ArticleNature communications2014
Cryo-electron tomography reveals ciliary defects underlying human RSPH1 primary ciliary dyskinesia.
Article in Nature communications, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 87 papers.
What it found
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Who cites it
87 citing papers in PubMed, 164 citations in OpenAlex.
- Preclinical human models of primary ciliary dyskinesia.European respiratory review : an official journal of the European Respiratory Society · 2026Review
- Loss-of-function variants in ODAD1 disrupt ODA docking and induce actin cytoskeletal remodeling in primary ciliary dyskinesia.Cell discovery · 2026Article
- Novel pathogenic variant inFrontiers in molecular biosciences · 2026Article
- BiallelicFrontiers in genetics · 2026Article
- TRiC Is a Structural Component of Mammalian Sperm Axonemes.Cytoskeleton (Hoboken, N.J.) · 2025Review
- Advances in high-resolution imaging of centrioles and cilia.Journal of cell science · 2025Review
- 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
- Deficiency in DNAH12 causes male infertility by impairing DNAH1 and DNALI1 recruitment in humans and mice.eLife · 2025Article
- Cryo-electron tomography of eel sperm flagella reveals a molecular "minimum system" for motile cilia.Molecular biology of the cell · 2025Article
- IQUB mutation induces radial spoke 1 deficiency causing asthenozoospermia with normal sperm morphology in humans and mice.Cell communication and signaling : CCS · 2025Article
- Proteomic and structural comparison between cilia from primary ciliary dyskinesia patients with a DNAH5 defect.Frontiers in molecular biosciences · 2025Article
- Article
- The Increase in the Frequency and Amplitude of the Beating of Isolated Mouse Tracheal Cilia Reactivated by ATP and cAMP with Elevation in pH.International journal of molecular sciences · 2024Article
- Primary Ciliary Dyskinesia: A Clinical Review.Cells · 2024Review
- The MBO2/FAP58 heterodimer stabilizes assembly of inner arm dyneinMolecular biology of the cell · 2024Article
- Multi-scale structures of the mammalian radial spoke and divergence of axonemal complexes in ependymal cilia.Nature communications · 2024Article
- LRRC23 truncation impairs radial spoke 3 head assembly and sperm motility underlying male infertility.eLife · 2023Article
- Integrated modeling of the Nexin-dynein regulatory complex reveals its regulatory mechanism.Nature communications · 2023Article
- Article
27 more citing papers are in PubMed but not listed here.
Corrections and comments
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Authors and funding
9 authors at 2 institutions in 1 country.
Funding
Abstract
Cilia play essential roles in normal human development and health; cilia dysfunction results in diseases such as primary ciliary dyskinesia (PCD). Despite their importance, the native structure of human cilia is unknown, and structural defects in the cilia of patients are often undetectable or remain elusive because of heterogeneity. Here we develop an approach that enables visualization of human (patient) cilia at high-resolution using cryo-electron tomography of samples obtained noninvasively by nasal scrape biopsy. We present the native 3D structures of normal and PCD-causing RSPH1-mutant human respiratory cilia in unprecedented detail; this allows comparisons of cilia structure across evolutionarily distant species and reveals the previously unknown primary defect and the heterogeneous secondary defects in RSPH1-mutant cilia. Our data provide evidence for structural and functional heterogeneity in radial spokes, suggest a mechanism for the milder RSPH1 PCD phenotype and demonstrate that cryo-electron tomography can be applied to human disease by directly imaging patient samples.
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