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Article Dans Une Revue Molecular Biology of the Cell Année : 2016

Arl13b and the exocyst interact synergistically in ciliogenesis

Cecília Seixas
  • Fonction : Auteur
Soo Young Choi
  • Fonction : Auteur
Noemi Polgar
  • Fonction : Auteur
Nicole Umberger
  • Fonction : Auteur
Michael East
  • Fonction : Auteur
Xiaofeng Zuo
  • Fonction : Auteur
Hugo Moreiras
  • Fonction : Auteur
Alexandre Benmerah
  • Fonction : Auteur
Richard Kahn
  • Fonction : Auteur
Ben Fogelgren
  • Fonction : Auteur
Tamara Caspary
  • Fonction : Auteur
Joshua Lipschutz
  • Fonction : Auteur
Duarte Barral
  • Fonction : Auteur

Résumé

Arl13b belongs to the ADP-ribosylation factor family within the Ras superfamily of regulatory GTPases. Mutations in Arl13b cause Joubert syndrome, which is characterized by congenital cerebellar ataxia, hypotonia, oculomotor apraxia, and mental retardation. Arl13b is highly enriched in cilia and is required for ciliogenesis in multiple organs. Nevertheless, the precise role of Arl13b remains elusive. Here we report that the exocyst subunits Sec8, Exo70, and Sec5 bind preferentially to the GTP-bound form of Arl13b, consistent with the exocyst being an effector of Arl13b. Moreover, we show that Arl13b binds directly to Sec8 and Sec5. In zebrafish, depletion of arl13b or the exocyst subunit sec10 causes phenotypes characteristic of defective cilia, such as curly tail up, edema, and abnormal pronephric kidney development. We explored this further and found a synergistic genetic interaction between arl13b and sec10 morphants in cilia-dependent phenotypes. Through conditional deletion of Arl13b or Sec10 in mice, we found kidney cysts and decreased ciliogenesis in cells surrounding the cysts. Moreover, we observed a decrease in Arl13b expression in the kidneys from Sec10 conditional knockout mice. Taken together, our results indicate that Arl13b and the exocyst function together in the same pathway leading to functional cilia.

Dates et versions

hal-03209200 , version 1 (27-04-2021)

Identifiants

Citer

Cecília Seixas, Soo Young Choi, Noemi Polgar, Nicole Umberger, Michael East, et al.. Arl13b and the exocyst interact synergistically in ciliogenesis. Molecular Biology of the Cell, 2016, 27 (2), pp.308-320. ⟨10.1091/mbc.E15-02-0061⟩. ⟨hal-03209200⟩
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