Affinage

CEP97

Centrosomal protein of 97 kDa · UniProt Q8IW35

Round 2 corrected
Length
865 aa
Mass
97.0 kDa
Annotated
2026-04-28
63 papers in source corpus 24 papers cited in narrative 24 extracted findings

Mechanistic narrative

Synthesis pass · prose summary of the discoveries below

CEP97 is a centriole distal-end capping protein that, together with CP110, forms an inhibitory cap complex suppressing inappropriate ciliogenesis in cycling cells. CEP97 recruits CP110 to centrosomes and is itself recruited via direct binding to MPP9, which is positioned by KIF24; removal of this cap at ciliogenesis onset is triggered by TTBK2-mediated phosphorylation and proteasomal degradation of MPP9, LUBAC-dependent linear ubiquitination of CP110, competitive displacement by ENKD1, and satellite-mediated displacement by PCM1 (PMID:17719545, PMID:30375385, PMID:34813648, PMID:35301795, PMID:36790165). Beyond its ciliogenesis-gating role, CEP97 functions as the central scaffold of the Drosophila distal tip complex required for centriole length control and structural integrity in cooperation with the microtubule acetylation machinery, and in vertebrate multiciliated cells it is phosphorylated by Dyrk1a to recruit Plk1 and promote Separase-mediated centriole disengagement (PMID:32589908, PMID:37729913, PMID:34787650). CEP97 re-capping of mother centrioles in post-mitotic cerebellar neurons enforces permanent loss of ciliation, and its levels can be regulated post-transcriptionally by miR-106b-5p to modulate ciliogenesis in astrocytes (PMID:39705308, PMID:37012704).

Mechanistic history

Synthesis pass · year-by-year structured walk · 12 steps
  1. 2007 High

    The foundational question of how primary cilia formation is suppressed in proliferating cells was answered by demonstrating that CEP97 recruits CP110 to centrosomes and that their co-depletion triggers ectopic ciliogenesis, establishing the CEP97–CP110 capping model.

    Evidence Protein complex purification, reciprocal co-IP, siRNA depletion, and dominant-negative expression in human cell lines

    PMID:17719545

    Open questions at the time
    • Mechanism by which CEP97 itself is recruited to the centriole distal end was unknown
    • Whether removal of the cap is an active regulated process or passive consequence of cell-cycle exit was unresolved
    • No structural information on CEP97 or the CEP97–CP110 interface
  2. 2008 High

    The CP110 capping complex was shown to be modular: CEP290 forms a distinct subcomplex with CP110 required for cilia suppression and linking the cap to Rab8a vesicle trafficking, expanding the cap's functional reach beyond simple physical obstruction.

    Evidence Co-immunoprecipitation and siRNA depletion with ciliogenesis and Rab8a localization readouts

    PMID:18694559

    Open questions at the time
    • Whether CEP97 and CEP290 bind CP110 simultaneously or mutually exclusively was not resolved
    • No direct binding assay between CEP97 and CEP290
  3. 2011 High

    KIF24, a depolymerizing kinesin-13, was identified as a mother-centriole-specific interactor of CP110 and CEP97 whose loss causes CP110 displacement and ectopic ciliogenesis, revealing that active microtubule remodeling maintains the cap.

    Evidence Co-IP, siRNA depletion, in vitro microtubule depolymerization assay in human cells

    PMID:21620453

    Open questions at the time
    • Whether KIF24 directly contacts CEP97 or acts only through CP110 was not distinguished
    • Mechanism linking microtubule depolymerization to cap retention was unclear
  4. 2014 High

    TTBK2 was placed as the kinase that triggers ciliogenesis initiation by phosphorylating CEP97 and CEP164, with CEP164-dependent centriolar recruitment of TTBK2 required for CP110/CEP97 removal, establishing the first enzymatic trigger of cap displacement.

    Evidence In vitro kinase assay, TTBK2 binding-motif mutagenesis, siRNA rescue in human cells

    PMID:25297623

    Open questions at the time
    • Direct phosphorylation sites on CEP97 were not mapped
    • Whether TTBK2 phosphorylation of CEP97 itself is sufficient for cap removal was untested
  5. 2016 High

    Biophysical characterization of the CEP104–CP110–CEP97 interaction network and crystal structure of the CEP104 TOG domain defined a microtubule-EB-CEP104-tubulin-CP110 bridge, placing CEP97 within a structurally resolved centriole distal-end interaction network.

