ArticleMolecular biology of the cell2014
Cby1 promotes Ahi1 recruitment to a ring-shaped domain at the centriole-cilium interface and facilitates proper cilium formation and function.
Article in Molecular biology of the cell, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 47 papers.
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Who cites it
47 citing papers in PubMed, 62 citations in OpenAlex.
- Neuroimaging-derived brain endophenotypes link molecular mechanisms to Alzheimer's disease and aging.medRxiv : the preprint server for health sciences · 2025Article
- Advances in high-resolution imaging of centrioles and cilia.Journal of cell science · 2025Review
- PathogeniciScience · 2025Article
- ciBAR1 loss in mice causes laterality defects, pancreatic degeneration, and altered glucose tolerance.Life science alliance · 2025Article
- The Cby3/ciBAR1 complex positions the annulus along the sperm flagellum during spermiogenesis.The Journal of cell biology · 2024Article
- Single-molecule imaging in the primary cilium.Methods in cell biology · 2023Article
- Composition, organization and mechanisms of the transition zone, a gate for the cilium.EMBO reports · 2022Review
- Article
- DZIP1 regulates mammalian cardiac valve development through a Cby1-β-catenin mechanism.Developmental dynamics : an official publication of the American Association of Anatomists · 2021Article
- Review
- Loss of the ciliary protein Chibby1 in mice leads to exocrine pancreatic degeneration and pancreatitis.Scientific reports · 2021Article
- The Transition Zone Protein AHI1 Regulates Neuronal Ciliary Trafficking of MCHR1 and Its Downstream Signaling Pathway.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2021Article
- STORM imaging reveals the spatial arrangement of transition zone components and IFT particles at the ciliary base in Tetrahymena.Scientific reports · 2021Article
- Superresolution characterization of core centriole architecture.The Journal of cell biology · 2021Article
- Motile cilia genetics and cell biology: big results from little mice.Cellular and molecular life sciences : CMLS · 2021Review
- Loss of CBY1 results in a ciliopathy characterized by features of Joubert syndrome.Human mutation · 2020Article
- Transient Primary Cilia Mediate Robust Hedgehog Pathway-Dependent Cell Cycle Control.Current biology : CB · 2020Article
- Mechanism of Methylprednisolone-Induced Primary Cilia Formation Disorder and Autophagy in Osteoblasts.Orthopaedic surgery · 2020Article
- Article
- Ahi1 promotes Arl13b ciliary recruitment, regulates Arl13b stability and is required for normal cell migration.Journal of cell science · 2019Article
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Authors and funding
10 authors at 3 institutions in 1 country.
Funding
Abstract
Defects in centrosome and cilium function are associated with phenotypically related syndromes called ciliopathies. Cby1, the mammalian orthologue of the Drosophila Chibby protein, localizes to mature centrioles, is important for ciliogenesis in multiciliated airway epithelia in mice, and antagonizes canonical Wnt signaling via direct regulation of β-catenin. We report that deletion of the mouse Cby1 gene results in cystic kidneys, a phenotype common to ciliopathies, and that Cby1 facilitates the formation of primary cilia and ciliary recruitment of the Joubert syndrome protein Arl13b. Localization of Cby1 to the distal end of mature centrioles depends on the centriole protein Ofd1. Superresolution microscopy using both three-dimensional SIM and STED reveals that Cby1 localizes to an ∼250-nm ring at the distal end of the mature centriole, in close proximity to Ofd1 and Ahi1, a component of the transition zone between centriole and cilium. The amount of centriole-localized Ahi1, but not Ofd1, is reduced in Cby1(-/-) cells. This suggests that Cby1 is required for efficient recruitment of Ahi1, providing a possible molecular mechanism for the ciliogenesis defect in Cby1(-/-) cells.
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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.