Linkage between Fuz and Gpr161 genes regulates sonic hedgehog signaling during mouse neural tube development.

IF 3.7 2区生物学 Q1 DEVELOPMENTAL BIOLOGY

Development Pub Date : 2024-10-01 Epub Date: 2024-10-04 DOI:10.1242/dev.202705

Sung-Eun Kim, Hyun-Yi Kim, Bogdan J Wlodarczyk, Richard H Finnell

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引用次数: 0

Abstract

Sonic hedgehog (Shh) signaling regulates embryonic morphogenesis utilizing the primary cilium, the cell's antenna, which acts as a signaling hub. Fuz, an effector of planar cell polarity signaling, regulates Shh signaling by facilitating cilia formation, and the G protein-coupled receptor 161 (Gpr161) is a negative regulator of Shh signaling. The range of phenotypic malformations observed in mice bearing mutations in either of the genes encoding these proteins is similar; however, their functional relationship has not been previously explored. This study identified the genetic and biochemical linkage between Fuz and Gpr161 in mouse neural tube development. Fuz was found to be genetically epistatic to Gpr161 with respect to regulation of Shh signaling in mouse neural tube development. The Fuz protein biochemically interacts with Gpr161, and Fuz regulates Gpr161-mediated ciliary localization, a process that might utilize β-arrestin 2. Our study characterizes a previously unappreciated Gpr161-Fuz axis that regulates Shh signaling during mouse neural tube development.

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Fuz和Gpr161基因之间的联系调控了小鼠神经管发育过程中的声刺猬信号传导。

音速刺猬素（Shh）信号通过作为信号枢纽的初级纤毛（细胞的触角）调节胚胎形态发生。Fuz是平面细胞极性信号转导的效应因子，它通过促进纤毛的形成来调节Shh信号转导，而G蛋白偶联受体161（Gpr161）是Shh信号转导的负调节因子。编码这些蛋白的任一基因发生突变的小鼠所观察到的表型畸形的范围是相似的；然而，以前从未探讨过它们之间的功能关系。本研究确定了 Fuz 和 Gpr161 在小鼠神经管发育过程中的遗传和生化联系。研究发现，在小鼠神经管发育过程中，Fuz 与 Gpr161 在调控 Shh 信号转导方面存在遗传外显关系。Fuz 蛋白与 Gpr161 存在生化相互作用，Fuz 可调控 Gpr161 介导的纤毛定位，这一过程可能利用了 β-arrestin 2。我们的研究描述了以前未被认识的 Gpr161-Fuz 轴在小鼠神经管发育过程中调控 Shh 信号的特性。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Development 生物-发育生物学

CiteScore

6.70

自引率

4.30%

发文量

433

审稿时长

3 months

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