    Evidence X-ray crystallography, ITC, and SEC binding assays

    PMID:27402853

    Open questions at the time
    • No atomic-resolution structure of CEP97 itself or the CEP97–CP110 interface
    • Functional consequence of disrupting the CEP97–CEP104 interaction in cells was not tested
  6. 2018 High

    The recruitment hierarchy was resolved: MPP9 directly binds CEP97 and is required to recruit the CP110–CEP97 cap, while TTBK2-mediated phosphorylation of MPP9 triggers its proteasomal degradation and consequent cap loss, unifying the kinase signal with cap displacement.

    Evidence Reciprocal co-IP, super-resolution microscopy, phosphorylation assays, and mouse kidney validation

    PMID:30375385

    Open questions at the time
    • Whether MPP9 phosphodegron is the sole route for cap removal or acts in parallel with other mechanisms
    • Structural basis of the MPP9–CEP97 interaction is unknown
  7. 2020 High

    Drosophila null mutants revealed that Cep97 is essential for full-length centriole formation and ciliogenesis in flies, and identified Sirt2 (a microtubule deacetylase) as a Cep97 interactor, linking the cap to centriole structural integrity via tubulin acetylation.

    Evidence Drosophila null mutant analysis, co-IP, electron microscopy across multiple tissue types

    PMID:32589908

    Open questions at the time
    • Whether tubulin acetylation directly stabilizes CEP97 binding or acts indirectly through centriole architecture
    • Relevance of Sirt2–CEP97 interaction in vertebrates was untested
  8. 2021 High

    Two parallel cap-removal mechanisms were identified: LUBAC-mediated linear ubiquitination of CP110 (read by PRPF8) and competitive displacement of CEP97 by ENKD1, revealing that cap disassembly is a multi-pathway convergence rather than a single trigger.

    Evidence Ubiquitination assays, co-IP, super-resolution microscopy, knockout mouse models, double-KD epistasis

    PMID:34813648 PMID:35301795

    Open questions at the time
    • How LUBAC and ENKD1 pathways are temporally coordinated during ciliogenesis
    • Whether ENKD1 competition and TTBK2/MPP9 degradation are independent or redundant
  9. 2021 High

    In multiciliated cells, CEP97 was shown to have a ciliogenesis-promoting function: Dyrk1a phosphorylates CEP97 to recruit Plk1, which activates Separase-mediated centriole disengagement, revealing a context-dependent reversal of CEP97's canonical cilia-inhibitory role.

    Evidence In vitro kinase assay, morpholino knockdown and Separase rescue in Xenopus multiciliated epithelium

    PMID:34787650

    Open questions at the time
    • Phosphorylation sites on CEP97 targeted by Dyrk1a were not mapped
    • Whether this pathway operates in mammalian multiciliated cells is untested
  10. 2022 Medium

    Cell-cycle-entrained oscillation of CP110/Cep97 levels at the distal end was shown to influence Plk4 dynamics and cartwheel growth at the proximal end in Drosophila, establishing unexpected crosstalk between distal capping and proximal centriole duplication.

    Evidence Live imaging and genetic manipulation in Drosophila embryos with quantitative fluorescence analysis

    PMID:35707992

    Open questions at the time
    • Molecular mechanism linking distal-end CP110/Cep97 levels to proximal Plk4 regulation is unknown
    • Whether this crosstalk occurs in vertebrate cells
  11. 2023 Medium

    Drosophila proximity labeling established Cep97 as the central scaffold of the distal tip complex, required for recruiting Cep104 and other DTC components, and showed Cep97–Cep104 cooperation is essential for spermatid alignment during spermiogenesis.

    Evidence Proximity-labeling screen, null mutant analysis, EM in Drosophila testes

    PMID:37729913

    Open questions at the time
    • Whether CEP97's scaffolding role in the DTC is conserved in vertebrates
    • Binding interfaces between Cep97 and DTC components remain unmapped
  12. 2024 Medium

    In vivo evidence from mouse cerebellar granule neurons showed that re-recruitment of CEP97 to the mother centriole accompanies permanent cilia loss during neuronal differentiation, extending the capping model to irreversible fate decisions in post-mitotic cells.

