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"High-throughput screening of some nine and a half thousand bioactive compounds with the USP1/UAF1 complex using a Ub-Rho substrate by the Zhuang group identified several low μM hits [68]."

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"The USP1 and UAF1 complex has also been reported to participate in homologous recombination [XREF_BIBR, XREF_BIBR, XREF_BIBR]."

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"The compound was found to be ‘largely reversible’, not binding at the active site and not acting via USP1/UAF1 complex disruption."

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"We carried out SEC-MALLS of the purified USP1 and UAF1 complex and the molecular weight obtained was approximately 220 KDa which closely corresponds to the 1:2 complex (235 KDa) rather than a 1:1 complex (160 KDa)."

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"Furthermore, by genetically tagging UAF1 with a fluorescent protein, such as RFP, and using automated microscopy procedures, the assay can be readily adapted to a high throughput format in order to allow a high content screening for molecules that interfere with USP1 and UAF1 complex formation."

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"This observation suggests that the USP1 and UAF1 complex may be an important novel target in cancer treatment and thus, interfering with the formation and/or inducing the dissociation of the complex may represent a potential therapeutic strategy."

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"However, not much has been studied regarding the roles of the USP1 and UAF1 complex in centrosome regulation."

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"Importantly, the striking change in the nucleocytoplasmic localization of Xpress-UAF1 that results from GFP-USP1 co-expression provides the basis for a relocation assay to test USP1 and UAF1 complex formation in cells."

sparser
"Binding of USP1 by WDR48 was sensitive to deletions within the WD-repeats, indicating the importance of a presumed propeller domain ( Cohn et al., 2007 )."

sparser
"The end of the FA pathway is marked with the deubiquitination of FANCD2/ FANCI heterodimer by the USP1-UAF1 complex xref ."

sparser
"Indeed, the structure of USP1UAF1 complex bound to ID2 Ub ‐DNA revealed ID2 movements towards the open‐state conformation, affecting not only FANCI helices in the region where ubiquitin‐conjugation occurs, but also, and most profoundly, the FANCD2 N‐terminus involved in interaction with FANCI's ubiquitin (Rennie et al ,  xref )."

sparser
"The subsequent unhooking of ICL would be followed by TLS and deubiquitination of the ID complex with USP1-UAF1 complex."

sparser
"This model, however, seems difficult to reconcile with the finding that the carboxy-terminal region of USP1 is unable to interact with UAF1 [ xref ], and would require further experimental validation."

sparser
"To some extent, this may be due to USP1UAF1 preferentially targeting FANCD2, via USP1's N‐terminal extension (Arkinson et al ,  xref ) and through UAF1‐FANCI interactions acting as a USP1‐FANCD2 enzyme‐substrate recruitment module (Rennie et al ,  xref )."

sparser
"Indeed, we have shown here that disruption of this extended FANCD2‐ubiquitin interface (via FANCD2 mutagenesis), results in I Ub D2‐DNA being more sensitive to USP1UAF1 action (Fig  xref )."

sparser
"Recent studies corroborate that Ser313 is not critical for USP1:WDR48 binding and function ( Olazabal-Herrero et al., 2015 )."

sparser
"Further structural work will be required to elucidate how USP1UAF1 targets the ubiquitin on FANCI and how dsDNA may interfere with such targeting."

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"We demonstrated that the binding of UAF1 to USP1 does not affect the binding of ubiquitin to USP1."

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"Several studies showed that UAF1 forms a complex with USP1 and stimulates the deubiquitination activity of USP1 both in vitro and in vivo."

sparser
"However, while the mechanism of FANCD2 ubiquitination and deubiquitination has been sufficiently elucidated (Chaugule et al ,  xref ; Rennie et al ,  xref ; Wang et al ,  xref ), we are still lacking information on how UBE2T and FA‐core engage the mono‐ubiquitinated ID2 complex for FANCI ubiquitination, and how the ubiquitin from FANCI is removed by USP1UAF1."

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"Recent work showed that phosphorylation of USP1 by kinase CDK1 promotes the complex formation between USP1 and UAF1."

sparser
"Moreover, mutagenesis of equivalent residues in the WDR48:USP1 system abrogates both USP1 binding to WDR48 and deubiquitination of a USP1 substrate in cells, suggesting that this interface, distinct f[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

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"The formation of USP1 and UAF1 complex greatly stimulates the DUB activity of USP1."

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"The exact mode of interaction between UAF1 and USP1 remains to be determined."

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"The USP1 and UAF1 complex promotes double-strand break repair through homologous recombination."

