasaki, Kazuhiko
Zhang, Hong
Zhang, Chuhua
Saunders, Thomas L.
Simmer, James P.
Hu, Jan C.-C.
author_sort Liang, Tian
collection PubMed
description 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 recessive pattern, as it is in humans. Odaph is specifically expressed by ameloblasts starting with the onset of post-secretory transition and continues until mid-maturation. Based upon histological and ultrastructural analyses, we determined that the secretory stage of amelogenesis is not affected in Odaph(C41*/C41*) mice. The enamel layer achieves a normal shape and contour, normal thickness, and normal rod decussation. The fundamental problem in Odaph(C41*/C41*) mice starts during post-secretory transition, which fails to generate maturation stage ameloblasts. At the onset of what should be enamel maturation, a cyst forms that separates flattened ameloblasts from the enamel surface. The maturation stage fails completely.
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spelling pubmed-78070252021-01-14 Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph(C41*/C41*) mice Liang, Tian Hu, Yuanyuan Kawasaki, Kazuhiko Zhang, Hong Zhang, Chuhua Saunders, Thomas L. Simmer, James P. Hu, Jan C.-C. Sci Rep Article 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 recessive pattern, as it is in humans. Odaph is specifically expressed by ameloblasts starting with the onset of post-secretory transition and continues until mid-maturation. Based upon histological and ultrastructural analyses, we determined that the secretory stage of amelogenesis is not affected in Odaph(C41*/C41*) mice. The enamel layer achieves a normal shape and contour, normal thickness, and normal rod decussation. The fundamental problem in Odaph(C41*/C41*) mice starts during post-secretory transition, which fails to generate maturation stage ameloblasts. At the onset of what should be enamel maturation, a cyst forms that separates flattened ameloblasts from the enamel surface. The maturation stage fails completely. Nature Publishing Group UK 2021-01-13 /pmc/articles/PMC7807025/ /pubmed/33441959 http://dx.doi.org/10.1038/s41598-020-80912-y Text en © The Author(s) 2021 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.
spellingShingle Article
Liang, Tian
Hu, Yuanyuan
Kawasaki, Kazuhiko
Zhang, Hong
Zhang, Chuhua
Saunders, Thomas L.
Simmer, James P.
Hu, Jan C.-C.
Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph(C41*/C41*) mice
title Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph(C41*/C41*) mice
title_full Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph(C41*/C41*) mice
title_fullStr Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph(C41*/C41*) mice
title_full_unstemmed Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph(C41*/C41*) mice
title_short Odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in Odaph(C41*/C41*) mice
title_sort odontogenesis-associated phosphoprotein truncation blocks ameloblast transition into maturation in odaph(c41*/c41*) mice
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7807025/
https://www.ncbi.nlm.nih.gov/pubmed/33441959
http://dx.doi.org/10.1038/s41598-020-80912-y
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