ArticlePLoS genetics2020
CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components.
Article in PLoS genetics, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.
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Who cites it
13 citing papers in PubMed, 37 citations in OpenAlex.
- Identification of novel biallelic mutations in CFAP53 associated with fetal situs inversus totalis and literature review.Journal of applied genetics · 2026Review
- Novel Compound Heterozygous CFAP53 Variants in a Fetus With Situs Inversus Totalis: A Case Report.Cureus · 2026Article
- Broadening horizons: new links between cilia and heart development and disease.Frontiers in cardiovascular medicine · 2026Review
- The Heterotaxy Gene CCDC11 Is Important for Cytokinesis via RhoA Regulation.Cytoskeleton (Hoboken, N.J.) · 2025Article
- A New Method for Lateral Visualization of the Primary Cilia on the Surfaces of Cells Cultured on White Glass Rods.Translational vision science & technology · 2024Article
- Biallelic Variants inCells · 2024Article
- Tektin bundle interacting protein, TEKTIP1, functions to stabilize the tektin bundle and axoneme in mouse sperm flagella.Cellular and molecular life sciences : CMLS · 2024Article
- FAP106 is an interaction hub for assembling microtubule inner proteins at the cilium inner junction.Nature communications · 2023Article
- The centriolar satellite protein Cfap53 facilitates formation of the zygotic microtubule organizing center in the zebrafish embryo.Development (Cambridge, England) · 2022Article
- Biallelic DNAH9 mutations are identified in Chinese patients with defective left-right patterning and cilia-related complex congenital heart disease.Human genetics · 2022Article
- Article
- Whole genome sequencing in the diagnosis of primary ciliary dyskinesia.BMC medical genomics · 2021Article
- Essential Role of CFAP53 in Sperm Flagellum Biogenesis.Frontiers in cell and developmental biology · 2021Article
Corrections and comments
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Authors and funding
13 authors at 5 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Motile cilia can beat with distinct patterns, but how motility variations are regulated remain obscure. Here, we have studied the role of the coiled-coil protein CFAP53 in the motility of different cilia-types in the mouse. While node (9+0) cilia of Cfap53 mutants were immotile, tracheal and ependymal (9+2) cilia retained motility, albeit with an altered beat pattern. In node cilia, CFAP53 mainly localized at the base (centriolar satellites), whereas it was also present along the entire axoneme in tracheal cilia. CFAP53 associated tightly with microtubules and interacted with axonemal dyneins and TTC25, a dynein docking complex component. TTC25 and outer dynein arms (ODAs) were lost from node cilia, but were largely maintained in tracheal cilia of Cfap53-/- mice. Thus, CFAP53 at the base of node cilia facilitates axonemal transport of TTC25 and dyneins, while axonemal CFAP53 in 9+2 cilia stabilizes dynein binding to microtubules. Our study establishes how differential localization and function of CFAP53 contributes to the unique motion patterns of two important mammalian cilia-types.
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