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"Moreover, WDR20 is specific for USP12 and it did not stimulate the DUB activity of the USP1 and UAF1 complex."

sparser
"However, how the fully engaged di-mono-ubiquitinated complex (Ub-FANCD2-Ub-FANCI), which is no longer a substrate for USP1-UAF1 xref , is removed, remains elusive."

sparser
"Recent structural studies of the factors involved in FA pathways, such as the FA core complex, the FANCI:FANCD2 heterodimer and the USP1-UAF1 DUB complex, are prime examples ( xref )."

sparser
"Interestingly, although the modifications in each pathway are assembled by distinct enzymes, they are removed by the same deubiquitinase (DUB), the USP1USP1-associated factor 1 (UAF1) complex ( xref , xref , xref )."

sparser
"Lastly, FANCD2 is deubiquitinated by USP1UAF1 complex to terminate the FA pathway ( xref , xref )."

sparser
"ML323 is a highly potent inhibitor of the USP1-UAF1 deubiquitinase complex."

sparser
"The cleavage rates show that the activity of a USP1 ΔNΔ1Δ2 –UAF1 complex is almost identical to that of USP1 FL –UAF1."

sparser
"Protein interaction mapping analysis identified some key residues within RAD51AP1 required for interacting with the USP1-UAF1 complex."

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"Altogether, these results support an idea that USP1 and UAF1 may have a function independent of FANCD2, and at a level downstream of RAD51 foci formation, in addition to deubiquitinating FANCD2.The USP1 and UAF1 complex interacts with RAD51AP1In order to search for the potential role of USP1 or UAF1 in HR repair, we undertook a proteomic approach to identify potential UAF1 or USP1 interacting proteins that regulate HR repair."

sparser
"In osteosarcoma and non-small cell lung cancer cells, ML323 inhibits the deubiquitination activity of the USP1-UAF1 complex in the DNA translesion synthesis and Fanconi anemia pathways in response to DNA damage."

sparser
"UAF1-USP1 has been reported to sustain pSTAT1 signaling by deubiquitinating and preventing proteasomal degradation of TBK1 [ xref ]."

sparser
"Likewise, mutation of USP1 E444K, analogous to the impactful USP46 E186K mutation, abrogated WDR48 binding and stimulation of USP1 deubiquitinase activity."

sparser
"The tri-phosphorylation of FANCI also inhibits the deubiquitinase activity of the USP1-UAF1 complex over the ID2 complex until ICL repair and replication are completed [ xref ]."
| PMC

sparser
"To search for the domains of NLRP3 responsible for the interaction with USP1 and UAF1, a series of Myc-tagged NLRP3 truncated mutants were constructed (Fig.  xref )."

sparser
"However, the PYD domain mutant (PYD) lost the ability to bind USP1 and UAF1 (Fig.  xref )."

sparser
"USP1UAF1 directly deubiquitinates monomeric FANCD2-Ub and FANCI-Ub."

sparser
"The same effect of I44A and F4A on FANCD2 deubiquitination was observed when using four times lower concentrations of USP1UAF1 in single time‐point DUB assays (Fig  xref A)."

sparser
"The deubiquitination is performed by the USP1-UAF1 complex resulting in the release of the ID2 complex from the chromatin to complete the ICL repair cycle [ xref ]."
| PMC

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"In this study, we identified that the USP1 and WDR48 complex could effectively enhance TGF-beta-mediated EMT and migration of TNBC cells."

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"Moreover, the USP1 and WDR48 complex was found to downregulate the polyubiquitination of TAK1 and mediate its in vitro stability."

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"Therefore, our findings have shed a light on the novel role of the USP1 and WDR48 complex in promoting TGF-beta-induced EMT and migration in TNBC via in vitro stabilization of TAK1."

sparser
"The USP1UAF1 complex can deubiquitinate several monoubiquitinated substrates, including human FANCD2-K561Ub ( hs FANCD2-Ub) ( xref ), human FANCI-K523Ub ( hs FANCI-Ub) ( xref ), and human PCNA-K164Ub (PCNA-Ub) ( xref )."

sparser
"Once the DSB is repaired, FANCD2-Ub has to be extracted from the lesion; for this to occur, it has to be deubiquitinated by the USP1-UAF1 deubiquitinase complex and p97."
| PMC

sparser
"De-ubiquitination of the monoubiquitinated ID2 complex by USP1-UAF1 stimulates its unloading from chromatin and knockdown of USP1 impairs subsequent DNA repair steps xref ."

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"Focusing on autophagy that makes a critical effect on tumor development, researchers have successively identified and screened various specific small-molecule inhibitors of autophagy-related DUBs, including USP1-UAF1 complex inhibitors GW7647 and Pimozide, which can successfully reverse NSCLC cell resistance to chemotherapeutic drugs (Chen et al., 2011)."

sparser
"These results, collectively with our observation that SIVA1 depletion did not affect the chromatin association of USP1 and UAF1 ( xref ), exclude the possibility that SIVA1 may act as an antagonist of PCNA deubiquitination by the USP1UAF1 complex."

reach
"However, UAF1 forms a complex with USP1 to activate its deubiquitinating activity."

sparser
"Furthermore, the mechanism for the removal of ubiquitin from FANCD2 was also clarified by cryo-EM and crystallography experiments with USP1-UAF1 ( xref )."

