CEP97 (also LRRIQ2) is a centrosomal protein with an N-terminal leucine-rich repeat domain and an IQ motif. It is the principal binding partner of CP110 and is required to recruit and stabilize CP110 at the distal ends of centrioles. Together CP110 and CEP97 form a cap on the distal end of the mother centriole that suppresses primary cilium formation in proliferating cells; the cap is anchored through a direct interaction between CEP97 and MPHOSPH9, which is itself recruited by KIF24, and it is removed after TTBK2 activity at the onset of ciliogenesis. CEP97 does not bind microtubules directly, unlike CP110. Depletion of CEP97 causes CP110 loss from centrosomes, spindle defects, polyploidy and inappropriate cilium formation in growing cells. CEP97 also contributes, with CP110 and CEP290, to centrosomal aggresome assembly under proteotoxic stress, and in multiciliated cells of Xenopus it acts in centriole disengagement from deuterosomes.
| GO Term | Evidence | Action | Reason |
|---|---|---|---|
| GO:0005515 protein binding | IPI PMID:17719545 Cep97 and CP110 suppress a cilia assembly program. | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:18694559 CP110 suppresses primary cilia formation through its interac... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:19460342 Cep76, a centrosomal protein that specifically restrains cen... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:21620453 Centriolar kinesin Kif24 interacts with CP110 to remodel mic... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:21620453 Centriolar kinesin Kif24 interacts with CP110 to remodel mic... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:22441691 Neurl4, a novel daughter centriole protein, prevents formati... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:24421332 The CP110-interacting proteins Talpid3 and Cep290 play overl... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:24421332 The CP110-interacting proteins Talpid3 and Cep290 play overl... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:24981860 Human-chromatin-related protein interactions identify a deme... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:26496610 A human interactome in three quantitative dimensions organiz... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:26638075 A Dynamic Protein Interaction Landscape of the Human Centros... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:27173435 An organelle-specific protein landscape identifies novel dis... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:27173435 An organelle-specific protein landscape identifies novel dis... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:30375385 M-Phase Phosphoprotein 9 regulates ciliogenesis by modulatin... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:33961781 Dual proteome-scale networks reveal cell-specific remodeling... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005515 protein binding | IPI PMID:35709258 Spatial centrosome proteome of human neural cells uncovers d... | REMOVE | Summary: Generic protein binding (IPI) for CEP97. Partners include CP110 (its main partner), CEP76, KIF24, Talpid3 and interactome hits; the CP110 recruitment role is captured by the adaptor and process terms. Reason: GO:0005515 protein binding is uninformative about the molecular activity of the gene product. Removal does not dispute that the reported physical interaction occurs; the biologically meaningful consequences of the interaction are captured by the process, location and more specific function terms reviewed elsewhere in this document. |
| GO:0005813 centrosome | IBA GO_REF:0000033 | ACCEPT | Summary: Centrosome localization (IBA). Reason: CEP97 localizes to centrosomes, at the distal ends of centrioles, where it recruits CP110. Supporting Evidence: PMID:17719545 identified a previously uncharacterized protein, Cep97, that recruits CP110 to centrosomes file:human/CEP97/CEP97-deep-research-falcon.md Its localization persists after cytoplasmic microtubule depolymerization, supporting an intrinsic centrosomal association. |
| GO:0005813 centrosome | IDA GO_REF:0000052 | ACCEPT | Summary: Centrosome localization from the Human Protein Atlas immunofluorescence (IDA, GO_REF:0000052). Reason: HPA reports CEP97 at the centrosome (Supported) and at the basal body and centriolar satellites (Approved), consistent with its established distal centriole localization. |
| GO:0005813 centrosome | IDA PMID:17719545 Cep97 and CP110 suppress a cilia assembly program. | ACCEPT | Summary: Centrosome localization (IDA). Reason: CEP97 localizes to centrosomes, at the distal ends of centrioles, where it recruits CP110. Supporting Evidence: PMID:17719545 identified a previously uncharacterized protein, Cep97, that recruits CP110 to centrosomes file:human/CEP97/CEP97-deep-research-falcon.md Its localization persists after cytoplasmic microtubule depolymerization, supporting an intrinsic centrosomal association. |
