ArticleThe Journal of cell biology2018
Proteins that control the geometry of microtubules at the ends of cilia.
Article in The Journal of cell biology, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 37 papers.
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Who cites it
37 citing papers in PubMed.
- Unraveling the molecular details of centriole elongation.Trends open · 2026Article
- Microtubule binding protein Togaram1 is required for proper development of mammalian forebrain and neural primary cilia.Developmental biology · 2026Article
- Structure Makes a Difference: IFT Complex in Ciliary Function and Ciliopathy.Cytoskeleton (Hoboken, N.J.) · 2026Review
- A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function.Nature communications · 2025Article
- Article
- Did axons evolve by activating cytokinesis during interphase? A hypothesis on the origin of neurons.Molecular biology of the cell · 2025Review
- Structure of the ciliary tip central pair reveals the unique role of the microtubule-seam binding protein SPEF1.Current biology : CB · 2025Article
- A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function.bioRxiv : the preprint server for biology · 2025Article
- A network of interacting ciliary tip proteins with opposing activities imparts slow and processive microtubule growth.Nature structural & molecular biology · 2025Article
- Left-right cortical interactions drive intracellular pattern formation in the ciliate Tetrahymena.PLoS genetics · 2025Article
- Axonemal microtubule dynamics in the assembly and disassembly of cilia.Biochemical Society transactions · 2025Review
- Tubulin tyrosination/detyrosination regulate the affinity and sorting of intraflagellar transport trains on axonemal microtubule doublets.Nature communications · 2025Article
- Article
- CEP104/FAP256 and associated cap complex maintain stability of the ciliary tip.The Journal of cell biology · 2023Article
- Cep104 is a component of the centriole distal tip complex that regulates centriole growth and contributes to Drosophila spermiogenesis.Current biology : CB · 2023Article
- A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans.The EMBO journal · 2023Article
- Modulation of inner junction proteins contributes to axoneme differentiation.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- Kif9 is an active kinesin motor required for ciliary beating and proximodistal patterning of motile axonemes.Journal of cell science · 2023Article
- Molecular architecture of the ciliary tip revealed by cryo-electron tomography.bioRxiv : the preprint server for biology · 2023Article
- Clinical and genetic characteristics of 36 children with Joubert syndrome.Frontiers in pediatrics · 2023Article
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Authors and funding
17 authors.
Funding
Abstract
Cilia, essential motile and sensory organelles, have several compartments: the basal body, transition zone, and the middle and distal axoneme segments. The distal segment accommodates key functions, including cilium assembly and sensory activities. While the middle segment contains doublet microtubules (incomplete B-tubules fused to complete A-tubules), the distal segment contains only A-tubule extensions, and its existence requires coordination of microtubule length at the nanometer scale. We show that three conserved proteins, two of which are mutated in the ciliopathy Joubert syndrome, determine the geometry of the distal segment, by controlling the positions of specific microtubule ends. FAP256/CEP104 promotes A-tubule elongation. CHE-12/Crescerin and ARMC9 act as positive and negative regulators of B-tubule length, respectively. We show that defects in the distal segment dimensions are associated with motile and sensory deficiencies of cilia. Our observations suggest that abnormalities in distal segment organization cause a subset of Joubert syndrome cases.
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