    Evidence Single-cell transcriptomics, confocal imaging, and immunocytology of mouse cerebellar tissue

    PMID:39705308

    Open questions at the time
    • Signal that triggers CEP97 re-capping in differentiating neurons is unknown
    • Whether preventing CEP97 re-capping restores ciliogenesis competence in these neurons

Open questions

Synthesis pass · forward-looking unresolved questions
  • No atomic-resolution structure of CEP97 or the CEP97–CP110 interface exists, and the precise phosphorylation sites on CEP97 targeted by TTBK2 and Dyrk1a have not been mapped; how the multiple parallel cap-removal pathways are coordinated temporally and whether CEP97's scaffolding role in the Drosophila DTC is conserved in vertebrates remain open questions.
  • No structure of CEP97 or CEP97–CP110 complex
  • Phosphosite mapping on CEP97 is incomplete
  • Temporal coordination of parallel cap-removal pathways is unresolved

Mechanism profile

Synthesis pass · controlled-vocabulary classification · explore literature graph →
Molecular activity
GO:0060090 molecular adaptor activity 3 GO:0005198 structural molecule activity 2 GO:0008092 cytoskeletal protein binding 2
Localization
GO:0005815 microtubule organizing center 7 GO:0005929 cilium 3
Pathway
R-HSA-1852241 Organelle biogenesis and maintenance 6 R-HSA-1640170 Cell Cycle 2
Partners
CEP104CEP290CP110ENKD1KIF24MPP9SIRT2TTBK2
Complex memberships
CP110–CEP97 capping complexCP110–CEP97–CEP290 complexDistal tip complex (Drosophila)

