{"doi":"10.1242/jcs.261816","title":"Roles for CEP170 in cilia function and dynein-2 assembly","abstract":"<jats:title>ABSTRACT</jats:title>\n                  <jats:p>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.</jats:p>","journal":"Journal of Cell Science","year":2024,"id":674130,"datarank":0.4335557636844247,"base_score":2.8903717578961645,"endowment":2.8903717578961645,"self_citation_contribution":0.4335557636844247,"citation_network_contribution":0.0,"self_endowment_contribution":0.4335557636844247,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":17,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":null,"is_data_producer":false,"deposit_databanks":null,"is_oa":false,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":null,"fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":1761340,"name":"Laura Vuolo","orcid":"0000-0002-9801-9206","position":1,"is_corresponding":false},{"id":1761341,"name":"Caroline Shak","orcid":"0000-0002-2109-1058","position":2,"is_corresponding":false},{"id":1761342,"name":"Anna Pugnetti","orcid":null,"position":3,"is_corresponding":false},{"id":1761343,"name":"Aakash G. Mukhopadhyay","orcid":"0000-0001-9397-9702","position":4,"is_corresponding":false},{"id":1761344,"name":"Lorna R. Hodgson","orcid":"0000-0001-6373-4405","position":5,"is_corresponding":false},{"id":483205,"name":"Kate J. Heesom","orcid":"0000-0002-5418-5392","position":6,"is_corresponding":false},{"id":632736,"name":"A. J. Roberts","orcid":"0000-0001-5277-6730","position":7,"is_corresponding":false},{"id":174054,"name":"David Stephens","orcid":"0000-0001-5297-3240","position":8,"is_corresponding":false},{"id":1761339,"name":"Johannes F. Weijman","orcid":"0000-0002-2082-5777","position":0,"is_corresponding":false}],"reference_count":0,"raw_metadata":{"has_enrichment":true,"resolved":true,"title":"Roles for CEP170 in cilia function and dynein-2 assembly","abstract":"<jats:title>ABSTRACT</jats:title>\n                  <jats:p>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.</jats:p>","is_dataset_classified":null,"base_score":2.833213344056216,"endowment":2.833213344056216,"datacite_reuse_total":0,"file_count":0,"downloads":0,"views":0,"has_version_chain":false,"is_dataset":false,"is_oa":false,"pmid":"38533689","pmcid":"PMC11112123","openalex_id":"https://openalex.org/W4393232713","authors":[],"funders":[{"funder_name":"Biotechnology and Biological Sciences Research Council","grant_id":"BB/S005390/1","title":"The role of dynein-2 in building a functional cilium."},{"funder_name":"Wellcome Trust","grant_id":"217186/Z/19/Z","title":null},{"funder_name":"Wellcome Trust","grant_id":"217186","title":"Multi-scale mechanisms of microtubule-based transport within cilia and flagella"},{"funder_name":"University of Bristol","grant_id":"","title":null},{"funder_name":"Wellcome Trust","grant_id":"","title":null},{"funder_name":"Wellcome Trust","grant_id":"","title":null}],"total_grants":6,"fwci":6.1537,"citation_percentile":0.96734737,"influential_citations":0,"citation_trend":[{"year":2024,"count":1},{"year":2025,"count":10},{"year":2026,"count":5}],"oa_status":"hybrid","license":"cc-by","oa_locations":[{"url":"https://journals.biologists.com/jcs/article-pdf/doi/10.1242/jcs.261816/3411676/jcs261816.pdf","host_type":"journal"},{"url":"https://journals.biologists.com/jcs/article-pdf/doi/10.1242/jcs.261816/3411676/jcs261816.pdf","host_type":"publisher"},{"url":"https://journals.biologists.com/jcs/article-pdf/doi/10.1242/jcs.261816/3422437/jcs261816.pdf","host_type":"publisher"},{"url":"https://doi.org/10.1242/jcs.261816","host_type":"journal"},{"url":"https://pubmed.ncbi.nlm.nih.gov/38533689","host_type":"repository"},{"url":"https://www.ncbi.nlm.nih.gov/pmc/articles/11112123","host_type":"repository"},{"url":"https://research-information.bris.ac.uk/en/publications/3b00f77f-9a17-49ff-8e8b-2ba0b46bb487","host_type":"repository"},{"url":"https://hdl.handle.net/1983/3b00f77f-9a17-49ff-8e8b-2ba0b46bb487","host_type":"repository"},{"url":"https://europepmc.org/articles/PMC11112123","host_type":"Europe_PMC"},{"url":"https://doi.org/10.1101/2023.11.20.567836","host_type":""},{"url":"http://dx.doi.org/10.1242/jcs.261816","host_type":""},{"url":"https://ora.ox.ac.uk/objects/uuid:9b22ba85-d2df-482b-bd09-c0857452b884","host_type":""},{"url":"https://doi.org/https://doi.org/10.1242/jcs.261816","host_type":""}],"fields_of_study":["Genetic and Kidney Cyst Diseases","Microtubule and mitosis dynamics","Fetal and Pediatric Neurological Disorders","0301 basic medicine","0303 health sciences","03 medical and health sciences","Cilia","Humans","Microtubule-Associated Proteins","Animals","Dyneins","Hedgehog Proteins","Signal Transduction","Mice","Flagella"],"mesh_terms":["Animals","Cilia","Dyneins","Flagella","Humans","Microtubule-Associated Proteins","Signal Transduction","Mice","Hedgehog Proteins"],"keywords":["Biology","Cilium","Dynein","Function (biology)","Intraflagellar transport","Cell biology","Flagellum","Microtubule","Genetics","Gene","Cilia","Centrosome","Microtubule Motors","Cep170","Dynein-2","570","610","Dyneins","Mice","Flagella","Humans","Animals","Hedgehog Proteins","Microtubule-Associated Proteins","Research Article","Signal Transduction"],"sdg_mappings":[],"linked_datasets":[],"clinical_trials":[],"software_tools":[],"database_accessions":[{"name":"hpa"},{"name":"pxd"},{"name":"doi"}],"source":"live","citation_network_status":"fetched"},"created_at":"2026-08-16T15:43:05.339516Z","pmid":null,"pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}