Affinage

CEP164

Centrosomal protein of 164 kDa · UniProt Q9UPV0

Length
1460 aa
Mass
164.3 kDa
Annotated
2026-06-09
22 papers in source corpus 19 papers cited in narrative 20 extracted findings
Cross-family judge vs UniProt: Affinage preferred faithfulness: 8/8 claims corpus-supported (100%)

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

CEP164 is a distal appendage protein of the mother centriole that serves as a master scaffold for ciliogenesis (PMID:17954613). It homodimerizes through its central coiled-coil region to achieve mother centriole localization, which is a prerequisite for its downstream recruiting functions (PMID:40305080). CEP164's central activity is the recruitment of the kinase TTBK2 via a proline-rich interaction; this complex, whose structural basis and disruption by nephronophthisis-associated mutations have been defined, triggers ciliogenesis and is reinforced by liquid-liquid phase separation driven by multivalent electrostatic interactions through CEP164's intrinsically disordered region (PMID:24982133, PMID:25297623, PMID:34499853, PMID:40483689). The CEP164-TTBK2 axis, dependent on TTBK2 kinase activity, drives removal of CP110 and recruitment of IFT-A, IFT-B, and dynein-2 complexes to the mother centriole (PMID:40305080), with CEP164 being required for stabilization of IFT-B at the basal body and connecting cilium in photoreceptors (PMID:36074756). Beyond intraflagellar transport, CEP164 governs membrane events of ciliogenesis: it drives enlargement of small vesicles docked to the mother centriole into ciliary vesicle intermediates, recruits Chibby1 (and its partners FAM92A/FAM92B) to enable basal body docking, and controls ciliary targeting of membrane-associated proteins including Rab8, Rab11, Arl13b, and INPP5E (PMID:23150559, PMID:29244804, PMID:34085951). CEP164 also anchors the GLI2 transcription factor at the mother centriole, restraining Hedgehog target-gene activation and Cyclin D-CDK6 expression in a cilia-independent manner (PMID:33251215, PMID:37199136). Loss of CEP164 abolishes primary cilia and causes nephronophthisis-related ciliopathy phenotypes, including postnatal renal cyst growth driven by tubular hyperproliferation that is reversible by cell-cycle inhibition (PMID:22863007, PMID:31248650). CEP164 was also characterized as an ATR/ATM-phosphorylated (Ser186) mediator of the DNA damage response (PMID:18283122), but rigorous genome-editing studies found no requirement for CEP164 in DDR signaling or nuclear localization (PMID:26966185).

Mechanistic history

Synthesis pass · year-by-year structured walk · 16 steps
  1. 2007 High

    Established CEP164 as a distal appendage component of the mother centriole that is required for primary cilium formation, defining the structural platform on which all later mechanism rests.

    Evidence siRNA screen, immunogold electron microscopy, and RNAi knockdown with ciliogenesis readout in human cells

    PMID:17954613

    Open questions at the time
    • Did not define molecular partners recruited by CEP164
    • Mechanism of distal appendage targeting unresolved
  2. 2008 Medium

    Addressed whether CEP164 has a nuclear function by identifying it as an ATR/ATM-phosphorylated (Ser186) mediator of the DNA damage response, implying a role beyond the centriole.

    Evidence Co-IP, in vitro kinase assay, phospho-specific antibodies, and checkpoint assays after replication stress/UV/IR

    PMID:18283122

    Open questions at the time
    • Did not reconcile a centriolar protein acting in nuclear DDR
    • DDR requirement later contested by genome editing
  3. 2009 Medium

    Extended the proposed DDR role by showing UV-dependent CEP164 interaction with XPA and recruitment to lesion sites, linking CEP164 to nucleotide excision repair signaling.

    Evidence Co-IP, domain mapping, ChIP, CPD colocalization, and siRNA knockdown with survival/CHK1 readouts

    PMID:19197159

    Open questions at the time
    • Single lab
    • Direct DDR requirement contradicted by later degron/CRISPR studies
  4. 2012 Medium

    Connected CEP164 loss to ciliopathy in vivo and to ciliary membrane-protein targeting, situating it within a discrete ARL13B-INPP5E-PDE6D network distinct from NPHP/MKS modules.

