ArticleeLife2024
CCDC113 stabilizes sperm axoneme and head-tail coupling apparatus to ensure male fertility.
Article in eLife, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed.
- Loss-of-function mutations in Ccdc113 cause male infertility in both humans and mice.Asian journal of andrology · 2026Article
- Togaram Ensures Axial Alignment of the Sperm Neck.bioRxiv : the preprint server for biology · 2026Article
- A novel hsa_circ_0070963/miR-223-3p/International journal of reproductive biomedicine · 2026Article
- The spermatozoon neck role in infertility and intracytoplasmic sperm injection outcomes.Journal of assisted reproduction and genetics · 2026Review
- Exploring retinal microglia: development, degeneration, and iPSC-derived model systems.Brain structure & function · 2026Review
- A Novel Homozygous Mutation inHuman mutation · 2026Article
- Mutations in CFAP57 disrupt the localization of MYH10 and IFT88, leading to flagellogenesis failure in humans and mice.Human genomics · 2025Article
- Molecular models of the sperm head-tail coupling apparatus.Journal of cell science · 2025Review
- Differentiated muscle cells of salamander Pleurodeles waltl re-enter the cell cycle.Histochemistry and cell biology · 2025Article
- A subset of evolutionarily conserved centriolar satellite core components is crucial for sperm flagellum biogenesis.Theranostics · 2025Article
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Authors and funding
11 authors.
Funding
Abstract
The structural integrity of the sperm is crucial for male fertility, defects in sperm head-tail linkage and flagellar axoneme are associated with acephalic spermatozoa syndrome (ASS) and the multiple morphological abnormalities of the sperm flagella (MMAF). Notably, impaired head-tail coupling apparatus (HTCA) often accompanies defects in the flagellum structure, however, the molecular mechanisms underlying this phenomenon remain elusive. Here, we identified an evolutionarily conserved coiled-coil domain-containing (CCDC) protein, CCDC113, and found the disruption of CCDC113 produced spermatozoa with disorganized sperm flagella and HTCA, which caused male infertility. Further analysis revealed that CCDC113 could bind to CFAP57 and CFAP91, and function as an adaptor protein for the connection of radial spokes, nexin-dynein regulatory complex (N-DRC), and doublet microtubules (DMTs) in the sperm axoneme. Moreover, CCDC113 was identified as a structural component of HTCA, collaborating with SUN5 and CENTLEIN to connect sperm head to tail during spermiogenesis. Together, our studies reveal that CCDC113 serve as a critical hub for sperm axoneme and HTCA stabilization in mice, providing insights into the potential pathogenesis of infertility associated with human
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