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Publication : ARL13B regulates Sonic hedgehog signaling from outside primary cilia.

First Author  Gigante ED Year  2020
Journal  Elife Volume  9
PubMed ID  32129762 Mgi Jnum  J:287091
Mgi Id  MGI:6415191 Doi  10.7554/eLife.50434
Citation  Gigante ED, et al. (2020) ARL13B regulates Sonic hedgehog signaling from outside primary cilia. Elife 9:e50434
abstractText  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 from its function outside of cilia. Furthermore, these data imply that the cilia defects upon complete absence of ARL13B do not underlie the alterations in Shh transduction, which is unexpected given the requirement of cilia for Shh transduction.
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