ArticlePLoS genetics2020
Rsph4a is essential for the triplet radial spoke head assembly of the mouse motile cilia.
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 28 papers.
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Who cites it
28 citing papers in PubMed, 40 citations in OpenAlex.
- Article
- Transcriptomic Analysis of Fermented Chinese Chive Selectively Attenuating Deoxynivalenol-Induced Ovarian Toxicity in Mice.Antioxidants (Basel, Switzerland) · 2026Article
- JHY enables the transition from switchable to fixed ciliary waveforms in metazoan evolution.EMBO reports · 2026Article
- BiallelicFrontiers in genetics · 2026Article
- Tppp3 determines basal body positioning and identity of respiratory cilia via microtubule assembly and sphingolipid homeostasis.Proceedings of the National Academy of Sciences of the United States of America · 2025Article
- Three-dimensional beating pattern of the ciliary tip in the live ciliate Tetrahymena.Journal of cell science · 2025Article
- Exon Skipping Therapy Restores Ciliary Function in USH2A-Related Retinal Degeneration.Investigative ophthalmology & visual science · 2025Article
- Heterogeneity of radial spoke components in Tetrahymena cilia.Cellular and molecular life sciences : CMLS · 2025Article
- Mouse radial spoke 3 is a metabolic and regulatory hub in cilia.Nature structural & molecular biology · 2025Article
- Analysis of clinical and genetic features in an adolescent patient with primary ciliary dyskinesia induced by homozygous mutation in theFrontiers in pediatrics · 2025Article
- Selection against domestication alleles in introduced rabbit populations.Nature ecology & evolution · 2024Article
- Multi-scale structures of the mammalian radial spoke and divergence of axonemal complexes in ependymal cilia.Nature communications · 2024Article
- A novel homozygous RSPH4A variant in a family with primary ciliary dyskinesia and literature review.Frontiers in genetics · 2024Article
- LRRC23 truncation impairs radial spoke 3 head assembly and sperm motility underlying male infertility.eLife · 2023Article
- Review
- TheInternational journal of molecular sciences · 2023Review
- LRRC46 Accumulates at the Midpiece of Sperm Flagella and Is Essential for Spermiogenesis and Male Fertility in Mouse.International journal of molecular sciences · 2022Article
- Development and characterization of human fetal female reproductive tract organoids to understand Müllerian duct anomalies.Proceedings of the National Academy of Sciences of the United States of America · 2022Article
- NovelFrontiers in genetics · 2022Article
- LRRC23 is a conserved component of the radial spoke that is necessary for sperm motility and male fertility in mice.Journal of cell science · 2021Article
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Authors and funding
8 authors at 4 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Motile cilia/flagella are essential for swimming and generating extracellular fluid flow in eukaryotes. Motile cilia harbor a 9+2 arrangement consisting of nine doublet microtubules with dynein arms at the periphery and a pair of singlet microtubules at the center (central pair). In the central system, the radial spoke has a T-shaped architecture and regulates the motility and motion pattern of cilia. Recent cryoelectron tomography data reveal three types of radial spokes (RS1, RS2, and RS3) in the 96 nm axoneme repeat unit; however, the molecular composition of the third radial spoke, RS3 is unknown. In human pathology, it is well known mutation of the radial spoke head-related genes causes primary ciliary dyskinesia (PCD) including respiratory defect and infertility. Here, we describe the role of the primary ciliary dyskinesia protein Rsph4a in the mouse motile cilia. Cryoelectron tomography reveals that the mouse trachea cilia harbor three types of radial spoke as with the other vertebrates and that all triplet spoke heads are lacking in the trachea cilia of Rsph4a-deficient mice. Furthermore, observation of ciliary movement and immunofluorescence analysis indicates that Rsph4a contributes to the generation of the planar beating of motile cilia by building the distal architecture of radial spokes in the trachea, the ependymal tissues, and the oviduct. Although detailed mechanism of RSs assembly remains unknown, our results suggest Rsph4a is a generic component of radial spoke heads, and could explain the severe phenotype of human PCD patients with RSPH4A mutation.
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