Evidence

Reading pass · 24 per-paper findings extracted from the source corpus
Year Finding Method Journal Conf PMIDs
2007 CEP97 recruits CP110 to centrosomes; depletion of CEP97 causes CP110 disappearance from centrosomes, spindle defects, and polyploidy. Loss of CEP97 or CP110 promotes primary cilia formation in growing cells, while enforced expression of CP110 in quiescent cells suppresses ciliogenesis, establishing that CEP97 and CP110 collaborate to inhibit a ciliogenesis program. Protein complex purification, co-immunoprecipitation, siRNA depletion, dominant-negative expression, live imaging Cell High 17719545
2008 CP110 interacts with CEP290 in a complex separable from other CP110 complexes; this CEP290-CP110 interaction is required for CP110-mediated suppression of primary cilia formation. CEP290 also interacts with Rab8a, and CEP290 depletion disrupts Rab8a localization to centrosomes/cilia. Co-immunoprecipitation, siRNA depletion, immunofluorescence Developmental cell High 18694559
2011 Kif24, a kinesin-13 subfamily motor protein, specifically interacts with CP110 and CEP97 at the mother centriole, preferentially localizing to mother centrioles. Loss of Kif24 causes CP110 disappearance from mother centrioles in cycling cells and aberrant cilia assembly. Kif24 can bind and depolymerize microtubules in vitro and specifically remodels centriolar microtubules when ectopically expressed. Co-immunoprecipitation, siRNA depletion, in vitro microtubule depolymerization assay, immunofluorescence Cell High 21620453
2012 CEP104 interacts with CP110 and CEP97 at the centriole and is required for ciliogenesis; CEP104 was identified as a microtubule plus-end tracking protein containing an SxIP motif. Proteome-wide SxIP motif screen, co-immunoprecipitation, siRNA depletion, fluorescence imaging Current biology : CB Medium 22885064
2013 MARK4 kinase is required for initiation of axoneme extension; upon MARK4 or ODF2 knockdown, the ciliary program arrests before complete removal of the CP110-CEP97 inhibitory complex from the mother centriole, placing MARK4 upstream of CP110-CEP97 removal in the ciliogenesis pathway. RNAi screen, siRNA depletion, ultrastructural analysis, immunofluorescence, co-immunoprecipitation The Journal of cell biology Medium 23400999
2014 TTBK2 phosphorylates CEP97 and CEP164 in vitro. Cep164 binding (not EB1) is essential for centriolar localization of TTBK2 and for CP110/CEP97 removal and ciliogenesis initiation. In vitro kinase assay, mutagenesis of TTBK2 binding motifs, siRNA rescue experiments, immunofluorescence Genes to cells : devoted to molecular & cellular mechanisms High 25297623
2015 In cytotoxic T lymphocytes (CTLs), CP110 and CEP97 remain associated with the mother centriole and neither axoneme nor transition zone ciliary structures form, demonstrating that CP110/CEP97 retention at the mother centriole prevents ciliogenesis during centrosome docking at the immunological synapse. High-resolution TEM tomography, immunofluorescence, siRNA depletion Current biology : CB Medium 26670998
2016 Crystal structure of the CEP104 TOG domain was solved; biophysical characterization defined the direct interaction network between CEP104, CP110, CEP97, EB protein, and tubulin, establishing CEP97 as part of a microtubule-EB-CEP104-tubulin-CP110 network at the centriole. X-ray crystallography, biophysical binding assays (ITC, SEC), sequence analysis The Journal of biological chemistry High 27402853
2016 Asterless (Drosophila ortholog of Cep152) controls centriole length via the centriole protein Cep97; in Asl-free centrioles, Cep97 function in length control is revealed as a duplication-independent role. Drosophila genetics, immunofluorescence, loss-of-function analysis in specific cell populations The Journal of cell biology Medium 27185836
2018 MPP9 is recruited by KIF24 to the distal end of the mother centriole where it forms a ring-like structure and directly binds CEP97 to recruit the CP110-CEP97 complex. Upon TTBK2-mediated phosphorylation of MPP9 at the onset of ciliogenesis, MPP9 is degraded via the ubiquitin-proteasome system, facilitating removal of CP110 and CEP97 from the mother centriole distal end. Co-immunoprecipitation, super-resolution microscopy, siRNA depletion, phosphorylation assays, mouse kidney models Nature communications High 30375385
2018 Truncated SALL1 (produced by TBS-causing mutations) interacts with CP110 and CEP97 as identified by proximity proteomics, leading to more frequent cilia formation in TBS-derived fibroblasts and altered SHH signaling. Proximity proteomics (BioID), CRISPR/Cas9 cell line generation, immunofluorescence, SHH signaling assays American journal of human genetics Medium 29395072
2020 In Drosophila, Cep97 is essential for formation of full-length centrioles and for ciliogenesis, acting as part of a protective cap at the centriole distal end. The microtubule deacetylase Sirt2 was identified as a Cep97 interactor; both Sirt2 deletion and Atat1 (acetylase) deletion impair centriole integrity, indicating Cep97 cooperates with the microtubule acetylation machinery to maintain centriole stability. Drosophila genetics (null mutants), co-immunoprecipitation, immunofluorescence, electron microscopy Current biology : CB High 32589908
