Affinage

CEP128

Centrosomal protein 128 · UniProt Q6ZU80

Length
1094 aa
Mass
128.0 kDa
Annotated
2026-06-09
10 papers in source corpus 3 papers cited in narrative 3 extracted findings
Cross-family judge vs UniProt: Affinage preferred faithfulness: 4/4 claims corpus-supported (100%)

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

CEP128 is a subdistal appendage (SDA) protein of the mother centriole that organizes appendage architecture and links the centriole to ciliary signaling (PMID:29514088, PMID:30623524). It associates with ODF2 and acts downstream of it in the hierarchical assembly of the SDA: loss of CEP128 dissociates downstream SDA components (centriolin, Ndel1, ninein, and CEP170) from the centriole while ODF2 and distal appendage components remain, and it is required for centriolar microtubule stability (PMID:30623524). At the primary cilium, CEP128 supports TGF-β/BMP signaling by maintaining ciliary localization of RAB11 and the phosphorylation of proteins controlling cilium-associated vesicle trafficking (PMID:29514088). In the testis, CEP128 has a tissue-specific ciliogenesis role required for sperm flagella formation and spermatogenesis, mediated in part through regulation of spermatogenesis gene expression and TGF-β/BMP pathway phosphorylation, with loss causing defective flagella, reduced sperm count, impaired motility, and male infertility (PMID:35296684).

Mechanistic history

Synthesis pass · year-by-year structured walk · 3 steps
  1. 2018 High

    Established CEP128 as a mother-centriole subdistal appendage protein whose loss disrupts ciliary TGF-β/BMP signaling, linking an appendage component to a signaling output via vesicle trafficking.

    Evidence Immunofluorescence localization, siRNA knockdown signaling assays, zebrafish loss-of-function, and quantitative phosphoproteomics in mammalian cells

    PMID:29514088

    Open questions at the time
    • Direct molecular mechanism connecting CEP128 to RAB11 recruitment not resolved
    • Identity of the kinase(s)/phosphatase(s) downstream of CEP128 in TGF-β signaling not defined
  2. 2019 High

    Placed CEP128 in the hierarchy of subdistal appendage assembly, showing it acts downstream of ODF2 to recruit other SDA components and stabilize centriolar microtubules.

    Evidence SR-SIM localization, reciprocal immunoprecipitation with ODF2, and siRNA knockdown scored against multiple SDA/DA markers

    PMID:30623524

    Open questions at the time
    • Structural basis of the CEP128-ODF2 interaction not determined
    • How CEP128 mechanically stabilizes centriolar microtubules not established
  3. 2022 High

    Demonstrated an in vivo, tissue-specific requirement for CEP128 in sperm flagella formation and spermatogenesis, distinguishing its testis role from general ciliogenesis.

    Evidence Knock-in (R222Q) and knockout mouse models with sperm morphology/motility analysis and gene expression/phosphorylation profiling

    PMID:35296684

    Open questions at the time
    • Why flagellar defects occur while cilia in other organs remain normal is unexplained
    • Direct targets linking CEP128 to spermatogenesis gene expression not identified

Open questions

Synthesis pass · forward-looking unresolved questions
  • How CEP128 mechanistically couples appendage architecture to RAB11-dependent vesicle trafficking and TGF-β/BMP signal transduction remains unresolved.
  • No structural model of CEP128 or its appendage interactions
  • No defined direct binding partner mediating RAB11 ciliary recruitment
  • Relationship between SDA assembly defects and signaling defects not causally dissected

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0060090 molecular adaptor activity 2 GO:0008092 cytoskeletal protein binding 1
Localization
GO:0005815 microtubule organizing center 2 GO:0005929 cilium 1
Pathway
R-HSA-162582 Signal Transduction 2 R-HSA-1474165 Reproduction 1 R-HSA-1852241 Organelle biogenesis and maintenance 1
Partners
CEP170NDEL1NINODF2RAB11
Complex memberships
subdistal appendage

Evidence

Reading pass · 3 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2018 CEP128 localizes to the subdistal appendages (SDA) of the mother centriole, and its loss impairs TGF-β/BMP signaling at the primary cilium; mechanistically, loss of CEP128 disrupts ciliary localization of RAB11, consistent with defective vesicle trafficking at the cilium, and quantitative phosphoproteomics showed CEP128 loss affects TGF-β1-induced phosphorylation of multiple proteins regulating cilium-associated vesicle trafficking. Immunofluorescence localization, siRNA knockdown with signaling assays, zebrafish loss-of-function, quantitative phosphoproteomics Cell reports High 29514088
2019 CEP128 associates with ODF2 at the subdistal appendage of the mother centriole; CEP128 knockdown causes dissociation of other SDA components (centriolin, Ndel1, ninein, CEP170) from the centriole while ODF2 and distal appendage components remain, demonstrating CEP128 acts downstream of ODF2 in hierarchical SDA assembly and is required for centriolar microtubule stability. Super-resolution structured illumination microscopy (SR-SIM), siRNA knockdown, immunoprecipitation, immunofluorescence of SDA/DA markers Genes to cells : devoted to molecular & cellular mechanisms High 30623524
2022 CEP128 is required for spermatogenesis; homozygous knock-in (R222Q) and knockout mice show defective sperm flagella, reduced sperm count, and impaired motility leading to male infertility, while cilia in other organs remain normal, indicating a testis-specific ciliogenesis role; mechanistically, CEP128 regulates expression of spermatogenesis genes and phosphorylation of TGF-β/BMP signaling members during spermatogenesis. Knock-in and knockout mouse models, sperm morphology/motility analysis, gene expression and phosphorylation profiling Nature communications High 35296684

Source papers

Stage 0 corpus · 10 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2018 Circular RNA CEP128 acts as a sponge of miR-145-5p in promoting the bladder cancer progression via regulating SOX11. Molecular medicine (Cambridge, Mass.) 80 30134837
2018 CEP128 Localizes to the Subdistal Appendages of the Mother Centriole and Regulates TGF-β/BMP Signaling at the Primary Cilium. Cell reports 61 29514088
2019 Circular RNA CEP128 promotes bladder cancer progression by regulating Mir-145-5p/Myd88 via MAPK signaling pathway. International journal of cancer 58 30939216
2022 CEP128 is involved in spermatogenesis in humans and mice. Nature communications 44 35296684
2019 Cep128 associates with Odf2 to form the subdistal appendage of the centriole. Genes to cells : devoted to molecular & cellular mechanisms 28 30623524
2019 Knockdown of circular RNA CEP128 suppresses proliferation and improves cytotoxic efficacy of temozolomide in glioma cells by regulating miR-145-5p. Neuroreport 28 31599823
2022 Downregulation of Circ-CEP128 Enhances the Paclitaxel Sensitivity of Cervical Cancer Through Regulating miR-432-5p/MCL1. Biochemical genetics 12 35391656
2018 CEP128 is a crucial risk locus for autoimmune thyroid diseases. Molecular and cellular endocrinology 12 30393005
2023 Modulation Effects of the CEP128 Gene on Radiotherapy-Related Brain Injury: A Longitudinal Structural Study Using Multi-Parametric Brain MR Images. Journal of magnetic resonance imaging : JMRI 5 37249021
2025 RETRACTION: Circular RNA CEP128 Promotes Bladder Cancer Progression by Regulating Mir-145-5p/Myd88 via MAPK Signaling Pathway. International journal of cancer 0 40019841

Missed literature

Know a paper Affinage missed for CEP128? Flag it for the maintainers and the community.

No submissions yet.