The hedgehog co-receptor BOC differentially regulates SHH signaling during craniofacial development.

ABSTRACT: The Hedgehog (HH) pathway controls multiple aspects of craniofacial development. HH ligands signal through the canonical receptor PTCH1, and three co-receptors: GAS1, CDON and BOC. Together, these co-receptors are required during embryogenesis to mediate proper HH signaling. Here, we investigated the individual and combined contributions of GAS1, CDON and BOC to HH-dependent mammalian craniofacial development. Notably, individual deletion of either Gas1 or Cdon results in variable holoprosencephaly phenotypes in mice, even on a congenic background. In contrast, we find that Boc deletion results in facial widening that correlates with increased HH target gene expression. In addition, Boc deletion in a Gas1 null background partially ameliorates the craniofa

SUBMITTER: Echevarria-Andino ML 

PROVIDER: S-EPMC7758635 | biostudies-literature | 2020 Dec

REPOSITORIES: biostudies-literature