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"ML323 is known to disrupt the interaction between USP1 and UAF1 [31], resulting in decreased interaction between USP1 and C/EBPβ."

sparser
"The I36A mutation resulted in partial loss, whereas the F4A and L73A mutations on ubiquitin resulted in complete loss of USP1UAF1 activity (Fig  xref D)."

sparser
"UAF1 also binds and activates two other DUBs, USP12 and USP46 ( xref ), and studies that reveal how UAF1 binds and activates USP12 and USP46 suggest that UAF1 will bind to USP1 in an analogous manner ( xref , xref )."

sparser
"Here, we report the reconstitution of substrate deubiquitination by USP1UAF1 that allows for a modular approach to understanding the molecular requirements for deubiquitination of physiological substrates."

sparser
"We find that UAF1 can bind the minimal USP1 catalytic domain (USP1 ΔNΔ1Δ2 ) by size exclusion chromatography (SEC) ( xref ) and stimulate DUB activity on a fluorescent ubiquitinated-dipeptide substrate, Ub-KG TAMRA ( xref )."

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"USP1 binds to USP1-associated factor 1 (UAF1), which stimulates the deubiquitinase activity of USP1 [45]."

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"ML323 exhibits reversible nanomolar inhibitory activity against the USP1/UAF1 complex with excellent selectivity [25, 27]."

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"The monoubiquitylation licensing of the ID complex is antagonized by the DUB complex USP1 and UAF1; however, it is currently unknown how PTMs regulate USP1 and UAF1 after DNA damage."

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"Pimozide and GW7647 were the first reported inhibitors of the USP1/UAF1 complex; these compounds are also known to regulate the activity of unrelated targets, whereas ML323 is known to specifically inhibit the activity of the USP1/UAF1 complex [46]."

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"When the repair process is completed, the FANCD2 and FANCI complex is deubiquitylated and dissociated from the repaired ICL site by the USP1 and UAF1 complex and is then released from the DNA [XREF_BIBR]."

sparser
"With the substrates purified ( xref ), we assessed whether hs FANCD2-Ub, hs FANCI-Ub, or hs PCNA-Ub is directly deubiquitinated by USP1UAF1 in vitro."

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"The UAF1 and USP1 complex deubiquitinates FANCD2 during execution of the Fanconi anemia DNA damage response pathway."

sparser
"A recent report suggests that xl FANCI is required for human USP1UAF1 to remove ubiquitin from xl FANCD2-Ub ( xref )."

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"USP1 stably associates with UAF1 and WDR48, and its DUB activity and intracellular stability are dependent on the latter."

sparser
"Taken together, these data establish that recombinant USP1UAF1 is able to directly deubiquitinate diverse monoubiquitinated substrates in vitro without the need for auxiliary factors."

sparser
"Crystals of the apo- and ubiquitin-bound form of USP1-UAF1, in conjunction with cryo-EM reconstructions of the enzyme-substrate complex, revealed important details of the specificity and regulation of this reaction."

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"However, like UAF1, neither USP1 nor the UAF1 and USP1 complex exerts any influence on the D-loop reaction (XREF_SUPPLEMENTARY; data not shown)."

sparser
"The UAF1-USP1 deubiquitinase complex selectively eliminates K48-linked ubiquitin chains of NLRP3 to stabilize its expression via interaction with the LRR and NACHT domains, which in turn promotes NLRP3 inflammasome activation [ xref ]."

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"One substrate of the USP1 and UAF1 complex is the monoubiquitinated FANCI and FANCD2 heterodimer, which is involved in the repair of DNA interstrand crosslinks via the Fanconi anemia pathway."

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"XREF_BIBR, XREF_BIBR Human ATAD5 regulates PCNA deubiquitination by recruiting the USP1 and UAF1 complex to ubiquitinated PCNA."

sparser
"To test this, we purified xl FANCD2-Ub to homogeneity ( xref ) and assayed with human USP1UAF1."

sparser
"Our study also provides an additional basis for development of therapeutic inhibitors of USP1-UAF1 which may sensitize cancer 2644 S. CUKRAS ET AL. cells that depend on HR repair."

sparser
"Structural basis of FANCD2 deubiquitination by USP1-UAF1."

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"Human Elg1 and ATAD5 promotes PCNA deubiquitination by recruiting the USP1 and UAF1 complex to ubiquitinated PCNA."

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"ML323 and other commercially available inhibitors of the USP1/UAF-1 complex act by targeting the DUB complex as opposed to the USP1 active site [52]."

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"RAD51AP1’s SIM domain interacts with the SUMO-like domains (SLD) of the UAF1-USP1 complex."