2021 Dyrk1a phosphorylates CEP97, which promotes recruitment of Polo-like kinase 1 (Plk1) to multiciliated cells. CEP97 and Dyrk1a coordinate with Plk1 to promote Separase function for centriole disengagement during multiciliogenesis; knockdown of CEP97 or Dyrk1a disrupts cilia formation and centriole disengagement, rescued by Separase overexpression. Xenopus embryo model, in vitro kinase assay, morpholino knockdown, overexpression rescue, immunofluorescence The Journal of cell biology High 34787650
2021 LUBAC generates linear ubiquitin chains specifically on CP110, and a pre-mRNA splicing factor PRPF8 at the distal end of the mother centriole acts as receptor for these linear ubiquitin chains to facilitate CP110 (and consequently CEP97) removal at the initial stage of ciliogenesis. Co-immunoprecipitation, ubiquitination assay, siRNA depletion, immunofluorescence The Journal of cell biology Medium 34813648
2022 ENKD1 competes with CEP97 for binding to CP110; depletion of ENKD1 enhances the CP110-CEP97 interaction and detains CP110 at the mother centriole, inhibiting ciliogenesis. Simultaneous knockdown of ENKD1 and CP110 reverses the ciliogenesis defect, placing ENKD1 as a promoter of CP110 removal by competing with CEP97. Co-immunoprecipitation, super-resolution microscopy, siRNA/knockout mouse models, genetic epistasis (double knockdown) EMBO reports High 35301795
2022 The seeding of a phosphorylated HSP27 ring around centrioles during aggresome assembly depends on CP110, CEP97, and CEP290; satellites and the CP110-CEP97-CEP290 complex are required for aggresome growth and for aggregation of mutant huntingtin. siRNA depletion, high-resolution quantitative imaging, immunofluorescence, proteasome inhibitor treatment Nature cell biology Medium 35411088
2022 WDR8 and Cep135 are required for both ciliary vesicle docking to the mother centriole and for removing the CP110-CEP97 inhibitory complex from the mother centriole distal end, establishing a role for centriolar proximal-end proteins upstream of CP110-CEP97 displacement in ciliogenesis. siRNA depletion, immunofluorescence, electron microscopy Journal of cell science Medium 26675238
2022 In Drosophila embryos, CP110 and Cep97 form a complex at the distal end of centrioles whose levels oscillate with the cell cycle, entrained by the Cdk-Cyclin oscillator. Changing CP110/Cep97 levels alters Plk4 oscillation and cartwheel growth at the proximal end, revealing unexpected crosstalk between distal-end capping factors and proximal-end cartwheel assembly. Drosophila embryo live imaging, genetic manipulation (overexpression/depletion), quantitative fluorescence analysis Journal of cell science Medium 35707992
2023 PCM1 and centriolar satellites facilitate removal of CP110 and CEP97 from the distal mother centriole to initiate ciliogenesis; Pcm1-null RPE1 cells show reduced docking of the mother centriole to the ciliary vesicle and reduced removal of CP110/CEP97. Mouse knockout, siRNA depletion in human RPE1 cells, immunofluorescence, single-cell transcriptomics eLife Medium 36790165
2023 In Drosophila, Cep97 is the central scaffolding unit required to recruit distal tip complex (DTC) components including Cep104 to the distal tip of centrioles; Cep97 and Cep104 cooperate during spermiogenesis to align spermatids and coordinate individualization. Drosophila proximity-labeling screen, null mutant analysis, immunofluorescence, electron microscopy Current biology : CB Medium 37729913
2023 miR-106b-5p in morphine-stimulated astrocyte-derived extracellular vesicles induces primary ciliogenesis by targeting CEP97 mRNA, reducing CEP97 levels; delivery of anti-miR-106b restored CEP97 expression, inhibited ciliogenesis, and prevented morphine tolerance in mice. miRNA target validation, EV delivery experiments, mouse morphine tolerance model, immunofluorescence, western blot Molecular therapy Medium 37012704
2024 During cerebellar granule cell differentiation, mother centrioles in maturing neurons recruit CEP97 (centriole cap complex) while cilia disassemble and cells permanently lose the ability to re-ciliate, indicating that CEP97 re-capping of docked mother centrioles prevents cilia regrowth in post-mitotic neurons. Single-cell transcriptomics, immunocytology, confocal imaging of mouse cerebellar tissue Proceedings of the National Academy of Sciences of the United States of America Medium 39705308
2022 CUL3/KCTD10 E3 ubiquitin ligase complex ubiquitinates CEP97, mediating its degradation and thereby influencing primary cilium formation. Ubiquitination assay, co-immunoprecipitation, siRNA depletion BioEssays Low 32484264
2025 EHD1, through its membrane tubulation function, directly promotes CP110/CEP97 removal from the mother centriole cap during ciliogenesis, linking membrane vesicle dynamics to CP110-CEP97 displacement. 3D electron microscopy (isotropic ultrastructure imaging), siRNA depletion, quantitative analysis bioRxivpreprint Medium 40894589

