Language
English
Publication Date
10-1-2024
Journal
Development
DOI
10.1242/dev.202705
PMID
39369306
PMCID
PMC11463954
PubMedCentral® Posted Date
10-4-2024
PubMedCentral® Full Text Version
Post-print
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.
Keywords
Animals, Hedgehog Proteins, Receptors, G-Protein-Coupled, Neural Tube, Signal Transduction, Mice, Cilia, Gene Expression Regulation, Developmental, beta-Arrestin 2, Epistasis, Genetic, Female, Cytoskeletal Proteins, Intracellular Signaling Peptides and Proteins, Sonic hedgehog signaling, Fuz, Gpr161, Primary cilia, Mouse neural tube development
Published Open-Access
yes
Recommended Citation
Kim, Sung-Eun; Kim, Hyun-Yi; Wlodarczyk, Bogdan J; et al., "Linkage Between Fuz and Gpr161 Genes Regulates Sonic Hedgehog Signaling During Mouse Neural Tube Development" (2024). Faculty and Staff Publications. 4863.
https://digitalcommons.library.tmc.edu/baylor_docs/4863
Included in
Health Services Research Commons, Medical Cell Biology Commons, Medical Molecular Biology Commons, Medical Specialties Commons, Microbiology Commons