ArticleNature communications2022
The inner junction protein CFAP20 functions in motile and non-motile cilia and is critical for vision.
Article in Nature communications, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 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
23 citing papers in PubMed, 32 citations in OpenAlex.
- Microtubule Inner Protein CFAP77 Contributes to Sperm Motility and Male Fertility in Mice.Andrology · 2026Article
- Systemic and Ocular Manifestations of a Ciliopathy: A Case Report of Renal-Retinal Involvement in Senior-Loken Syndrome.Journal of clinical medicine · 2026Article
- Loss-of-function variants in SAXO6, encoding a microtubule inner protein of photoreceptor cilia, cause a late-onset retinal dystrophy.American journal of human genetics · 2026Article
- Ciliary Defects in Inherited Retinal Diseases.Advanced genetics (Hoboken, N.J.) · 2026Review
- Article
- A single-cell multiomics roadmap of zebrafish spermatogenesis reveals regulatory principles of male germline formation.Molecular systems biology · 2026Article
- Research Hotspots and Trends in Ciliopathies: A Bibliometric and Visualization Analysis.BioMed research international · 2026Article
- NSUN7 is a catalytically inactive RNA mNature communications · 2025Article
- The core outer junction protein CFAP77 connects A- and B-tubules within doublet microtubules of cilia and flagella.PLoS biology · 2025Article
- Senior-Loken Syndrome: Ocular Perspectives on Genetics, Pathogenesis, and Management.Biomolecules · 2025Review
- Investigating cigarette smoke-induced airway inflammation and sperm activity impairment in rats based on cilia-associated proteins.3 Biotech · 2025Article
- Long-Read Whole-Genome Sequencing as a Tool for Variant Detection in Inherited Retinal Dystrophies.International journal of molecular sciences · 2025Article
- LRRC56 deletion causes primary ciliary dyskinesia in mice characterized by dynein arms defects.Biology open · 2025Article
- Adaptation in the Alleyways: Candidate Genes Under Potential Selection in Urban Coyotes.Genome biology and evolution · 2025Article
- Case Report: Identification of a novel hemizygousFrontiers in medicine · 2025Article
- FAP20 is required for flagellum assembly inMolecular biology of the cell · 2024Article
- Neurons dispose of hyperactive kinesin into glial cells for clearance.The EMBO journal · 2024Article
- Long-read sequencing improves the genetic diagnosis of retinitis pigmentosa by identifying an Alu retrotransposon insertion in the EYS gene.Mobile DNA · 2024Article
- Centriole and transition zone structures in photoreceptor cilia revealed by cryo-electron tomography.Life science alliance · 2024Article
- FAP20 is required for flagellum assembly inbioRxiv : the preprint server for biology · 2024Article
Corrections and comments
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Authors and funding
30 authors at 15 institutions in 4 countries.
Funding
Abstract
Motile and non-motile cilia are associated with mutually-exclusive genetic disorders. Motile cilia propel sperm or extracellular fluids, and their dysfunction causes primary ciliary dyskinesia. Non-motile cilia serve as sensory/signalling antennae on most cell types, and their disruption causes single-organ ciliopathies such as retinopathies or multi-system syndromes. CFAP20 is a ciliopathy candidate known to modulate motile cilia in unicellular eukaryotes. We demonstrate that in zebrafish, cfap20 is required for motile cilia function, and in C. elegans, CFAP-20 maintains the structural integrity of non-motile cilia inner junctions, influencing sensory-dependent signalling and development. Human patients and zebrafish with CFAP20 mutations both exhibit retinal dystrophy. Hence, CFAP20 functions within a structural/functional hub centered on the inner junction that is shared between motile and non-motile cilia, and is distinct from other ciliopathy-associated domains or macromolecular complexes. Our findings suggest an uncharacterised pathomechanism for retinal dystrophy, and potentially for motile and non-motile ciliopathies in general.
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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.