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However, the mechanisms that regulate dynein-2 motility remain poorly understood. Here, we identify the Caenorhabditis elegans WDR60 homologue, WDR-60, and dissect the roles of this intermediate chain using genome editing and live imaging of endogenous dynein-2\/IFT components. We find that loss of WDR-60 impairs dynein-2 recruitment to cilia and its incorporation onto anterograde IFT trains, reducing retrograde motor availability at the ciliary tip. Consistent with this, we show that fewer dynein-2 motors power WDR-60\u2013deficient retrograde IFT trains, which move at reduced velocities and fail to exit cilia, accumulating on the distal side of the transition zone. Remarkably, disrupting the transition zone\u2019s NPHP module almost fully restores ciliary exit of underpowered retrograde trains in wdr-60 mutants. This work establishes WDR-60 as a major contributor to IFT, and the NPHP module as a roadblock to dynein-2 passage through the transition zone.<\/jats:p>","DOI":"10.1083\/jcb.202010178","type":"journal-article","created":{"date-parts":[[2021,10,14]],"date-time":"2021-10-14T02:22:15Z","timestamp":1634178135000},"update-policy":"https:\/\/doi.org\/10.1083\/jcb.crossmarkpolicy","source":"Crossref","is-referenced-by-count":36,"title":["WDR60-mediated dynein-2 loading into cilia powers retrograde IFT and transition zone crossing"],"prefix":"10.1083","volume":"221","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5471-8590","authenticated-orcid":true,"given":"Ana R.G.","family":"De-Castro","sequence":"first","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1203-8447","authenticated-orcid":true,"given":"Diogo R.M.","family":"Rodrigues","sequence":"additional","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"},{"name":"Instituto de Ci\u00eancias Biom\u00e9dicas Abel Salazar (ICBAS), Universidade do Porto, Porto, Portugal 3"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3920-6893","authenticated-orcid":true,"given":"Maria J.G.","family":"De-Castro","sequence":"additional","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7166-4336","authenticated-orcid":true,"given":"Neide","family":"Vieira","sequence":"additional","affiliation":[{"name":"Life and Health Sciences Research Institute (ICVS), School of Medicine, University of Minho, Campus Gualtar, Braga, Portugal 4"},{"name":"ICVS\/3B\u2019s - PT Government Associate Laboratory, Braga\/Guimar\u00e3es, Portugal 5"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1560-5937","authenticated-orcid":true,"given":"C\u00e1rmen","family":"Vieira","sequence":"additional","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2256-7134","authenticated-orcid":true,"given":"Ana X.","family":"Carvalho","sequence":"additional","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0360-2977","authenticated-orcid":true,"given":"Reto","family":"Gassmann","sequence":"additional","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8070-8388","authenticated-orcid":true,"given":"Carla M.C.","family":"Abreu","sequence":"additional","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0120-4895","authenticated-orcid":true,"given":"Tiago J.","family":"Dantas","sequence":"additional","affiliation":[{"name":"i3S - Instituto de Investiga\u00e7\u00e3o e Inova\u00e7\u00e3o em Sa\u00fade, Universidade do Porto, Porto, Portugal 1"},{"name":"IBMC - Instituto de Biologia Molecular e Celular, Universidade do Porto, Porto, Portugal 2"}]}],"member":"291","published-online":{"date-parts":[[2021,11,5]]},"reference":[{"key":"2023072717474077900_bib1","doi-asserted-by":"publisher","first-page":"95","DOI":"10.1111\/boc.201800042","article-title":"Cell type-specific structural plasticity of the ciliary transition zone in C. elegans","volume":"111","author":"Akella","year":"2019","journal-title":"Biol. 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