    Evidence DNA-damage foci colocalization, sensitivity assays, zebrafish morpholino, and pulldown/Co-IP with ciliary targeting analysis

    PMID:22863007 PMID:23150559

    Open questions at the time
    • Direct vs. indirect interaction with INPP5E network not fully resolved
    • Mechanistic link between DDR and ciliopathy phenotype unclear
  5. 2014 High

    Defined the central ciliogenic mechanism: CEP164 recruits TTBK2 via a proline-rich motif to the mother centriole, an event required for CP110 removal and cilium initiation, with chimeric rescue proving sufficiency.

    Evidence Co-IP, domain mapping, binding-motif mutagenesis, chimeric protein rescue, and in vitro phosphorylation across two independent labs

    PMID:24982133 PMID:25297623

    Open questions at the time
    • Did not resolve the structural basis of the interaction
    • Downstream IFT recruitment steps not yet mapped
  6. 2014 Medium

    Showed CEP164 depletion perturbs cell cycle progression and induces EMT/pro-fibrotic changes rescuable by wild-type but not disease-mutant CEP164, linking centriolar dysfunction to proliferative pathology.

    Evidence RPE-FUCCI live imaging, FACS, proliferation assays, RT-qPCR, zebrafish knockdown, and WT vs. mutant rescue

    PMID:25340510

    Open questions at the time
    • Causal link between cilium loss and EMT not isolated
    • Single lab
  7. 2016 Medium

    Directly challenged the DDR model by showing that conditional degron depletion and CRISPR disruption of CEP164 block ciliogenesis without affecting irradiation sensitivity, and detected no nuclear CEP164.

    Evidence Auxin-inducible degron, genome editing, clonogenic survival assays, and multiple tagged CEP164 constructs in DT40 and RPE cells

    PMID:26966185

    Open questions at the time
    • Cell-type-specific DDR contributions not excluded
    • Single lab negative result
  8. 2017 High

    Distinguished CEP164's roles in multiciliogenesis versus primary ciliogenesis, showing it controls vesicle recruitment, Chibby1 recruitment, basal body docking, and membrane-protein targeting but is dispensable for IFT recruitment in multicilia.

    Evidence FoxJ1-Cre conditional KO mouse, primary tracheal multiciliated cell cultures, immunofluorescence, and EM

    PMID:29244804

    Open questions at the time
    • Molecular mechanism of vesicle recruitment not defined
    • Why IFT requirement differs between cilium types unexplained
  9. 2019 High

    Established the disease mechanism of renal cystogenesis: collecting-duct Cep164 deletion abolishes cilia and drives hyperproliferative cyst growth reversible by a cell-cycle inhibitor.

    Evidence Collecting duct-specific conditional KO mouse with roscovitine pharmacological rescue

    PMID:31248650

    Open questions at the time
    • Signaling pathway linking cilium loss to hyperproliferation not fully mapped
  10. 2020 Medium

    Revealed a cilia-independent signaling role: CEP164 anchors GLI2 at the mother centriole to restrain Hedgehog-driven Cyclin D-CDK6 expression, with loss enhancing cancer cell clonogenicity.

    Evidence CRISPR KO, colocalization, cell cycle analysis, clonogenic assays, and gene expression in pancreatic cancer cells

    PMID:33251215

    Open questions at the time
    • Single lab
    • Direct GLI2 binding not yet domain-mapped at this stage
  11. 2021 High

    Provided the structural basis of the CEP164-TTBK2 complex and showed how ciliopathic CEP164 mutations disrupt it, mechanistically connecting genotype to ciliogenesis failure.

    Evidence Structural/biophysical analysis, biochemical interaction assays, and mutagenesis of nephronophthisis variants with ciliogenesis readouts

    PMID:34499853

    Open questions at the time
    • Full-length complex architecture in cellular context not resolved
  12. 2021 High

    Confirmed in efferent ducts that CEP164 recruits Chibby1 to mediate basal body docking, with loss causing undocked cytoplasmic basal bodies and diminished transition-zone marker localization.

    Evidence FoxJ1-Cre conditional KO mouse, immunofluorescence, TEM, and histology

    PMID:34085951

    Open questions at the time
    • Direct biochemical CEP164-Chibby1 interaction not isolated here
    • Order of docking versus recruitment events unresolved
  13. 2022 High

    Demonstrated a post-ciliogenesis maintenance role: CEP164 is required for recruitment and stabilization of IFT-B particles and basal body docking in photoreceptors, with IFT88/57/140 reduced upon loss.