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"The end of the FA pathway is marked with the deubiquitination of FANCD2/ FANCI heterodimer by the USP1-UAF1 complex ."

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"The subsequent unhooking of ICL would be followed by TLS and deubiquitination of the ID complex with USP1-UAF1 complex."

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"The only currently accepted negative regulator of TLS is the deubiquitinase complex USP1 and UAF1."

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"Interestingly, depletion of FANCI did not affect the interaction between PHLPP1 and the USP1 and UAF1 heterodimer indicating that FANCI specifically affects the Akt-PHLPP1 interaction."

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"ML323, an inhibitor of the USP1/UAF1 complex, was developed by Zhuang Lab [80, 81] and stood to be the only USP family DUB inhibitor that met the chemical probe criteria [82] by 2016."

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"However, the exact structural details on how ML323 binds to the USP1/UAF1 complex remained elusive, which prevented further structure-based optimization of the inhibitor."

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"In any case, HU counteracts USP1 and p21 with alternative strategies : USP1 interaction with its activating partner UAF1 is eliminated by HU and the interaction of p21 and PCNA is specifically prevented after HU."

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"The USP1/UAF1 complex maintains FANCD2 levels during normal cellular growth."

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"USP1 interacts with the activating protein partner UAF1 (USP1 associated factor 1) and de-ubiquitylate Ubi-PCNA in the absence of DNA damage."

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"27 In addition to BRCA1, DUBs such as USP38 and the USP1UAF1 complex are involved in HR and NHEJ by deubiquitinating other important proteins in DNA repair pathways."

sparser
"Finally, we found that in the full-length USP1UAF1 context, a single point mutation in its N-terminus (R22E) is sufficient for specifically disrupting hs FANCD2-Ub deubiquitination but not hs FANCI-Ub or hs PCNA-Ub ( xref )."

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"Using high throughput screening, we identified a small molecule inhibitor of the USP1 and UAF1 complex."

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"In addition, monoubiquitinated FANCD2/FANCI proteins are deubiquitinated by the USP1/UAF1 complex after DNA removal, completing the ICL repair procedure [42–45]."

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"The USP1 and UAF1 complex is responsible for regulating DNA break repair pathways such as trans-lesion synthesis pathway, Fanconi anemia pathway and homologous recombination."

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"We therefore utilized a purified USP1 and UAF1 complex and performed a high throughput screen to identify a USP1 inhibitors (XREF_FIG)."

sparser
"Several studies showed that UAF1 forms a complex with USP1 and stimulates the deubiquitination activity of USP1 both in vitro and in vivo ( xref , xref - xref )."

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"A small molecule, C527 (heterocyclic tricyclic 1,4-dihydro-1,4-dioxo-1H-naphthalene) (XREF_FIG) was identified which inhibited the USP1 and UAF1 complex."

reach
"We next confirmed the ability of C527 to inhibit the USP1 and UAF1 complex in a dose dependent and time dependent manner, using Ub-AMC as a substrate (XREF_FIG)."

sparser
"We assayed interaction with hs FANCD2 and found that a USP1 Δ1Δ2 –UAF1 complex can capture the non-ubiquitinated substrate ( xref )."

reach
"Removal of the two ubiquitins, through isopeptide cleavage by the USP1UAF1 complex, is also required for ICL repair and maintenance of genomic stability (Oestergaard et al, 2007; Kim et al, 2009; Murai et al, 2011)."

sparser
"USP1 associates with its activating factor UAF1, and the USP1-UAF1 complex removes monoubiquitin from FANCD2 to complete the repair ( xref ) ( xref )."

reach
"Indeed, the structure of USP1UAF1 complex bound to ID2 ‐DNA revealed ID2 movements towards the open‐state conformation, affecting not only FANCI helices in the region where ubiquitin‐conjugation occurs, but also, and most profoundly, the FANCD2 N‐terminus involved in interaction with FANCI's ubiquitin (Rennie et al, 2021)."

reach
"In human cells, ATAD5, the ortholog of yeast Elg1 appears to have a somehow different role from its yeast counterpart as it interacts, at stalled replication forks, with the USP1 and UAF1 complex and facilitates USP1 mediated PCNA de-ubiquitylation."

reach
"USP1 by itself exhibits low DUB activity; however, this activity is significantly enhanced when bound as a USP1 and UAF1 complex."

sparser
"The association of USP1 and UAF1 in cells is likely regulated in response to DNA damage."

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"Since USP1 affects centrosome duplication and ATAD5 interacts with the USP1 and UAF1 complex [XREF_BIBR, XREF_BIBR], we hypothesized that ATAD5 plays a role in centrosome duplication."

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"However, the IC 50 of C527 for these DUB enzymes was higher in comparison to USP1 and UAF1 complex (XREF_TABLE)."

