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Updated: Jun 16, 2026

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Volumetric Imaging and Analysis of Primary Cilia in Musculoskeletal Tissue using the ARL13B-CENTRIN-2 Mouse Model
Published on: March 28, 2025
Characterization of membrane structures regulating primary ciliogenesis by quantitative isotropic ultrastructure
Quanlong Lu1, Huijie Zhao1, Ziam Khan1
1Laboratory of Cellular and Developmental Signaling, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Frederick, MD, USA.
Nature Communications
|June 13, 2026
Summary
Vesicle enlargement and fusion at the mother centriole (MC) are crucial for initiating primary cilia assembly. This process involves novel tubular and toroidal membrane intermediates regulated by EHD1 and RAB8, enabling ciliogenesis progression.
Area of Science:
- Cell Biology
- Membrane Trafficking
- Organelle Biogenesis
Background:
- Primary cilia are essential cellular structures involved in various signaling pathways.
- The initial steps of primary cilia assembly, particularly membrane vesicle trafficking to the mother centriole (MC), are not fully understood.
Purpose of the Study:
- To elucidate the three-dimensional (3D) membrane ultrastructures and protein associations involved in early primary cilia assembly.
- To identify key regulators and intermediates in the membrane trafficking pathway upstream of axoneme growth.
Main Methods:
- Isotropic ultrastructural imaging to visualize 3D membrane structures.
- Quantitative 3D analysis to study protein localization and membrane dynamics.
- Investigated the roles of CEP164, EHD1, RAB8, and IFT88 in ciliogenesis.
Main Results:
- Identified vesicle enlargement and fusion at the MC as a trigger for ciliogenesis, requiring CEP164.
- Characterized novel tubular C-shaped and toroidal membrane intermediates during ciliogenesis.
- Demonstrated that EHD1 and RAB8 orchestrate the formation of these intermediates, associated with IFT88.
- Showed EHD1-mediated membrane tubulation promotes CP110/CEP97 removal from the MC cap, facilitating ciliogenesis.
- Observed recruitment of ciliary gate transition zone proteins to these tubular structures.
Conclusions:
- Redefined the architectural framework of early primary cilia assembly.
- Highlighted the importance of vesicle fusion and membrane remodeling at the MC.
- Emphasized the role of EHD1 in regulating ciliogenesis progression via membrane tubulation and protein removal.
- Underscored the utility of advanced imaging and 3D analysis for studying membrane trafficking and organelle biogenesis.
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