    Evidence Multiple conditional and inducible KO mouse models, immunofluorescence, EM, and disc labeling in rod photoreceptors

    PMID:36074756

    Open questions at the time
    • Mechanism of IFT-B stabilization at the basal body not molecularly defined
  14. 2023 Medium

    Mapped the GLI2-binding region of CEP164 and showed its ectopic expression displaces centriolar GLI2 and de-represses Hedgehog targets in cells with or without cilia, confirming a cilia-independent signaling control point.

    Evidence Co-IP, domain mapping, ectopic binding-region expression, colocalization, and RT-PCR of Hh targets

    PMID:37199136

    Open questions at the time
    • Single lab
    • How GLI2 anchoring is regulated physiologically unknown
  15. 2024 Medium

    Revealed cell-type-specific in vivo functions in skeleton, with osteoblast Cep164 loss increasing γH2AX-positive cells while chondrocyte deletion was benign, reopening a possible DDR-linked role in specific lineages.

    Evidence Mesodermal-, osteoblast-, and chondrocyte-specific conditional KO mice with γH2AX immunofluorescence and histology

    PMID:39612644

    Open questions at the time
    • Whether γH2AX accumulation reflects a direct CEP164 DDR function is unresolved
    • Mechanism of cell-type specificity unknown
  16. 2025 High

    Integrated the recruiting mechanism: CEP164 homodimerizes via its coiled-coil for centriole localization, and forms phase-separated condensates with TTBK2 that, via TTBK2 kinase activity, drive CP110 removal and IFT-A/B and dynein-2 recruitment; vesicle enlargement was placed upstream of axoneme growth.

    Evidence KO cell lines with chimeric domain constructs, Co-IP, phase separation assays, live-cell condensate imaging, and FIB-SEM ultrastructure (one source a preprint)

    PMID:40305080 PMID:40483689

    Open questions at the time
    • Physiological regulation of condensate formation not defined
    • Coupling between CP110 removal and IFT recruitment is variable and unexplained

Open questions

Synthesis pass · forward-looking unresolved questions
  • It remains unresolved whether CEP164 has any genuine, cell-type-restricted DNA damage response function, given the contradiction between early DDR mediator claims, the rigorous negative genome-editing study, and recent osteoblast γH2AX findings.
  • No reconciliation of nuclear DDR role with absence of detectable nuclear localization
  • Lineage-specific DDR contributions not mechanistically tested

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0060090 molecular adaptor activity 4 GO:0005198 structural molecule activity 2
Localization
GO:0005815 microtubule organizing center 5 GO:0005929 cilium 3 GO:0005856 cytoskeleton 1
Pathway
R-HSA-1852241 Organelle biogenesis and maintenance 5 R-HSA-1643685 Disease 3 R-HSA-162582 Signal Transduction 2
Partners
ARL13BATMATRCBY1GLI2INPP5ETTBK2XPA
Complex memberships
centriole distal appendage

