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JBMR Plus. 2019 Jul 31;3(8):e10205. doi: 10.1002/jbm4.10205. eCollection 2019 Aug.

Downregulation of FGF Signaling by Spry4 Overexpression Leads to Shape Impairment, Enamel Irregularities, and Delayed Signaling Center Formation in the Mouse Molar.

Author information

1
Program in Craniofacial Biology and Department of Orofacial Sciences University of California San Francisco CA USA.
2
Institut de Génomique Fonctionnelle de Lyon Univ Lyon, CNRS UMR 5242, ENS de Lyon, Université Claude Bernard Lyon 1 Lyon France.
3
Stowers Institute for Medical Research Kansas City MO USA.
4
Faculty of Dentistry, University of Toronto ON Canada.
5
Department of Anatomy and Cell Biology Kansas University Medical Center Kansas City KS USA.
6
Department of Pediatrics and Institute for Human Genetics University of California San Francisco CA USA.

Abstract

FGF signaling plays a critical role in tooth development, and mutations in modulators of this pathway produce a number of striking phenotypes. However, many aspects of the role of the FGF pathway in regulating the morphological features and the mineral quality of the dentition remain unknown. Here, we used transgenic mice overexpressing the FGF negative feedback regulator Sprouty4 under the epithelial keratin 14 promoter (K14-Spry4) to achieve downregulation of signaling in the epithelium. This led to highly penetrant defects affecting both cusp morphology and the enamel layer. We characterized the phenotype of erupted molars, identified a developmental delay in K14-Spry4 transgenic embryos, and linked this with changes in the tooth developmental sequence. These data further delineate the role of FGF signaling in the development of the dentition and implicate the pathway in the regulation of tooth mineralization. © 2019 The Authors. JBMR Plus is published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

KEYWORDS:

ENAMEL MINERALIZATION DEFECT; FGF SIGNALING; SPRY4; TOOTH DEVELOPMENT

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