PUBLICATION

Early steps in primary cilium assembly require EHD1/EHD3-dependent ciliary vesicle formation

Authors
Lu, Q., Insinna, C., Ott, C., Stauffer, J., Pintado, P.A., Rahajeng, J., Baxa, U., Walia, V., Cuenca, A., Hwang, Y.S., Daar, I.O., Lopes, S., Lippincott-Schwartz, J., Jackson, P.K., Caplan, S., Westlake, C.J.
ID
ZDB-PUB-150429-18
Date
2015
Source
Nature cell biology   17: 228-40 (Journal)
Registered Authors
Lopes, Susana
Keywords
none
MeSH Terms
  • Animals
  • Axoneme/metabolism
  • Axoneme/ultrastructure
  • Carrier Proteins/antagonists & inhibitors
  • Carrier Proteins/genetics*
  • Carrier Proteins/metabolism
  • Cell Line
  • Centrioles/metabolism
  • Centrioles/ultrastructure
  • Cilia/metabolism*
  • Cilia/ultrastructure
  • Embryo, Nonmammalian
  • Epithelial Cells/metabolism*
  • Epithelial Cells/ultrastructure
  • Gene Expression Regulation, Developmental*
  • Humans
  • Kidney Tubules, Collecting/metabolism
  • Kidney Tubules, Collecting/ultrastructure
  • Mice
  • Morphogenesis/genetics
  • Qb-SNARE Proteins/genetics
  • Qb-SNARE Proteins/metabolism
  • Qc-SNARE Proteins/genetics
  • Qc-SNARE Proteins/metabolism
  • RNA, Small Interfering/genetics
  • RNA, Small Interfering/metabolism
  • Retinal Pigment Epithelium/metabolism
  • Retinal Pigment Epithelium/ultrastructure
  • Signal Transduction
  • Transport Vesicles/metabolism*
  • Transport Vesicles/ultrastructure
  • Vesicular Transport Proteins/antagonists & inhibitors
  • Vesicular Transport Proteins/genetics*
  • Vesicular Transport Proteins/metabolism
  • Zebrafish
  • rab GTP-Binding Proteins/genetics
  • rab GTP-Binding Proteins/metabolism
PubMed
25686250 Full text @ Nat. Cell Biol.
Abstract
Membrane association with mother centriole (M-centriole) distal appendages is critical for ciliogenesis initiation. How the Rab GTPase Rab11-Rab8 cascade functions in early ciliary membrane assembly is unknown. Here, we show that the membrane shaping proteins EHD1 and EHD3, in association with the Rab11-Rab8 cascade, function in early ciliogenesis. EHD1 and EHD3 localize to preciliary membranes and the ciliary pocket. EHD-dependent membrane tubulation is essential for ciliary vesicle formation from smaller distal appendage vesicles (DAVs). Importantly, this step functions in M-centriole to basal body transformation and recruitment of transition zone proteins and IFT20. SNAP29, a SNARE membrane fusion regulator and EHD1-binding protein, is also required for DAV-mediated ciliary vesicle assembly. Interestingly, only after ciliary vesicle assembly is Rab8 activated for ciliary growth. Our studies uncover molecular mechanisms informing a previously uncharacterized ciliogenesis step, whereby EHD1 and EHD3 reorganize the M-centriole and associated DAVs before coordinated ciliary membrane and axoneme growth.
Errata / Notes
This article is corrected by ZDB-PUB-220906-14.
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