Cited by 146dental development and anomaliesDevelopmental Biology and Gene RegulationHair Growth and Disorders

The activation level of the TNF family receptor, Edar, determines cusp number and tooth number during tooth development.

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Tucker, A.S, Headon, D.J, Courtney, J.-M, Overbeek, P, Sharpe, P.T·2004·Developmental Biology

Abstract

Mutations in members of the ectodysplasin (TNF-related) signalling pathway, EDA, EDAR, and EDARADD in mice and humans produce an ectodermal dysplasia phenotype that includes missing teeth and smaller teeth with reduced cusps. Using the keratin 14 promoter to target expression of an activated form of Edar in transgenic mice, we show that expression of this transgene is able to rescue the tooth phenotype in Tabby (Eda) and Sleek (Edar) mutant mice. High levels of expression of the transgene in wild-type mice result in molar teeth with extra cusps, and in some cases supernumerary teeth, the opposite of the mutant phenotype. The level of activation of Edar thus determines cusp number and tooth number during tooth development.

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Research works printed but not linked: 3

  1. Pirinen, S., 1998. Dental and oral aspects in anhidrotic ectodermal dyspla- sia. In: Bergendahl, B., Koch, G., Kurol, J., Wanndahl, G. (Eds.), Con- sensus Conference on Ectodermal Dysplasia with special reference to Dental Treatment. Forlagshuset Gothia AB, Stockholm, pp. 13 -21.
  2. Srivatava, A.K., Pispa, J., Hartung, A.J., Du, Y., Ezer, S., Jenks, T., Shi- mada, T., Pekkanen, M., Mikkola, M.L., Ko, M.S.H., et al., 1997. The tabby phenotype is caused by mutations in a mouse homologue of the EDA gene that reveals novel mouse and human exons and encodes a protein (ectodysplasin-A) with collagenous domains. Proc. Natl. Acad. Sci. U. S. A. 94, 13069 -13074.
  3. Vassar, R., Rosenberg, M., Ross, S., Tyner, A., Fuchs, E., 1989. Tissue- specific and differentiation-specific expression of a human K14 keratin gene in transgenic mice. Proc. Natl. Acad. Sci. U. S. A. 86, 1563 -1567.

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Authors
Tucker, A. S., Headon, D. J., Courtney, J.-M., Overbeek, P., & Sharpe, P. T.
Affiliations
  • Guy's Hospital (GB)
  • University of Manchester (GB)
  • Baylor College of Medicine (US)
Corresponding author
Sharpe, P.T
Venue
Developmental Biology
Publisher
Elsevier BV
Published
2004-04
Volume
268
Issue
1
Pages
185-194
ISSN
0012-1606 (ISSN-L)
OpenAlex
W2096819604
Cited by
146
References
2
Citations by year
2026: 3 · 2024: 2 · 2023: 5 · 2022: 6 · 2021: 3 · 2020: 4 · 2019: 5 · 2018: 6 · 2017: 5 · 2016: 3 · 2015: 3 · 2014: 12 · 2013: 16 · 2012: 6
FWCI
2.85
Type
journal article
Language
en
Open access
Locations
  • Developmental Biology (publishedVersion)
  • PubMed (publishedVersion)
  • eCite Digital Repository (University of Tasmania) (submittedVersion)
  • Figshare (submittedVersion, cc-by)
  • Research Portal (King's College London) (submittedVersion)
  • Research Explorer (The University of Manchester) (submittedVersion)
  • Edinburgh Research Explorer (University of Edinburgh) (submittedVersion, other-oa)
Funders
  • Wellcome Trust
  • Medical Research Council
  • Biotechnology and Biological Sciences Research Council
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › dental development and anomalies
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
  • Health Sciences › Medicine › Urology › Hair Growth and Disorders
Keywords
EDAR, cusp number, number of teeth, tooth development, ectodermal dysplasia, supernumerary teeth, molars, keratin 14 promoter, transgenic rescue
MeSH terms
Animals, Base Sequence, Dental Enamel, Humans, Membrane Proteins, Mice, Transgenic, Tooth, DNA Primers, Receptors, Tumor Necrosis Factor, Mice, Receptors, Ectodysplasin, Edar Receptor

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