Co-ordinated brain and craniofacial development depend upon Patched1/XIAP regulation of cell survival.

ABSTRACT: Congenital brain and craniofacial defects often occur together as a consequence of their developmental dependency on common progenitor tissue interactions and signaling pathways during embryogenesis. A classic example of this is perturbation of midline embryo development, and disruption of Hedgehog (Hh) signaling in the pathogenesis of holoprosencephaly. However, our understanding of how Hh signaling governs cell and tissue survival remains incomplete. Patched1 (Ptch1) is a well-known receptor for Hh ligands and Ptch1 overexpression is associated with cell and tissue-specific apoptosis. Here, we demonstrate that the X-linked inhibitory apoptosis protein (XIAP) associates with the C terminus of Ptch1 (Ptch1-C) in primary cilia to inhibit Ptch1-mediated cell death. Consistent with this obser

SUBMITTER: Aoto K 

PROVIDER: S-EPMC4291248 | biostudies-literature | 2015 Feb

REPOSITORIES: biostudies-literature