ARL13B regulates Sonic hedgehog signaling from outside primary cilia.

ABSTRACT: ARL13B is a regulatory GTPase highly enriched in cilia. Complete loss of Arl13b disrupts cilia architecture, protein trafficking and Sonic hedgehog signaling. To determine whether ARL13B is required within cilia, we knocked in a cilia-excluded variant of ARL13B (V358A) and showed it retains all known biochemical function. We found that ARL13B^V358A protein was expressed but could not be detected in cilia, even when retrograde ciliary transport was blocked. We showed Arl13b^V358A/V358A mice are viable and fertile with normal Shh signal transduction. However, in contrast to wild type cilia, Arl13b^V358A/V358A cells displayed short cilia and lacked ciliary ARL3 and INPP5E. These data indicate that ARL13B's role within cilia can be uncoupled f

SUBMITTER: Gigante ED 

PROVIDER: S-EPMC7075693 | biostudies-literature | 2020 Mar

REPOSITORIES: biostudies-literature