Evidence

Reading pass · 20 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2007 CEP164 localizes to the distal appendages of mature centrioles (mother centriole), as determined by immunogold electron microscopy, and is indispensable for primary cilium formation. Unlike subdistal appendage proteins ninein and Cep170, CEP164 persists at centrioles throughout mitosis, and its localization is mutually independent of ninein/Cep170 during interphase. siRNA screen, immunogold electron microscopy, immunofluorescence, RNAi knockdown with ciliogenesis phenotype readout The Journal of cell biology High 17954613
2008 CEP164 (KIAA1052) interacts with both ATR and ATM kinases, is phosphorylated at Ser186 by ATR/ATM in vitro and in vivo upon replication stress, UV, and ionizing radiation, and functions as a mediator in the DNA damage response. MDC1 knockdown severely reduces Ser186 phosphorylation, and CEP164 knockdown diminishes DNA damage-induced phosphorylation of RPA, H2AX, MDC1, CHK2, and CHK1 (but not NBS1), and causes G2/M checkpoint defects. Co-immunoprecipitation, in vitro kinase assay, siRNA knockdown, phospho-specific antibody analysis, cell cycle checkpoint assays Genes & development Medium 18283122
2009 Upon UV irradiation, CEP164 interacts with XPA (Xeroderma pigmentosum group A) in a UV-dependent manner; CEP164 binds to amino acids 4–97 of XPA. XPA is required for recruitment of CEP164 to cyclobutane pyrimidine dimer (CPD) sites. CEP164 knockdown compromises cell survival upon UV damage and impairs UV-induced CHK1 phosphorylation. Co-immunoprecipitation, chromatin immunoprecipitation (ChIP), immunofluorescence colocalization with CPD, siRNA knockdown, complementation with XPA deletion mutants Cell cycle (Georgetown, Tex.) Medium 19197159
2012 Upon induced DNA damage, CEP164 co-localizes to nuclear foci positive for TIP60 (an ATM activator) together with ZNF423 and NPHP10. CEP164 knockdown causes sensitivity to DNA damaging agents. Zebrafish cep164 knockdown results in dysregulated DDR and a nephronophthisis-related ciliopathy phenotype. Immunofluorescence colocalization at DNA damage foci, siRNA knockdown with DNA damage sensitivity assays, zebrafish morpholino knockdown Cell Medium 22863007
2012 CEP164 physically interacts with INPP5E (inositol polyphosphate-5-phosphatase E) and participates in a functional network with ARL13B, INPP5E, and PDE6D for ciliary targeting of INPP5E. This network is distinct from those defined by NPHP and MKS proteins. Protein-protein interaction assays (pulldown/Co-IP), genetic analysis, ciliary targeting experiments Proceedings of the National Academy of Sciences of the United States of America Medium 23150559
2014 CEP164 recruits TTBK2 (Tau tubulin kinase 2) to the mother centriole to trigger ciliogenesis. CEP164 is a likely physiological substrate of TTBK2. Complex formation between CEP164 and TTBK2 is mediated by mapped interaction domains, and is essential for TTBK2 recruitment to basal bodies. Ciliogenesis can be rescued in CEP164-depleted cells by chimeric proteins fusing TTBK2 to the C-terminal centriole-targeting domain of CEP164. TTBK2 acts upstream of CEP164 to contribute to distal appendage assembly. Co-immunoprecipitation, domain mapping, siRNA knockdown, chimeric protein rescue, immunofluorescence Proceedings of the National Academy of Sciences of the United States of America High 24982133
2014 CEP164 is essential for TTBK2 centriolar localization via a proline-rich motif on TTBK2 (not via SxIP/EB1 motifs). TTBK2 non-Cep164-binding mutants fail to rescue CP110 removal and ciliogenesis in TTBK2-depleted cells. TTBK2 can phosphorylate CEP164 and Cep97, and TTBK2 kinase activity inhibits the CEP164–Dishevelled-3 interaction. Co-immunoprecipitation, site-directed mutagenesis of TTBK2 binding motifs, rescue experiments in TTBK2-depleted cells, in vitro phosphorylation assay Genes to cells : devoted to molecular & cellular mechanisms High 25297623
2014 CEP164 knockdown promotes cells to accumulate in S-phase and causes cell cycle acceleration, apoptosis, and epithelial-to-mesenchymal transition. These effects can be rescued by wild-type CEP164 but not disease-associated CEP164 mutants. Overexpression of dominant-negative CEP164 Q525X also induces EMT and pro-fibrotic gene upregulation. siRNA knockdown with live-cell imaging (RPE-FUCCI), FACS, CyQuant proliferation assay, immunofluorescence, RT-qPCR, zebrafish morpholino knockdown, rescue with WT vs. mutant CEP164 PLoS genetics Medium 25340510
2016 NEGATIVE FINDING: Conditional depletion of CEP164 in DT40 cells (auxin-inducible degron) caused no increase in sensitivity to ionising or UV irradiation. Disruption of CEP164 in human RPE cells blocked primary cilium formation but did not affect proliferation or responses to ionising/UV irradiation. No nuclear localization of CEP164 was detected by immunofluorescence or analysis of multiple tagged forms of CEP164. These data suggest CEP164 is not required for the DNA damage response. Auxin-inducible degron conditional depletion, genome editing (CRISPR/reverse genetics), clonogenic survival assays, immunofluorescence with multiple tagged CEP164 constructs Journal of cell science Medium 26966185
2017 CEP164 is required for multiciliogenesis via regulation of small vesicle recruitment, ciliary vesicle formation, and basal body docking. CEP164 is necessary for proper recruitment of Chibby1 (Cby1) and its binding partners FAM92A and FAM92B to the ciliary base in multiciliated cells. CEP164 controls ciliary targeting of membrane-associated proteins including Rab8, Rab11, and Arl13b. Unlike in primary ciliogenesis, CEP164 is dispensable for IFT component recruitment to multicilia. Conditional knockout mouse model (FoxJ1-Cre;CEP164fl/fl), primary tracheal multiciliated cell cultures, immunofluorescence, electron microscopy PLoS genetics High 29244804
