Abstract
Sensory and signaling pathways are exquisitely organized in primary cilia. Bardet-Biedl syndrome (BBS) patients have compromised cilia and signaling. BBS proteins form the BBSome, which binds Rabin8, a guanine nucleotide exchange factor (GEF) activating the Rab8 GTPase, required for ciliary assembly. We now describe serum-regulated upstream vesicular transport events leading to centrosomal Rab8 activation and ciliary membrane formation. Using live microscopy imaging, we show that upon serum withdrawal Rab8 is observed to assemble the ciliary membrane in ∼100 min. Rab8-dependent ciliary assembly is initiated by the relocalization of Rabin8 to Rab11-positive vesicles that are transported to the centrosome. After ciliogenesis, Rab8 ciliary transport is strongly reduced, and this reduction appears to be associated with decreased Rabin8 centrosomal accumulation. Rab11-GTP associates with the Rabin8 COOH-terminal region and is required for Rabin8 preciliary membrane trafficking to the centrosome and for ciliogenesis. Using zebrafish as a model organism, we show that Rabin8 and Rab11 are associated with the BBS pathway. Finally, using tandem affinity purification and mass spectrometry, we determined that the transport protein particle (TRAPP) II complex associates with the Rabin8 NH(2)-terminal domain and show that TRAPP II subunits colocalize with centrosomal Rabin8 and are required for Rabin8 preciliary targeting and ciliogenesis.
MeSH Terms
Analysis of Variance
Animals
Bardet-Biedl Syndrome/metabolism,physiopathology
Carrier Proteins/metabolism
Centrosome/metabolism
Cilia/physiology
Fluorescent Antibody Technique
Guanine Nucleotide Exchange Factors/metabolism
Humans
Mass Spectrometry
Membranes/growth & development
Signal Transduction/physiology
Time-Lapse Imaging
Transfection
Two-Hybrid System Techniques
Zebrafish
rab GTP-Binding Proteins/metabolism
Chemicals
Carrier Proteins
Guanine Nucleotide Exchange Factors
rab11 protein
RAB8A protein, human
rab GTP-Binding Proteins
Authors & Affiliations
13 authors, click to expand affiliations / ORCID
Westlake Christopher J
Genentech, South San Francisco, CA 94080, USA.
Baye Lisa M
Nachury Maxence V
Wright Kevin J
Ervin Karen E
Phu Lilian
Chalouni Cecile
Beck John S
Kirkpatrick Donald S
Slusarski Diane C
Sheffield Val C
Scheller Richard H
Jackson Peter K
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