@article{98f25fd1252a4ea28e20d152f1b560f7,
title = "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.",
author = "Quanlong Lu and Christine Insinna and Carolyn Ott and Jimmy Stauffer and Pintado, {Petra A.} and Juliati Rahajeng and Ulrich Baxa and Vijay Walia and Adrian Cuenca and Hwang, {Yoo Seok} and Daar, {Ira O.} and Susana Lopes and Jennifer Lippincott-Schwartz and Jackson, {Peter K.} and Steve Caplan and Westlake, {Christopher J.}",
note = "Funding Information: We thank K. Nagashima and A. Kamata for help with IEM and EM, C. Lamont for data analysis, S. Lockett for help with SIM imaging, S. Specht for help with cell culture, A. Peden for SNAP29 antibodies, J. Goldenring for Rab11 antibodies, Z. Sun for the transgenic line and S. Burgess for assistance raising zebrafish embryos. We are grateful to J. Donaldson for critical reading of this manuscript. This research was supported by the Intramural Research Program of the National Institutes of Health, National Cancer Institute, Center for Cancer Research. This project has also been funded in part with federal funds from the Frederick National Laboratory for Cancer Research, National Institutes of Health, under contract HHSN261200800001E. Publisher Copyright: {\textcopyright} 2015 Macmillan Publishers Limited.",
year = "2015",
month = mar,
day = "2",
doi = "10.1038/ncb3109",
language = "English (US)",
volume = "17",
pages = "228--240",
journal = "Nature Cell Biology",
issn = "1465-7392",
publisher = "Nature Publishing Group",
number = "3",
}