2019 Collecting duct-specific deletion of Cep164 in mice abolishes primary cilia from collecting duct epithelium and leads to rapid postnatal cyst growth driven by tubular hyperproliferation. Administration of the cell cycle inhibitor roscovitine blocked cyst growth, confirming cell cycle dysregulation as the primary cystogenesis mechanism. Conditional knockout mouse model (collecting duct-specific Cre), cell cycle analysis, biochemical studies, roscovitine pharmacological treatment Kidney international High 31248650
2020 CEP164 co-localizes with GLI2 transcription factor at the mother centriole, controls GLI2 activation, and thereby regulates Cyclin D-CDK6 expression. Loss of CEP164 in pancreatic cancer cells enhances clonogenicity and alters cell cycle progression through cilia-independent GLI2-Cyclin D/CDK6 activation. CEP164 CRISPR/gene editing, immunofluorescence colocalization, cell cycle analysis, clonogenic assay, gene expression analysis Frontiers in cell and developmental biology Medium 33251215
2021 Structural and biochemical analysis reveals the molecular basis of the CEP164–TTBK2 complex and how it is disrupted by ciliopathic (nephronophthisis) CEP164 mutations. Binding to CEP164 is coordinated with TTBK2 kinase activities. Biochemical interaction assays, structural analysis (NMR/biophysical), mutagenesis of ciliopathic variants, functional ciliogenesis assays Structure (London, England : 1993) High 34499853
2021 CEP164 recruits Chibby1 (Cby1) to basal bodies to facilitate basal body docking and multiciliogenesis in efferent ducts. FoxJ1-Cre;CEP164fl/fl mice show loss of multicilia in efferent ducts with accumulation of undocked basal bodies in the cytoplasm. The apical localization of Cby1 and the transition zone marker NPHP1 is severely diminished, indicating basal body docking defects. Conditional knockout mouse model, immunofluorescence, TEM, histology Reproduction (Cambridge, England) High 34085951
2022 Deletion of CEP164 post-ciliogenesis in rod photoreceptors impairs intraflagellar transport (IFT): IFT components IFT88, IFT57, and IFT140 were reduced at basal body and ciliary tip. CEP164 is also required for basal body docking to the apical membrane; retina-specific KO at embryonic stage prevents connecting cilium and outer segment formation. CEP164 is key for recruitment and stabilization of IFT-B particles at the basal body/connecting cilium. Conditional knockout mouse models (Six3Cre, iCre75, Prom1-ETCre tamoxifen-inducible), immunofluorescence, electron microscopy, fluorescent dye disc labeling PLoS genetics High 36074756
2023 CEP164 physically interacts with GLI2 transcription factor at the mother centriole. The GLI2-binding region of CEP164, when ectopically expressed, reduces centriolar GLI2 localization and enhances expression of Hedgehog target genes, both in cells with and without primary cilia, demonstrating a cilia-independent role for CEP164 in controlling Hh signaling at the mother centriole. Co-immunoprecipitation, domain mapping, ectopic expression of CEP164 binding region, immunofluorescence colocalization, RT-PCR for Hh target genes Biochemical and biophysical research communications Medium 37199136
2024 Osteoblast-specific deletion of Cep164 in mice causes bone development defects and an increased number of γH2AX-positive cells in osteoblasts, indicating defective DNA damage response contributes to skeletal pathology. Chondrocyte-specific deletion causes no overt skeletal abnormalities, revealing cell-type-specific CEP164 function. Mesodermal cell-specific deletion results in severe bone defects. Conditional knockout mouse models (mesodermal-, osteoblast-, and chondrocyte-specific Cre), immunofluorescence for γH2AX, histology Biochemical and biophysical research communications Medium 39612644
2025 CEP164 contains a long intrinsically disordered region and forms dynamic condensates with TTBK2 through liquid-liquid phase separation driven by multivalent electrostatic interactions. This phase separation facilitates efficient recruitment of TTBK2 to distal appendages to initiate ciliogenesis. Phase separation assays, electrostatic interaction analysis, live-cell imaging of condensates, TTBK2 recruitment assays, ciliogenesis rescue experiments Cell reports Medium 40483689
2025 CEP164 homodimerizes via its central coiled-coil region, which is required for its mother centriole localization and subsequent TTBK2 recruitment. TTBK2 kinase activity plus its interaction with CEP164 are both required for recruitment of IFT-A, IFT-B, and dynein-2 complexes to, and removal of CP110 from, the mother centriole. CP110 removal is not always coupled with IFT protein recruitment. CEP164-KO and TTBK2-KO cell lines, chimeric/domain construct expression, co-immunoprecipitation, immunofluorescence for IFT and CP110 Molecular biology of the cell High 40305080
2025 CEP164 at distal appendages is required for the enlargement of small vesicles docked to the mother centriole, a key trigger for ciliogenesis progression upstream of axoneme growth. These vesicles subsequently fuse to form tubular C-shaped and toroidal membrane intermediates that organize into the ciliary vesicle. Quantitative isotropic 3D ultrastructure imaging (focused ion beam SEM), protein localization, CEP164 loss-of-function bioRxivpreprint Medium