| GO:0005813 centrosome | IDA PMID:21399614 Novel asymmetrically localizing components of human centroso... | ACCEPT | Summary: Centrosome localization (IDA). Reason: CEP97 localizes to centrosomes, at the distal ends of centrioles, where it recruits CP110. Supporting Evidence: PMID:17719545 identified a previously uncharacterized protein, Cep97, that recruits CP110 to centrosomes file:human/CEP97/CEP97-deep-research-falcon.md Its localization persists after cytoplasmic microtubule depolymerization, supporting an intrinsic centrosomal association. |
| GO:0005813 centrosome | IDA PMID:30375385 M-Phase Phosphoprotein 9 regulates ciliogenesis by modulatin... | ACCEPT | Summary: Centrosome localization (IDA). Reason: CEP97 localizes to centrosomes, at the distal ends of centrioles, where it recruits CP110. Supporting Evidence: PMID:17719545 identified a previously uncharacterized protein, Cep97, that recruits CP110 to centrosomes file:human/CEP97/CEP97-deep-research-falcon.md Its localization persists after cytoplasmic microtubule depolymerization, supporting an intrinsic centrosomal association. |
| GO:0005813 centrosome | IEA GO_REF:0000120 | ACCEPT | Summary: Centrosome localization (IEA). Reason: CEP97 localizes to centrosomes, at the distal ends of centrioles, where it recruits CP110. Supporting Evidence: PMID:17719545 identified a previously uncharacterized protein, Cep97, that recruits CP110 to centrosomes file:human/CEP97/CEP97-deep-research-falcon.md Its localization persists after cytoplasmic microtubule depolymerization, supporting an intrinsic centrosomal association. |
| GO:0005814 centriole | IBA GO_REF:0000033 | ACCEPT | Summary: Centriole localization (IBA). Reason: CEP97 is a distal centriole cap protein colocalizing with CP110, KIF24 and MPHOSPH9 at the distal end of the mother centriole. Supporting Evidence: PMID:30375385 Surprisingly, MPP9 appeared as a ring-like structure surrounding the centriole distal-end marker Centrin-3 and was colocalized with other distal-end markers, such as CP110, CEP97, and KIF24 |
| GO:0005814 centriole | IDA PMID:30375385 M-Phase Phosphoprotein 9 regulates ciliogenesis by modulatin... | ACCEPT | Summary: Centriole localization (IDA). Reason: CEP97 is a distal centriole cap protein colocalizing with CP110, KIF24 and MPHOSPH9 at the distal end of the mother centriole. Supporting Evidence: PMID:30375385 Surprisingly, MPP9 appeared as a ring-like structure surrounding the centriole distal-end marker Centrin-3 and was colocalized with other distal-end markers, such as CP110, CEP97, and KIF24 |
| GO:0005814 centriole | IEA GO_REF:0000044 | ACCEPT | Summary: Centriole localization (IEA). Reason: CEP97 is a distal centriole cap protein colocalizing with CP110, KIF24 and MPHOSPH9 at the distal end of the mother centriole. Supporting Evidence: PMID:30375385 Surprisingly, MPP9 appeared as a ring-like structure surrounding the centriole distal-end marker Centrin-3 and was colocalized with other distal-end markers, such as CP110, CEP97, and KIF24 |
| GO:0005829 cytosol | TAS Reactome:R-HSA-5626220 | KEEP AS NON CORE | Summary: Reactome-derived cytosol localization (TAS) for CEP97, from pathway reactions in which CEP97 participates. Reason: Reactome places soluble, non-membrane participants of its reactions in the cytosol compartment. This is not wrong in the broad sense, but the informative location of CEP97 is the distal end of centrioles; cytosol is retained as a non-core, low-information location. |
| GO:0005829 cytosol | TAS Reactome:R-HSA-5626223 | KEEP AS NON CORE | Summary: Reactome-derived cytosol localization (TAS) for CEP97, from pathway reactions in which CEP97 participates. Reason: Reactome places soluble, non-membrane participants of its reactions in the cytosol compartment. This is not wrong in the broad sense, but the informative location of CEP97 is the distal end of centrioles; cytosol is retained as a non-core, low-information location. |
| GO:0005829 cytosol | TAS Reactome:R-HSA-5626227 | KEEP AS NON CORE | Summary: Reactome-derived cytosol localization (TAS) for CEP97, from pathway reactions in which CEP97 participates. Reason: Reactome places soluble, non-membrane participants of its reactions in the cytosol compartment. This is not wrong in the broad sense, but the informative location of CEP97 is the distal end of centrioles; cytosol is retained as a non-core, low-information location. |
| GO:0005829 cytosol | TAS Reactome:R-HSA-5626228 | KEEP AS NON CORE | Summary: Reactome-derived cytosol localization (TAS) for CEP97, from pathway reactions in which CEP97 participates. Reason: Reactome places soluble, non-membrane participants of its reactions in the cytosol compartment. This is not wrong in the broad sense, but the informative location of CEP97 is the distal end of centrioles; cytosol is retained as a non-core, low-information location. |
| GO:0005829 cytosol | TAS Reactome:R-HSA-5626699 | KEEP AS NON CORE | Summary: Reactome-derived cytosol localization (TAS) for CEP97, from pathway reactions in which CEP97 participates. Reason: Reactome places soluble, non-membrane participants of its reactions in the cytosol compartment. This is not wrong in the broad sense, but the informative location of CEP97 is the distal end of centrioles; cytosol is retained as a non-core, low-information location. |