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"Since ATAD5 interacts with the USP1 and UAF1 complex and USP1has been reported to regulate centrosome duplication by increasing ID1 levels [XREF_BIBR, XREF_BIBR], we speculated that ATAD5 modulates ID1 to regulate centrosome duplication."

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"Taken together, C527 is a pan deubiquitinating enzyme inhibitor in vitro, with a high nanomolar IC 50 for the USP1 and UAF1 complex."

reach
"Taken together, these data demonstrate that SJB2-043 is an inhibitor of the native USP1 and UAF1 complex."

reach
"Given the structural similarity between these USP14 inhibitors and the USP1 inhibitor ML323 (12), it is tempting to speculate that this equally drug-like lead may bind into a similar region of the USP1/UAF1 complex.In June 2015, Genentech and Almac Discovery announced a collaboration on an undisclosed USP target, whereby Almac Discovery's small-molecule leads would provide the starting point for a two-year joint research programme."

sparser
"Elegant work by Rennie et al. (2021) revealed that, when USP1-UAF1 binds, the I-D2-Ub complex is remodeled to a more open conformation, allowing USP1 to extract Ub from the complex and enclose it with[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

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"For instance, the USP1 and UAF1 complex deubiquitinates the cellular monoubiquitinated substrates Ub-FANCD2 and Ub-PCNA."

sparser
"We demonstrated that the binding of UAF1 to USP1 does not affect the binding of ubiquitin to USP1."

sparser
"USP1 interacts with WDR48 in MHCC97H and SK‐Hep‐1 cells."

sparser
"A recent report has showed that the di-monoubiquitinated frog ID2 complex (both FANCD2 and FANCI monoubiquitinated), generated in the presence of plasmid DNA, remains resistant to deubiquitination by a GST-tagged USP1UAF1 complex and only the removal of DNA permits efficient DUB activity ( xref )."

sparser
"We subsequently treated half of the substrate sample with benzonase nuclease to eliminate the DNA before subjecting both the nuclease-treated and untreated substrate samples to USP1UAF1 activity."

sparser
"The exact mode of interaction between UAF1 and USP1 remains to be determined."

sparser
"In addition, we confirmed the interaction between USP1 and WDR48 in HCC cell lines using co‐immunoprecipitation, and the results showed that USP1 interacted with WDR48 in MHCC97H and SK‐Hep‐1 cells (Figure  xref )."

sparser
"The recruitment of USP1-UAF1 is thought to be via a SUMO-like domain on UAF1 that recognises the SUMO-interacting motif (SIM) on ELG1, a PCNA-binding RFC-like adaptor for Ub removal from PCNA [ xref , xref ]."

sparser
"USP1-UAF1 also contributes to genome stability as a DUB for FA repair of ICLs caused by MMC or cisplatin [ xref , xref , xref ]."

sparser
"The USP1-UAF1 deubiquitinase complex facilitates TLR3/4-, RIG-I-, and cGAS-induced beta interferon (IFN-β) production to enhance antiviral responses during Sendai virus and vesicular stomatitis virus infection ( xref )."

reach
"Mutational analysis suggests the same is true of WDR48 interaction and activation of USP1 (140)."

sparser
"When the repair process is completed, the FANCD2 and FANCI complex is deubiquitylated and dissociated from the repaired ICL site by the USP1-UAF1 complex and is then released from the DNA [ xref ]."

sparser
"Interestingly, and despite the presence of FANCI, which may also recruit the DUB complex ( xref ), USP1 ΔN –UAF1 and USP1 R22E –UAF1 are not able to fully deubiquitinate hs FANCD2-Ub, suggesting that any indirect recruitment of USP1UAF1 by FANCI does not completely compensate for the loss of the USP1 N-terminus."

sparser
"Moreover, we confirmed the interaction of USP1WDR48 in HCC cells using co‐immunoprecipitation."

sparser
"As RAD51AP1 amplification is associated with multiple types of cancers,18,43-45 therapeutic targeting of the USP1-UAF1 complex may be beneficial in suppressing these tumors."

sparser
"As reported, USP1 alone had low deubiquitinase activity, and its activity was significantly promoted when forming a complex with its binding partner WDR48 . xref USP1WDR48 mainly works by maintaining the activity of their substrates."

sparser
"Removal of the ubiquitin signal from FANCD2 is conducted by the USP1UAF1 DUB, and disruption of USP1 catalytic activity results in an accumulation of FANCD2-Ub and FA-like phenotypes ( xref , xref , xref , xref )."

sparser
"Yu et al reported that the USP1WDR48 complex stabilized TANK‐binding kinase 1 by moving its K48‐linked polyubiquitination, and this process could be attenuated using the inhibitor ML‐323. xref Intriguingly, we found that WDR48 was also overexpressed in HCC tissues compared with normal tissues and was associated with poor prognosis in patients."

