ArticleeLife2018
Dynein-2 intermediate chains play crucial but distinct roles in primary cilia formation and function.
Article in eLife, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 37 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
37 citing papers in PubMed.
- In situ structure of the human ciliary transition zone links linker defects to primary ciliary dyskinesia.Science (New York, N.Y.) · 2026Article
- Hepatic manifestations of ciliopathies: genetic causes and clinical update.European journal of pediatrics · 2026Review
- WDR34 Deficiency Disrupts Retrograde Intraflagellar Transport and Induces Unfolded Protein Response-Driven Inflammation and Retinal Degeneration.Investigative ophthalmology & visual science · 2026Article
- Soft matrix promotes ciliogenesis in human retinal pigment epithelial cells.Scientific reports · 2026Article
- Base editing-derived models of human WDR34 and WDR60 disease alleles replicate retrograde intraflagellar transport (IFT) and hedgehog signaling defects.Communications biology · 2026Article
- Radioprotective Efficacy of Jujube Aqueous Extract and Arbutin: Synergistic Antioxidant and Anti-Apoptotic Mechanisms in Radiation Injury.Food science & nutrition · 2025Article
- The Kelch 3 motif on gigaxonin mediates the interaction with NUDCD3 and regulates vimentin filament morphology.European journal of cell biology · 2025Article
- Cilia at the crossroad: convergence of regulatory mechanisms to govern cilia dynamics during cell signaling and the cell cycle.Cell & bioscience · 2025Review
- The genetic landscape of pediatric postural orthostatic tachycardia syndrome.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2025Article
- TCTEX1D2 is essential for sperm flagellum formation in mice.Scientific reports · 2025Article
- Functional heterogeneity of meniscal fibrochondrocytes and microtissue models is dependent on modality of fibrochondrocyte isolation.Cell proliferation · 2025Article
- Virus infection and vesicle trafficking.Frontiers in immunology · 2025Review
- m6Am methyltransferase PCIF1 negatively regulates ciliation by inhibiting BICD2 expression.The Journal of cell biology · 2024Article
- Roles for CEP170 in cilia function and dynein-2 assembly.Journal of cell science · 2024Article
- Structure and tethering mechanism of dynein-2 intermediate chains in intraflagellar transport.The EMBO journal · 2024Article
- Review
- Multi-scale Modelling of Shear Stress on the Syncytiotrophoblast: Could Maternal Blood Flow Impact Placental Function Across Gestation?Annals of biomedical engineering · 2023Article
- Hyperoxia impairs intraflagellar transport and causes dysregulated metabolism with resultant decreased cilia length.American journal of physiology. Lung cellular and molecular physiology · 2023Article
- Disease-associated mutations in WDR34 lead to diverse impacts on the assembly and function of dynein-2.Journal of cell science · 2023Article
- Multiple interactions of the dynein-2 complex with the IFT-B complex are required for effective intraflagellar transport.Journal of cell science · 2023Article
Corrections and comments
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Authors and funding
4 authors.
Funding
Abstract
The dynein-2 microtubule motor is the retrograde motor for intraflagellar transport. Mutations in dynein-2 components cause skeletal ciliopathies, notably Jeune syndrome. Dynein-2 contains a heterodimer of two non-identical intermediate chains, WDR34 and WDR60. Here, we use knockout cell lines to demonstrate that each intermediate chain has a distinct role in cilium function. Using quantitative proteomics, we show that WDR34 KO cells can assemble a dynein-2 motor complex that binds IFT proteins yet fails to extend an axoneme, indicating complex function is stalled. In contrast, WDR60 KO cells do extend axonemes but show reduced assembly of dynein-2 and binding to IFT proteins. Both proteins are required to maintain a functional transition zone and for efficient bidirectional intraflagellar transport. Our results indicate that the subunit asymmetry within the dynein-2 complex is matched with a functional asymmetry between the dynein-2 intermediate chains. Furthermore, this work reveals that loss of function of dynein-2 leads to defects in transition zone architecture, as well as intraflagellar transport.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.