Source papers

Stage 0 corpus · 63 papers · ranked by NIH iCite citations
Year Title Journal Citations PMID
2002 Generation and initial analysis of more than 15,000 full-length human and mouse cDNA sequences. Proceedings of the National Academy of Sciences of the United States of America 1479 12477932
2015 The BioPlex Network: A Systematic Exploration of the Human Interactome. Cell 1118 26186194
2017 Architecture of the human interactome defines protein communities and disease networks. Nature 1085 28514442
2015 A human interactome in three quantitative dimensions organized by stoichiometries and abundances. Cell 1015 26496610
2018 VIRMA mediates preferential m6A mRNA methylation in 3'UTR and near stop codon and associates with alternative polyadenylation. Cell discovery 829 29507755
2003 Complete sequencing and characterization of 21,243 full-length human cDNAs. Nature genetics 754 14702039
2021 Dual proteome-scale networks reveal cell-specific remodeling of the human interactome. Cell 705 33961781
2011 Phylogenetic-based propagation of functional annotations within the Gene Ontology consortium. Briefings in bioinformatics 656 21873635
2018 High-Density Proximity Mapping Reveals the Subcellular Organization of mRNA-Associated Granules and Bodies. Molecular cell 580 29395067
1994 Oligo-capping: a simple method to replace the cap structure of eukaryotic mRNAs with oligoribonucleotides. Gene 492 8125298
2004 The status, quality, and expansion of the NIH full-length cDNA project: the Mammalian Gene Collection (MGC). Genome research 438 15489334
2015 A Dynamic Protein Interaction Landscape of the Human Centrosome-Cilium Interface. Cell 433 26638075
2022 OpenCell: Endogenous tagging for the cartography of human cellular organization. Science (New York, N.Y.) 432 35271311
2010 Systematic analysis of human protein complexes identifies chromosome segregation proteins. Science (New York, N.Y.) 421 20360068
2005 Diversification of transcriptional modulation: large-scale identification and characterization of putative alternative promoters of human genes. Genome research 409 16344560
2007 Cep97 and CP110 suppress a cilia assembly program. Cell 385 17719545
2009 Mammalian BTBD12/SLX4 assembles a Holliday junction resolvase and is required for DNA repair. Cell 375 19596235
2021 A proximity-dependent biotinylation map of a human cell. Nature 339 34079125
2014 A quantitative chaperone interaction network reveals the architecture of cellular protein homeostasis pathways. Cell 325 25036637
2018 Enhancer Activity Requires CBP/P300 Bromodomain-Dependent Histone H3K27 Acetylation. Cell reports 274 30110629
2011 Novel asymmetrically localizing components of human centrosomes identified by complementary proteomics methods. The EMBO journal 265 21399614
2008 CP110 suppresses primary cilia formation through its interaction with CEP290, a protein deficient in human ciliary disease. Developmental cell 220 18694559
2021 Trans-ancestry analysis reveals genetic and nongenetic associations with COVID-19 susceptibility and severity. Nature genetics 211 33888907
2016 An organelle-specific protein landscape identifies novel diseases and molecular mechanisms. Nature communications 211 27173435
2015 A deep proteomics perspective on CRM1-mediated nuclear export and nucleocytoplasmic partitioning. eLife 198 26673895
2012 A Proteome-wide screen for mammalian SxIP motif-containing microtubule plus-end tracking proteins. Current biology : CB 175 22885064
2013 The protein interaction landscape of the human CMGC kinase group. Cell reports 174 23602568
2011 Centriolar kinesin Kif24 interacts with CP110 to remodel microtubules and regulate ciliogenesis. Cell 165 21620453
2014 Proximity interactions among centrosome components identify regulators of centriole duplication. Current biology : CB 162 24613305
2019 Mapping the proximity interaction network of the Rho-family GTPases reveals signalling pathways and regulatory mechanisms. Nature cell biology 137 31871319
2020 PROTAC-mediated degradation reveals a non-catalytic function of AURORA-A kinase. Nature chemical biology 129 32989298
2017 The human cytoplasmic dynein interactome reveals novel activators of motility. eLife 118 28718761
2007 Toward a confocal subcellular atlas of the human proteome. Molecular & cellular proteomics : MCP 114 18029348