sparser
"Structural data of USP1-UAF1, with Ub and in an enzyme-substrate complex with ubiquitylated FANCI-D2, reveals critical mechanistic and regulatory details for Ub removal from FANCD2 [ xref ]."

sparser
"By reconstituting FANCD2-Ub, FANCI-Ub, and PCNA-Ub deubiquitination by USP1UAF1, we identified the minimal components required for deubiquitination and discovered a unique sequence within the N-terminus of USP1 critical for removing ubiquitin from FANCD2 ( xref )."

sparser
"Hence, we hypothesized that the USP1WDR48 complex played critical roles in HCC by stabilizing the activity of their substrates."

sparser
"This finding suggested that the USP1WDR48 complex played a tumour‐promoting role in HCC by stabilizing and deubiquitinating these hub genes."

sparser
"In the past years, several agents aimed at USP1 have been reported, such as pimozide and ML‐323. xref , xref These agents inhibit the activity of USP1WDR48 complex in a non‐competitive manner. xref In the present study, we demonstrated that siRNA‐USP1 transfection or ML‐323 treatment decreased the proliferation of HCC cells."

reach
"The inhibition of the deubiquitinase complex USP1-UAF1 or the expression of constitutive fusion of ubiquitin to PCNA provides the missing clue required for DNA polymerase η recruitment and thereafter the introduction of A/T base pair (bp) mutations during the process of IgV gene diversification."

sparser
"Further studies may reveal additional layers of specificity for substrates such as PCNA-Ub, which could also be mediated by the N-terminus of USP1UAF1."

reach
"Recent studies have found that inhibiting the activity of the USP1/UAF1 complex can improve cisplatin sensitivity in NSCLC (85)."

reach
"The cata-lytic activity and stability of USP1 is promoted by itsstoichiometric binding partner UAF1 (USP1 Associated Factor 1; WDR48), a WD40 repeat containing protein.11 Both USP1and UAF1 are regulators of the HR repair, as knockouts ofUSP1 or UAF1 in DT40 cells show reduced HR repair efficiency.12 The USP1 and UAF1 complex also deubiquitinates FANCI, which interacts with FANCD2,13 and a replicative polymerase processive factor PCNA.14 Altogether, USP1 andUAF1 are important contributors to the genome integrity atleast in part by regulating the HR and TLS DNA repairpathways.CONTACT Younghoon Kee Ykee@usf.edu Department of Cell Biology, Microbiology, and Molecular Biology, College of Arts and Sciences, University of South Florida, Tampa, FL 33620, USA."

sparser
"Lastly, FANCD2-Ub is downregulated by the USP1-UAF1 deubiquitinating enzyme complex, and the remaining adduct is removed via nucleotide excision repair (NER) to complete the ICL repair ( xref ; xref ) ( xref )."

sparser
"USP1UAF1, therefore, works to recycle and maintain appropriate levels of both monoubiquitinated and non-ubiquitinated FANCD2."

sparser
"Inhibition of USP1UAF1 activity by the specific small molecule ML323 leads to an increased sensitivity to platinum-based compounds in resistant cells ( xref )."

sparser
"Ubiquitinated FANCI/FANCD2 complex is finally deubiquitinated by ubiquitin specific peptidase 1-USP1-associated factor 1 (USP1-UAF1)."

sparser
"Furthermore, the USP1UAF1 complex has been shown to play critical roles in homologous recombination ( xref )."

sparser
"However, USP1UAF1 targets multiple substrates, including the inhibitor of DNA-binding proteins and TBK1, meaning a catalytic activity inhibitor may affect multiple processes."

sparser
"The USP1-UAF1 complex interacts with RAD51AP1 In order to search for the potential role of USP1 or UAF1 in HR repair, we undertook a proteomic approach to identify potential UAF1 or USP1 interacting proteins that regulate HR repair."

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"It has been shown to inhibit the USP1/UAF1 complex and exhibit strong dose-dependent tumor response in xenograft-derived BRCA-mutated triple-negative breast cancer with synergy seen when given with a PARP inhibitor."

reach
"The scaffold protein UAF1 forms a complex with USP1, USP12, and USP46 that is essential for their activity."

reach
"Disruption of USP1/UAF1 complex promote level of FANCD2/FANCI ubiquitination and DNA repair defects, suggesting a failure in the completion of FA pathway (Nijman et al., 2005b, Smogorzewska et al., 2007)."

sparser
"RAD51AP1’s SIM domain interacts with the SUMO-like domains (SLD) of the UAF1-USP1 complex."

sparser
"Both UAF1 and USP1 bind DNA ( xref )."

sparser
"Yu et al. ( xref ) found that USP1-UAF1 deubiquitinase complex stabilizes TBK1 and enhances antiviral responses."

