| 2002 |
CP110 is phosphorylated by CDKs (CDK2) in vitro and in vivo, localizes to centrosomes, and is required for centrosome duplication; RNAi-mediated depletion inhibits centrosome duplication, and long-term disruption of CP110 phosphorylation leads to unscheduled centrosome separation and polyploidy. |
CDK substrate screen, in vitro kinase assay, immunofluorescence localization, RNAi knockdown with centrosome phenotype readout |
Developmental cell |
High |
12361598
|
| 2006 |
CP110 directly interacts with calmodulin (CaM) and centrin in vivo; CP110 exists in large complexes (~300 kDa to 3 MDa) containing both centrin and CaM; CP110 depletion or expression of a CaM-binding-deficient CP110 mutant causes failure at a late stage of cytokinesis and binucleate cell formation. |
Yeast two-hybrid, co-immunoprecipitation, in vitro binding assay, RNAi knockdown, expression of dominant-negative mutant |
Molecular biology of the cell |
High |
16760425
|
| 2007 |
CP110 forms a complex with Cep97, which 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, and enforced CP110 expression in quiescent cells suppresses cilia assembly, demonstrating that Cep97 and CP110 collaborate to inhibit ciliogenesis. |
Biochemical complex purification, RNAi knockdown, dominant-negative expression, immunofluorescence, cilia formation assay |
Cell |
High |
17719545
|
| 2008 |
CP110 interacts with CEP290 in a discrete complex separable from other CP110 complexes; interaction with CEP290 is required for CP110's ability to suppress primary cilia formation. CEP290 and CP110 also interact with Rab8a; depletion of CEP290 prevents ciliogenesis and mislocalizes Rab8a at centrosomes and cilia. |
Co-immunoprecipitation, RNAi knockdown, immunofluorescence, ciliogenesis assay |
Developmental cell |
High |
18694559
|
| 2009 |
CP110 acts as a distal end-capping protein of centrioles; depletion of CP110 leads to elongated microtubule structures extending from centrioles. CP110 and CPAP play antagonistic roles in controlling the length of newly formed centrioles, with CPAP promoting tubulin addition and CP110 restraining it. |
siRNA knockdown, overexpression, immunofluorescence, electron microscopy of centriole ultrastructure |
Current biology : CB |
High |
19481458
|
| 2010 |
Cyclin F (Fbxo1) is the substrate recognition subunit of SCF(Cyclin F) ubiquitin ligase complex that physically associates with CP110 at centrioles during G2 and ubiquitylates CP110, leading to its proteasomal degradation. SCF(Cyclin F)-mediated CP110 degradation is required for mitotic fidelity; stable CP110 causes multipolar spindles and chromosome instability. |
Unbiased protein interaction screen, co-immunoprecipitation, ubiquitination assay, siRNA knockdown, expression of stable CP110 mutant, cell biology phenotype readouts |
Nature |
High |
20596027
|
| 2011 |
Kif24, a kinesin-13 subfamily motor protein, specifically interacts with CP110 and Cep97 at mother centrioles; loss of Kif24 causes disappearance of CP110 from mother centrioles in cycling cells and leads to aberrant cilia assembly. Kif24 depolymerizes microtubules in vitro and specifically remodels centriolar microtubules in cells. |
Co-immunoprecipitation, RNAi knockdown, in vitro microtubule depolymerization assay, immunofluorescence, ciliogenesis assay |
Cell |
High |
21620453
|
| 2012 |
In Drosophila, CP110 depletion results in centriole length diminution (in contrast to mammalian cells where depletion causes elongation); co-depletion of CP110 and Klp10A (kinesin-13) gives longer centrioles than usual, indicating functional interaction between these two proteins in controlling centriole length. |
RNAi in Drosophila cultured cells and testes, electron microscopy of centriole ultrastructure, co-depletion epistasis |
Current biology : CB |
Medium |
22365849
|
| 2013 |
USP33, a deubiquitinating enzyme, interacts with CP110 and localizes to centrioles primarily in S and G2/M phases; USP33 potently and specifically deubiquitinates CP110 (but not other cyclin-F substrates), counteracting SCF(cyclin F)-mediated ubiquitination. USP33 overactivity promotes supernumerary centrioles; USP33 ablation destabilizes CP110 and inhibits centrosome amplification. |
Co-immunoprecipitation, in vitro deubiquitination assay, RNAi knockdown, overexpression, centrosome number assay |
Nature |
High |
23486064
|
| 2013 |
In Drosophila, CP110 subtly influences centriole length by counteracting centriole-elongating activity of duplication proteins; CP110 ensures centriolar microtubules do not extend beyond the distal centriole end; CP110 suppresses centriole overduplication induced by overexpression of duplication proteins. |
CP110 null mutant fly analysis, overexpression, immunofluorescence and electron microscopy of centrioles |