Source papers

Stage 0 corpus · 22 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2007 Cep164, a novel centriole appendage protein required for primary cilium formation. The Journal of cell biology 427 17954613
2012 Exome capture reveals ZNF423 and CEP164 mutations, linking renal ciliopathies to DNA damage response signaling. Cell 316 22863007
2012 ARL13B, PDE6D, and CEP164 form a functional network for INPP5E ciliary targeting. Proceedings of the National Academy of Sciences of the United States of America 199 23150559
2014 Cep164 triggers ciliogenesis by recruiting Tau tubulin kinase 2 to the mother centriole. Proceedings of the National Academy of Sciences of the United States of America 156 24982133
2008 Cep164 is a mediator protein required for the maintenance of genomic stability through modulation of MDC1, RPA, and CHK1. Genes & development 79 18283122
2014 Nephronophthisis-associated CEP164 regulates cell cycle progression, apoptosis and epithelial-to-mesenchymal transition. PLoS genetics 63 25340510
2014 Binding to Cep164, but not EB1, is essential for centriolar localization of TTBK2 and its function in ciliogenesis. Genes to cells : devoted to molecular & cellular mechanisms 55 25297623
2017 Conditional knockout mice for the distal appendage protein CEP164 reveal its essential roles in airway multiciliated cell differentiation. PLoS genetics 53 29244804
2009 UV-dependent interaction between Cep164 and XPA mediates localization of Cep164 at sites of DNA damage and UV sensitivity. Cell cycle (Georgetown, Tex.) 34 19197159
2016 CEP164-null cells generated by genome editing show a ciliation defect with intact DNA repair capacity. Journal of cell science 33 26966185
2021 CEP164 is essential for efferent duct multiciliogenesis and male fertility. Reproduction (Cambridge, England) 25 34085951
2022 Deletion of CEP164 in mouse photoreceptors post-ciliogenesis interrupts ciliary intraflagellar transport (IFT). PLoS genetics 20 36074756
2021 Molecular mechanisms underlying the role of the centriolar CEP164-TTBK2 complex in ciliopathies. Structure (London, England : 1993) 20 34499853
2019 Roscovitine blocks collecting duct cyst growth in Cep164-deficient kidneys. Kidney international 15 31248650
2020 CEP164 Deficiency Causes Hyperproliferation of Pancreatic Cancer Cells. Frontiers in cell and developmental biology 13 33251215
2020 Embryonic and foetal expression patterns of the ciliopathy gene CEP164. PloS one 8 31990917
2022 Biallelic variants in CEP164 cause a motile ciliopathy-like syndrome. Clinical genetics 7 36273371
2023 CEP164-GLI2 association ensures the hedgehog signaling in pancreatic cancer cells. Biochemical and biophysical research communications 3 37199136
2025 Phase separation of TTBK2 and CEP164 is necessary for ciliogenesis. Cell reports 2 40483689
2025 Coordinated roles of the CEP164 homodimer and TTBK2 are required for recruitment of the IFT machinery to the mother centriole for ciliogenesis. Molecular biology of the cell 1 40305080
2024 Disruption of distal appendage protein CEP164 causes skeletal malformation in mice. Biochemical and biophysical research communications 1 39612644
2026 Ameloblastoma Displays Primary Cilia Maintenance and CEP164 Overexpression. Journal of dental research 0 41733187

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