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"15, NO. 19, 2636-2646 http://dx.doi.org/10.1080/15384101.2016.1209613REPORTThe USP1 and UAF1 complex interacts with RAD51AP1 to promote homologous recombination repairScott Cukrasa, Euiho Leea, Emily Palumboa, Pamela Benavideza, George-Lucian Moldovanb, and Younghoon KeeaaDepartment of Cell Biology, Microbiology, and Molecular Biology, College of Arts and Sciences, University of South Florida, Tampa, FL, USA; bDepartment of Biochemistry and Molecular Biology, Pennsylvania State University College of Medicine, Hershey, PA, USAABSTRACTUSP1 deubiquitinating enzyme and its stoichiometric binding partner UAF1 play an essential role in promoting DNA homologous recombination (HR) repair in response to various types of DNA damaging agents."

sparser
"ML323 (a specific USP1-UAF1 inhibitor) and USP1/UAF1 knockdown attenuated IFN-β expression and enhanced viral replication both in vitro and in vivo ( xref )."

sparser
"40 However, we were unable to detect the ubiquitinated species of RAD51AP1, thus we are not able to conclude whether the USP1-UAF1 complex protects RAD51AP1 from proteasomal degradation by deubiquitination."

sparser
"For example, USP1 interaction with UAF1 is necessary for DUB activity in vitro and deubiquitination of cellular substrates PCNA and FANCD2 ( xref ; xref ; xref )."

sparser
"USP1 binds to DNA independently of UAF1 and is stimulated preferentially by DNA structures mimicking a replication fork."

sparser
"To test this possibility, we examined the ubiquitin protease activity of full length USP1 bound in a complex with UAF1, with or without DNA, using the ubiquitin-AMC release assay and the ubiquitin CHOP2-Reporter assay ( xref ) ( xref ; xref ; xref ; xref ; xref )."

sparser
"Removal of FANCD2 from stalled replication forks is regulated by the deubiquitinating enzyme USP1-UAF1 and is necessary following repair to allow for replication restart xref ."

reach
"As K48-linked ubiquitination is linked with protein degradation, the UAF1/USP1 complex stabilizes the degradation of NLRP3, which increases cellular NLRP3 levels and promotes NLRP3 inflammasome activation."
| PMC

reach
"The amount of FANCA in chromatin is affected in the former cell line but not in the latter.The de and ubiquitinase complex USP1 and UAF1 [86,87] counteracts the mono-ubiquitination of FANCD2-FANCI."

reach
"Since ML323 has an inhibitory effect on the UAF1/USP1 complex, its treatment keeps NLRP3 in check at the cellular level and prevents NLRP3 inflammasome activation."
| PMC

reach
"To validate a novel role of USP1 along this RSK1-FLT3 axis, we treated MV4-11 and MOLM-13 cell lines with USP1 inhibitor SJB3-019A which reduced expression of FLT3 and USP1-UAF1 complex and suppressed FLT3 and S6 phosphorylation (Fig. 4J)."

sparser
"USP1 (ubiquitin-specific peptidase 1), which promotes de-ubiquitination of mono-ubiquitinated PCNA [57] , forms a stable complex with UAF1 (USP1-associated factor 1), which stabilizes and activates U[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"Recent studies have progressively elucidated the immunoregulatory roles of the USP1UAF1 complex. [ xref ] For instance, one study indicates that USP1UAF1 modulates the differentiation of T helper cell 17 and Treg cells by stabilizing Tafazzin, [ xref ] while another demonstrates that USP1UAF1 stabilizes NLR family pyrin domain containing 3 (NLRP3), thereby activating the NLRP3‐mediated inflammatory phenotype. [ xref ] In the context of type I interferon pathway regulation during RNA virus infection, a report suggests that USP1UAF1 augments antiviral responses by stabilizing TBK1. [ xref ] However, the regulatory mechanism of USP1UAF1 within the STING‐TBK1‐IRF3 signaling pathway remains uncharted."

reach
"The USP1 and UAF1 complex keeps in check FANCD2 ubiquitination under normal conditions."

reach
"The tri phosphorylation of FANCI also inhibits the deubiquitinase activity of the USP1 and UAF1 complex over the ID2 complex until ICL repair and replication are completed [XREF_BIBR]."
| PMC

reach
"These results, collectively with our observation that SIVA1 depletion did not affect the chromatin association of USP1 and UAF1 (XREF_FIG), exclude the possibility that SIVA1 may act as an antagonist of PCNA deubiquitination by the USP1 and UAF1 complex."

reach
"The deubiquitination is performed by the USP1 and UAF1 complex resulting in the release of the ID2 complex from the chromatin to complete the ICL repair cycle [XREF_BIBR]."
| PMC

sparser
"How this ISG15- and USP10-mediated signalling functions in parallel to the more established USP1-UAF1 mediated regulation of PCNA mono-ubiquitylation will need further exploration."

reach
"In addition, the function of the USP1/UAF1 complex in inflammation was discovered through its interaction with NLRP3 [13]."

