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There is renewed research interest in primary cilia due to their essential roles in development, tissue homeostasis, and human diseases. Primary cilia dysfunction causes a large spectrum of human diseases, collectively known as ciliopathies. Despite significant advances in our understanding of primary cilia, there are still no effective agents for treating ciliopathies. Primary ciliogenesis is a highly ordered process involving membrane trafficking, basal body maturation, vesicle docking and fusion, transition zone assembly, and axoneme extension, in which actin and microtubule networks play critical and multiple roles. Actin and microtubule network architecture, isotropy, and dynamics are tightly controlled by cytoskeleton\u2010associated proteins, a growing number of which are now recognized as responsible for cilium formation and maintenance. Here we summarize the roles of actin and microtubules and their associated proteins in primary ciliogenesis and maintenance. In doing so, we highlight that targeting cytoskeleton\u2010associated proteins may be a promising therapeutic strategy for the treatment of ciliopathies.<\/jats:p>","DOI":"10.1002\/jcp.30865","type":"journal-article","created":{"date-parts":[[2022,8,24]],"date-time":"2022-08-24T07:35:12Z","timestamp":1661326512000},"page":"3975-3983","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["Cytoskeletal networks in primary cilia: Current knowledge and perspectives"],"prefix":"10.1002","volume":"237","author":[{"given":"Ruixin","family":"Ge","sequence":"first","affiliation":[{"name":"Department of Cell Biology, College of Life Sciences, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, Institute of Biomedical Sciences Shandong Normal University Jinan China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Minghui","family":"Cao","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, College of Life Sciences, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, Institute of Biomedical Sciences Shandong Normal University Jinan China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miao","family":"Chen","sequence":"additional","affiliation":[{"name":"Department of Bioscience, School of Life Sciences and Medicine Shandong University of Technology Zibo China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1626-7100","authenticated-orcid":false,"given":"Min","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, College of Life Sciences, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, Institute of Biomedical Sciences Shandong Normal University Jinan China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Songbo","family":"Xie","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, College of Life Sciences, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, Institute of Biomedical Sciences Shandong Normal University Jinan China"},{"name":"Department of Bioscience, School of Life Sciences and Medicine Shandong University of Technology Zibo China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,8,24]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"crossref","unstructured":"Afanasyeva T. 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