2013 The microtubule affinity regulating kinase MARK4 promotes axoneme extension during early ciliogenesis. The Journal of cell biology 69 23400999
2018 M-Phase Phosphoprotein 9 regulates ciliogenesis by modulating CP110-CEP97 complex localization at the mother centriole. Nature communications 67 30375385
2015 Mother Centriole Distal Appendages Mediate Centrosome Docking at the Immunological Synapse and Reveal Mechanistic Parallels with Ciliogenesis. Current biology : CB 61 26670998
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
2018 LRRC45 contributes to early steps of axoneme extension. Journal of cell science 42 30131441
2023 Centriolar satellites expedite mother centriole remodeling to promote ciliogenesis. eLife 37 36790165
2015 TTBK2: a tau protein kinase beyond tau phosphorylation. BioMed research international 36 25950000
2022 Aggresome assembly at the centrosome is driven by CP110-CEP97-CEP290 and centriolar satellites. Nature cell biology 33 35411088
2018 Truncated SALL1 Impedes Primary Cilia Function in Townes-Brocks Syndrome. American journal of human genetics 32 29395072
2015 WDR8 is a centriolar satellite and centriole-associated protein that promotes ciliary vesicle docking during ciliogenesis. Journal of cell science 32 26675238
2016 Biophysical and Structural Characterization of the Centriolar Protein Cep104 Interaction Network. The Journal of biological chemistry 31 27402853
2022 Genome-wide association studies of COVID-19: Connecting the dots. Infection, genetics and evolution : journal of molecular epidemiology and evolutionary genetics in infectious diseases 30 36280088
2016 Asterless is required for centriole length control and sperm development. The Journal of cell biology 29 27185836
2020 Cep97 Is Required for Centriole Structural Integrity and Cilia Formation in Drosophila. Current biology : CB 27 32589908
2022 ENKD1 promotes CP110 removal through competing with CEP97 to initiate ciliogenesis. EMBO reports 26 35301795
2022 A modified TurboID approach identifies tissue-specific centriolar components in C. elegans. PLoS genetics 26 35442950
2021 LUBAC regulates ciliogenesis by promoting CP110 removal from the mother centriole. The Journal of cell biology 21 34813648
2023 Extracellular vesicle-mediated delivery of anti-miR-106b inhibits morphine-induced primary ciliogenesis in the brain. Molecular therapy : the journal of the American Society of Gene Therapy 15 37012704
2014 The nuclear pool of tetraspanin CD9 contributes to mitotic processes in human breast carcinoma. Molecular cancer research : MCR 13 25103498
2020 KCTD10 Biology: An Adaptor for the Ubiquitin E3 Complex Meets Multiple Substrates: Emerging Divergent Roles of the cullin-3/KCTD10 E3 Ubiquitin Ligase Complex in Various Cell Lines. BioEssays : news and reviews in molecular, cellular and developmental biology 12 32484264
2022 Centriole distal-end proteins CP110 and Cep97 influence centriole cartwheel growth at the proximal end. Journal of cell science 11 35707992
2024 Permanent cilia loss during cerebellar granule cell neurogenesis involves withdrawal of cilia maintenance and centriole capping. Proceedings of the National Academy of Sciences of the United States of America 10 39705308
2021 CEP97 phosphorylation by Dyrk1a is critical for centriole separation during multiciliogenesis. The Journal of cell biology 8 34787650
2023 Cep104 is a component of the centriole distal tip complex that regulates centriole growth and contributes to Drosophila spermiogenesis. Current biology : CB 4 37729913
2025 Disrupted glucocorticoid receptor cell signalling causes a ciliogenesis defect in the fetal mouse renal tubule. EMBO reports 1 40247090
2025 CEP76 impairment at the centrosome-cilium interface contributes to a spectrum of ciliopathies. Science advances 1 41105778
2026 Visualizing Drosophila centrioles by expansion microscopy. Journal of cell science 0 41347334
2025 Epigenetic age acceleration and methylation differences in IgG4-related cholangitis and primary sclerosing cholangitis. Clinical epigenetics 0 39819503
2025 The interplay between Wnt and mTOR signaling modulates ciliogenesis in human retinal epithelial cells. PLoS biology 0 40892854
2025 Characterization of membrane structures regulating primary ciliogenesis by quantitative isotropic ultrastructure imaging. bioRxiv : the preprint server for biology 0 40894589