The Journal of cell biology |
Medium |
24297749
|
| 2014 |
Talpid3/KIAA0586 is a component of a CP110-containing protein complex; Talpid3 assembles a ring-like structure at the extreme distal end of centrioles. Depletion of Talpid3 causes abnormal centriolar satellite distribution and cilia assembly defects reminiscent of Cep290 loss, including mislocalization of Rab8a; activated Rab8a suppresses cilia defects caused by Talpid3 depletion. |
Co-immunoprecipitation, RNAi, immunofluorescence, activated GTPase rescue epistasis |
The Journal of cell biology |
Medium |
24421332
|
| 2014 |
miR-34/449 miRNAs promote motile ciliogenesis in part by post-transcriptionally repressing Cp110; cp110 knockdown in miR-34/449-deficient multiciliated cells (mouse and Xenopus) restored ciliogenesis by rescuing basal body maturation and docking. |
miRNA knockout mice, Xenopus model, morpholino knockdown, cp110 knockdown rescue epistasis, basal body imaging |
Nature |
High |
24899310
|
| 2015 |
Centrin2 controls CP110 removal from the mother centriole during ciliogenesis; CETN2-deficient human RPE cells fail to remove CP110 and show abnormal distal appendage protein localization; knockdown of CP110 in CETN2-deficient cells rescues ciliation, placing centrin2 upstream of CP110 removal. |
CETN2 gene disruption (reverse genetics), immunofluorescence, CP110 knockdown epistasis, ciliogenesis assay |
The Journal of cell biology |
High |
25753040
|
| 2015 |
CP110 is a critical mediator of CDK2 inhibition-driven anaphase catastrophe; siRNA-mediated repression of CP110 induced anaphase catastrophe in lung cancer cells, while CP110 overexpression antagonized CDK2 inhibitor-mediated anaphase catastrophe. Site-directed mutagenesis identified CDK phosphorylation sites Ser170 and Thr194 as critical for conferring anaphase catastrophe by altering centrosome clustering in mitosis. |
siRNA knockdown, overexpression, site-directed mutagenesis, live-cell imaging, CDK2 inhibitor treatment |
Cancer research |
Medium |
25808870 26304236
|
| 2016 |
CP110 is required for anchoring basal bodies to the membrane during cilia formation in vivo; Cp110-/- mice die shortly after birth with organogenesis defects including impaired Shh signaling and reduced primary cilia. CP110 loss results in abnormal distribution of subdistal appendage (SDA) core components and recycling endosomes, implicating CP110 in SDA assembly and ciliary vesicle docking. |
Cp110 knockout mouse, immunofluorescence, ciliogenesis assay in multiple tissues, Shh signaling readout |
Development (Cambridge, England) |
High |
26965371
|
| 2016 |
In Xenopus multiciliated cells, Cp110 at optimal levels promotes ciliogenesis by localizing to cilia-forming basal bodies and rootlets and being required for ciliary adhesion complexes that facilitate actin interactions; coiled-coil domains mediate preferential binding to centrioles over rootlets. Cp110 levels are precisely controlled by both ciliary transcription factors and miRNAs. |
Xenopus in vivo experiments, domain mutagenesis, immunofluorescence, knockdown/overexpression of transcription factors and miRNAs |
eLife |
Medium |
27623009
|
| 2017 |
PLK4 specifically phosphorylates CP110 at Ser98; phospho-resistant CP110 (S98A) inhibits centriole assembly while phospho-mimetic CP110 (S98D) induces centriole assembly even under PLK4-limiting conditions. Phosphorylated CP110 augments centrosomal SAS6 levels, suggesting involvement in cartwheel stabilization. |
In vitro kinase assay, site-directed mutagenesis, overexpression of phospho-mutants, immunofluorescence, SAS6 quantification |
Cell cycle (Georgetown, Tex.) |
Medium |
28562169
|
| 2018 |
MPP9 is recruited by KIF24 to the distal end of the mother centriole where it forms a ring-like structure that recruits the CP110-CEP97 complex by directly binding CEP97. Phosphorylation of MPP9 by TTBK2 at the onset of ciliogenesis targets MPP9 for ubiquitin-proteasome degradation, which facilitates removal of CP110 and CEP97 from the mother centriole. |
Co-immunoprecipitation, immunofluorescence, siRNA knockdown, super-resolution microscopy, TTBK2 kinase assay |
Nature communications |
High |
30375385
|
| 2021 |
NudCL2 acts as a selective autophagy receptor at mother centrioles, containing an LIR motif that mediates association of CP110 with the autophagosome marker LC3. Knockout of NudCL2 induces defective CP110 removal from mother centrioles and ciliogenesis defects rescued by wild-type NudCL2 but not its LIR mutant; CP110 knockdown attenuates ciliogenesis defects in NudCL2-deficient cells. |
NudCL2 knockout, LIR motif mutagenesis, Co-IP, autophagy/LC3 interaction assay, RNAi epistasis, zebrafish morphant analysis |
Cell research |
High |
34480124
|
| 2021 |
