Distinct impacts of Eda and Edar loss of function on the mouse dentition

Cyril Charles, Sophie Pantalacci, Paul Tafforeau, Denis Headon, Vincent Laudet, Laurent Viriot

Research output: Contribution to journalArticlepeer-review

Abstract

BACKGROUND: The Eda-A1-Edar signaling pathway is involved in the development of organs with an ectodermal origin, including teeth. In mouse, mutants are known for both the ligand, Eda-A1 (Tabby), and the receptor, Edar (Downless). The adult dentitions of these two mutants have classically been considered to be similar. However, previous studies mentioned differences in embryonic dental development between Eda(Ta) and Edar(dl-J) mutants. A detailed study of tooth morphology in mutants bearing losses of functions of these two genes thus appears necessary to test the pattern variability induced by the developmental modifications. METHODOLOGY/PRINCIPAL FINDINGS: 3D-reconstructions of the cheek teeth have been performed at the ESRF (Grenoble, France) by X-ray synchrotron microtomography to assess dental morphology. The morphological variability observed in Eda(Ta) and Edar(dl-J) mutants have then been compared in detail. Despite patchy similarities, our detailed work on cheek teeth in Eda(Ta) and Edar(dl-J) mice show that all dental morphotypes defined in Edar(dl-J) mice resolutely differ from those of Eda(Ta) mice. This study reveals that losses of function of Eda and Edar have distinct impacts on the tooth size and morphology, contrary to what has previously been thought. CONCLUSION/SIGNIFIANCE: The results indicate that unknown mechanisms of the Eda pathway are implicated in tooth morphogenesis. Three hypotheses could explain our results; an unexpected role of the Xedar pathway (which is influenced by the Eda gene product but not that of Edar), a more complex connection than has been appreciated between Edar and another protein, or a ligand-independent activity for Edar. Further work is necessary to test these hypotheses and improve our understanding of the mechanisms of development.
Original languageEnglish
Pages (from-to)e4985
JournalPLoS ONE
Volume4
Issue number4
DOIs
Publication statusPublished - 2009

Keywords

  • Animals
  • Base Sequence
  • DNA Primers
  • Ectodysplasins
  • Edar Receptor
  • Heterozygote
  • Homozygote
  • Mice
  • Mutation
  • Tomography, X-Ray Computed
  • Tooth

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