Downregulation of FGF Signaling by Spry4 Overexpression Leads to Shape Impairment, Enamel Irregularities, and Delayed Signaling Center Formation in the Mouse Molar.
Downregulation of FGF Signaling by Spry4 Overexpression Leads to Shape Impairment, Enamel Irregularities, and Delayed Signaling Center Formation in the Mouse Molar.
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Spry4 过表达下调 FGF 信号传导导致小鼠磨牙形状损伤、牙釉质不规则和信号传导中心形成延迟。
作者：
Marangoni,Pauline;Charles,Cyril;Ahn,Youngwook;Seidel,Kerstin;Jheon,Andrew;Ganss,Bernhard;Krumlauf,Robb;Viriot,Laurent;Klein,OphirD
FGF信号传导在牙齿发育中起着关键作用，并且该途径的调节剂的突变产生许多引人注目的表型。然而，FGF通路在调节牙列形态特征和矿物质质量中的作用的许多方面仍然未知。在这里，我们使用了在上皮角蛋白14启动子（K14Spry4）下过表达FGF负反馈调节因子Sputy4的转基因小鼠，以实现上皮中信号转导的下调。这导致高度渗透性缺陷，影响牙尖形态和釉质层。我们描述了萌出臼齿的表型，确定了K14 Spry 4转基因胚胎的发育延迟，并将其与牙齿发育序列的变化联系起来。这些数据进一步阐明了FGF信号在牙列发育中的作用，并暗示了牙齿矿化的调节途径。© 2019 The Authors.JBMR Plus由Wiley Periodicals，Inc.出版。美国骨与矿物质研究协会（American Society for Bone and Mineral Research）
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 (K14Spry4) 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 K14Spry4transgenic 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 Plusis published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.