FAM20C plays an essential role in the formation of murine teeth

J Biol Chem. 2012 Oct 19;287(43):35934-42. doi: 10.1074/jbc.M112.386862. Epub 2012 Aug 30.

Abstract

FAM20C is highly expressed in bone and tooth. Previously, we showed that Fam20C conditional knock-out (KO) mice manifest hypophosphatemic rickets, which highlights the crucial roles of this molecule in promoting bone formation and mediating phosphate homeostasis. In this study, we characterized the dentin, enamel, and cementum of Sox2-Cre-mediated Fam20C KO mice. The KO mice exhibited small malformed teeth, severe enamel defects, very thin dentin, less cementum than normal, and overall hypomineralization in the dental mineralized tissues. In situ hybridization and immunohistochemistry analyses revealed remarkable down-regulation of dentin matrix protein 1 (DMP1) and dentin sialophosphoprotein in odontoblasts, along with a sharply reduced expression of ameloblastin and amelotin in ameloblasts. Collectively, these data indicate that FAM20C is essential to the differentiation and mineralization of dental tissues through the regulation of molecules critical to the differentiation of tooth-formative cells.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Animals
  • Calcium-Binding Proteins / genetics
  • Calcium-Binding Proteins / metabolism*
  • Cell Differentiation / physiology
  • Dental Enamel Proteins / biosynthesis
  • Dental Enamel Proteins / genetics
  • Extracellular Matrix Proteins / biosynthesis
  • Extracellular Matrix Proteins / genetics
  • Extracellular Matrix Proteins / metabolism*
  • Gene Expression Regulation, Developmental / physiology
  • Homeostasis / physiology
  • Mice
  • Mice, Knockout
  • Odontoblasts / cytology
  • Odontoblasts / metabolism*
  • Osteogenesis / physiology
  • Phosphates / metabolism
  • Phosphoproteins / biosynthesis
  • Phosphoproteins / genetics
  • Sialoglycoproteins / biosynthesis
  • Sialoglycoproteins / genetics
  • Tooth / cytology
  • Tooth / embryology*
  • Tooth Calcification / physiology*

Substances

  • Ambn protein, mouse
  • Calcium-Binding Proteins
  • Dental Enamel Proteins
  • Dmp1 protein, mouse
  • Extracellular Matrix Proteins
  • FAM20C protein, mouse
  • Phosphates
  • Phosphoproteins
  • Sialoglycoproteins
  • dentin sialophosphoprotein