Roles for CEP170 in cilia function and dynein-2 assembly

Journal article

Primary cilia are essential eukaryotic organelles required for signalling and secretion. Dynein-2 is a microtubule-motor protein complex and is required for ciliogenesis via its role in facilitating retrograde intraflagellar transport (IFT) from the cilia tip to the cell body. Dynein-2 must be assembled and loaded onto IFT trains for entry into cilia for this process to occur, but how dynein-2 is assembled and how it is recycled back into a cilium remain poorly understood. Here, we identify centrosomal protein of 170 kDa (CEP170) as a dynein-2-interacting protein in mammalian cells. We show that loss of CEP170 perturbs intraflagellar transport and hedgehog signalling, and alters the stability of dynein-2 holoenzyme complex. Together, our data indicate a role for CEP170 in supporting cilia function and dynein-2 assembly.

Authors: Weijman, JF; Vuolo, L; Shak, C; Pugnetti, A; Mukhopadhyay, AG

• Publication status: Published
• Peer review status: Peer reviewed
• Publisher: The Company of Biologists
• Journal: Journal of Cell Science
• Volume: 137
• Issue: 8
• Article number: jcs261816
• Publication date: 2024-05-01
• Acceptance date: 2024-03-19
• DOI: 10.1242/jcs.261816
• EISSN: 1477-9137
• ISSN: 0021-9533
• Language: English
• Keywords: CEP170; Centrosome; Cilia; Intraflagellar transport; Microtubule motors; Dynein-2