| GO:0030674 protein-macromolecule adaptor activity | IDA PMID:17719545 Cep97 and CP110 suppress a cilia assembly program. | ACCEPT | Summary: Protein-macromolecule adaptor activity (IDA) based on CEP97-dependent recruitment of CP110 to centrosomes. Reason: CEP97 binds CP110 and is required for CP110 centrosomal localization, and it links the CP110-CEP97 cap to MPHOSPH9 by direct binding, i.e. it bridges CP110 to the centriolar recruitment machinery. This is the most informative molecular function available for CEP97. Supporting Evidence: PMID:17719545 Depletion of Cep97 or expression of dominant-negative mutants results in CP110 disappearance from centrosomes PMID:30375385 recruits CP110-CEP97 by directly binding CEP97 |
| GO:0032991 protein-containing complex | IDA PMID:24421332 The CP110-interacting proteins Talpid3 and Cep290 play overl... | KEEP AS NON CORE | Summary: Protein-containing complex (IDA), association with CP110- and Talpid3-containing complexes. Reason: Correct but uninformative generic complex term; no specific GO complex term exists for the CP110-CEP97 distal cap. Supporting Evidence: PMID:24421332 Collectively, these data indicate that Talpid3 robustly binds to CP110 and more weakly associates with Cep97, Cep290, and Kif24. |
| GO:1901673 regulation of mitotic spindle assembly | IMP PMID:17719545 Cep97 and CP110 suppress a cilia assembly program. | KEEP AS NON CORE | Summary: Regulation of mitotic spindle assembly (IMP) from CEP97 depletion causing spindle defects. Reason: CEP97 depletion produces spindle defects and polyploidy, but this is most plausibly an indirect consequence of loss of CP110 and centrosome/cytokinesis defects rather than a direct role of CEP97 in spindle assembly. Retained as non-core. Supporting Evidence: PMID:17719545 Depletion of Cep97 or expression of dominant-negative mutants results in CP110 disappearance from centrosomes, spindle defects, and polyploidy. |
| GO:1902018 negative regulation of cilium assembly | IBA GO_REF:0000033 | ACCEPT | Summary: Negative regulation of cilium assembly (IBA). Reason: Core function: CEP97 with CP110 forms the distal cap that suppresses ciliogenesis in cycling cells; loss of CEP97 promotes cilia formation in growing cells. Supporting Evidence: PMID:17719545 loss of Cep97 or CP110 promotes primary cilia formation in growing cells PMID:30375385 The CP110-CEP97 complex functions as a negative regulator of ciliogenesis |
| GO:1902018 negative regulation of cilium assembly | IEA GO_REF:0000107 | ACCEPT | Summary: Negative regulation of cilium assembly (IEA). Reason: Core function: CEP97 with CP110 forms the distal cap that suppresses ciliogenesis in cycling cells; loss of CEP97 promotes cilia formation in growing cells. Supporting Evidence: PMID:17719545 loss of Cep97 or CP110 promotes primary cilia formation in growing cells PMID:30375385 The CP110-CEP97 complex functions as a negative regulator of ciliogenesis |
| GO:1902018 negative regulation of cilium assembly | IMP PMID:17719545 Cep97 and CP110 suppress a cilia assembly program. | ACCEPT | Summary: Negative regulation of cilium assembly (IMP). Reason: Core function: CEP97 with CP110 forms the distal cap that suppresses ciliogenesis in cycling cells; loss of CEP97 promotes cilia formation in growing cells. Supporting Evidence: PMID:17719545 loss of Cep97 or CP110 promotes primary cilia formation in growing cells PMID:30375385 The CP110-CEP97 complex functions as a negative regulator of ciliogenesis |
| GO:1902018 negative regulation of cilium assembly | IMP PMID:30375385 M-Phase Phosphoprotein 9 regulates ciliogenesis by modulatin... | ACCEPT | Summary: Negative regulation of cilium assembly (IMP). Reason: Core function: CEP97 with CP110 forms the distal cap that suppresses ciliogenesis in cycling cells; loss of CEP97 promotes cilia formation in growing cells. Supporting Evidence: PMID:17719545 loss of Cep97 or CP110 promotes primary cilia formation in growing cells PMID:30375385 The CP110-CEP97 complex functions as a negative regulator of ciliogenesis |
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Download this section (compressed HTML)Q: HPA detects CEP97 at centriolar satellites as well as the basal body and centrosome; does a satellite pool of CEP97 have a function distinct from the distal cap (for example in CP110 delivery or turnover)?
Q: CEP97 depletion lengthens centrioles in human cells although CEP97 does not bind microtubules; is this effect fully explained by loss of CP110, and should a centriole-length regulation process be annotated to CEP97 or only to CP110?
Experiment: Map the CP110-binding and MPHOSPH9-binding surfaces of CEP97 and test separation-of-function mutants for CP110 cap retention, centriole length and ciliation in RPE1 cells.
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