CEP78 promotes ciliogenesis by negatively regulating CP110 levels via the EDD1-DYRK2-DDB1VPRBP E3 ubiquitin ligase complex involved in CP110 ubiquitination and degradation. CEP78 interacts with CEP350, which promotes centrosomal recruitment and stability of CEP78, leading to EDD1 centrosomal recruitment. Cells lacking CEP78 display significantly increased CP110 levels, and depletion of CP110 in CEP78-deficient cells restores ciliation frequency. |
Disease-based interactome screen, Co-IP, CEP78 knockout, RNAi epistasis, ciliogenesis assay |
eLife |
High |
34259627
|
| 2021 |
LUBAC (linear ubiquitin chain assembly complex) specifically generates linear ubiquitin chains on CP110, which is required for CP110 removal from the mother centriole during ciliogenesis. PRPF8, located at the distal end of the mother centriole, acts as a receptor for linear ubiquitin chains on CP110 to facilitate its removal at the initial stage of ciliogenesis. |
Co-immunoprecipitation, in vitro ubiquitination assay, siRNA knockdown, immunofluorescence, ciliogenesis assay |
The Journal of cell biology |
High |
34813648
|
| 2022 |
Aggresome assembly at the centrosome depends on CP110, CEP97, and CEP290; the seeding of a phosphorylated HSP27 ring around centrioles (the initial aggresome structure) requires these proteins. In senescent cells, reduced CP110 levels impair aggresome formation. |
siRNA knockdown, quantitative immunofluorescence, aggresome formation assay, mutant huntingtin aggregation assay |
Nature cell biology |
Medium |
35411088
|
| 2022 |
CP110 and Cep97 form a complex at the distal end of centrioles in Drosophila embryos whose levels rise and fall entrained by the Cdk-Cyclin oscillator; altering CP110 and Cep97 levels changes Plk4 oscillation and cartwheel growth at the proximal end, revealing unexpected crosstalk between distal-end factors and proximal centriole assembly. |
Live imaging in Drosophila embryos, quantitative fluorescence analysis, overexpression and knockdown of CP110/Cep97 |
Journal of cell science |
Medium |
35707992
|
| 2022 |
ENKD1 competes with CEP97 for binding to CP110; depletion of ENKD1 enhances the CP110-CEP97 interaction and retains CP110 at the mother centriole. Co-knockdown of ENKD1 and CP110 reverses ciliogenesis defects caused by ENKD1 depletion. Enkd1 knockout mice have ciliogenesis defects in multiple organs. |
Co-immunoprecipitation, competitive binding assay, siRNA knockdown, knockout mice, super-resolution microscopy, ciliogenesis assay |
EMBO reports |
High |
35301795
|
| 2023 |
EHD1 regulates CP110 ubiquitination during ciliogenesis; EHD1 controls centriolar satellite movement to the mother centriole, delivering the E3 ubiquitin ligase HERC2, which interacts with and ubiquitinates CP110. MIB1 also ubiquitinates CP110. HERC2 is required for ciliogenesis and localizes to centriolar satellites. |
Co-immunoprecipitation, ubiquitination assay, siRNA knockdown, immunofluorescence, centriolar satellite trafficking assay |
EMBO reports |
High |
37074924
|
| 2023 |
CCP5 and CCP6 interact with CP110 (CCP5 through its N-terminus) and retain CP110 at the mother centriole to suppress cilia formation in cycling cells. Loss of CCP5 or CCP6 causes disappearance of CP110 from the mother centriole and increased ciliation; these effects are independent of their enzymatic deglutamylation activity. |
CoIP-MS, co-immunoprecipitation, RNAi knockdown, immunofluorescence, ciliogenesis assay, domain mapping |
BMC biology |
Medium |
37226238
|
| 2023 |
ODF2 controls CP110 levels at centrioles; ODF2 knockdown leads to decreased CP110 levels, while ODF2 most likely acts as a scaffold for NEURL4 or HYLS1 binding to mediate CP110 degradation via the ubiquitin-dependent proteasome pathway. |
siRNA knockdown, rapamycin-mediated dimerization recruitment assay, immunofluorescence, ciliogenesis assay |
Cells |
Low |
37681926
|
| 2025 |
p97/VCP (valosin-containing protein), an AAA-ATPase, is responsible for removal of CP110 from the mother centriole; p97 knockdown or inhibition impairs ciliogenesis in a mechanism dependent on CP110. p97 is proposed to unfold and extract polyubiquitinated CP110 from the mother centriole. |
siRNA knockdown, p97 chemical inhibition, ciliogenesis assay, immunofluorescence, epistasis with CP110 |
Molecular biology of the cell |
Medium |
39785673
|
| 2025 |
EHD1 promotes CP110/CEP97 removal from the mother centriole cap through its membrane tubulation function during formation of tubular ciliogenesis intermediates; EHD1 and RAB8 orchestrate formation of tubular C-shaped and toroidal membrane intermediates upstream of the ciliary vesicle. |
3D electron microscopy (isotropic ultrastructure imaging), EHD1 depletion/inhibition, immunofluorescence, quantitative analysis of membrane structures |
bioRxivpreprint |
Medium |
|