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

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.

journal_name

Nat Cell Biol

journal_title

Nature cell biology

authors

Lu Q,Insinna C,Ott C,Stauffer J,Pintado PA,Rahajeng J,Baxa U,Walia V,Cuenca A,Hwang YS,Daar IO,Lopes S,Lippincott-Schwartz J,Jackson PK,Caplan S,Westlake CJ

doi

10.1038/ncb3109

subject

Has Abstract

pub_date

2015-03-01 00:00:00

pages

228-240

issue

3

eissn

1465-7392

issn

1476-4679

journal_volume

17

pub_type

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