reach
"For example, WDR48 interacts with USP1, USP12 and USP46 to activate their DUB activity."

sparser
"ATR-mediated phosphorylation of the USP1-UAF1 complex and of its substrate, ubiquitylated FANCI-D2, has also been shown to regulate the formation of the enzyme-substrate complex for efficient Ub removal."

sparser
"FANCI recruits the deubiquitinating enzyme USP1:UAF1 ( xref ), which prevents trapping of monoubiquitinated FANCD2 at non-productive DNA damage sites, but only ID2 Ub and not I Ub D2 Ub is a substrate ( xref )."

sparser
"Related to USP1-UAF1, is how some DUBs function in multiple different DDR signalling pathways."

sparser
"Although we showed that FANCD2-Ub with FANCI and DNA does not obviously affect USP1UAF1 deubiquitination in vitro, FANCI has been shown to regulate FANCD2 deubiquitination in cells via phosphomimetic mutations ( xref ) and SUMO-like interacting motif mutations ( xref )."

sparser
"For example, we and others have shown that di-monoubiquitinated DNA-bound ID2 complexes remain resistant to deubiquitination ( xref ), suggesting that FANCD2-Ub and FANCI-Ub in the presence of DNA reciprocally regulate their deubiquitination by USP1UAF1."

sparser
"How di-monoubiquitination of ID2–DNA regulates the ability of USP1UAF1 to remove ubiquitin from FANCD2 will be an interesting mechanism to investigate."

reach
"On the right are the representative images.immunoprecipitation also reversely pulled down both UAF1 and USP1, suggesting that RAD51AP1 associate with the USP1 and UAF1 complex."

sparser
"For example, ML323 is a selective USP1-UAF1 inhibitor that has shown promise to inhibit FA and TLS signalling to maximise cancer cell cytotoxicity with few off-target effects [ xref ]."

sparser
"Human USP1 forms a stable complex with UAF1 ( xref )."

reach
"Deubiquitination of FANCD2 and FANCI proteins by the multisubunit protein complex USP1 and UAF1 is required for the completion of the FA pathway."

reach
"HSCARG enhances the interaction between PCNA and the deubiquitinase complex USP1 and UAF1 and inhibits the monoubiquitination of PCNA, thereby impairing the recruitment of Y family polymerases and increasing cell sensitivity to stimuli that trigger replication fork blockades."

reach
"The Usp1/UAF1 complex directly binds to modified FANCD2 and PCNA and catalyzes their deubiquitination [ 25 •• ]."

reach
"Our findings indicate that these NLSs mediate the nuclear localization of the USP1 and UAF1 complex, supporting a model in which USP1 and UAF1 form a complex in the cytoplasm that subsequently relocates to the nucleus through active nuclear import mediated by USP1 NLSs."

sparser
"Our results indicate that the S313 phosphorylation site is not critical for USP1 binding to UAF1 or for PCNA deubiquitination in a cellular environment."

sparser
"Since only in vitro evidence for S313 phosphorylation-regulated USP1/UAF1 interaction has been provided, we decided to evaluate the role that this phosphorylation site may have in regulating USP1 binding to UAF1 in a cellular context."

sparser
"In this regard, phosphorylation at the S313 residue has been recently reported to be critical for USP1 activity and interaction with UAF1 in an in vitro setting [ xref ]."

reach
"Two Nuclear Localization Signals in USP1 Mediate Nuclear Import of the USP1 and UAF1 Complex."

reach
"The crucial role that UAF1 binding plays as regulator of USP1 deubiquitinating activity raises the possibility that interfering with the formation of the USP1 and UAF1 complex may constitute a useful therapeutic strategy."

reach
"Inhibiting the function of the USP1 and UAF1 complex sensitizes cancer cells to chemotherapy, suggesting that this complex is a relevant anticancer target."

reach
"Here, we identify two nuclear localization signals (NLSs) in USP1 and show that these NLSs mediate the nuclear import of the USP1 and UAF1 complex."

reach
"Our data support a model in which USP1 and UAF1 form a complex in the cytoplasm that subsequently translocates to the nucleus through import mediated by USP1 NLSs."

reach
"First, the UAF1 interacting region of USP1 identified here might be targeted to disrupt the USP1 and UAF1 interaction with therapeutic purposes."

reach
"On the other hand, we describe a cellular relocation assay that can be easily implemented in a high throughput setting to search for drugs that may dissociate the USP1 and UAF1 complex."

reach
"However, the mechanisms that may modulate the nucleocytoplasmic localization of the USP1 and UAF1 complex have not yet been studied."

reach
"Further supporting this view, a recent report has shown that inhibiting the deubiquitinase activity of the USP1 and UAF1 complex sensitizes non small cell lung cancer cells to DNA damaging agents XREF_BIBR."

reach
"In addition to providing further insight into the biological consequences of the USP1 and UAF1 interaction, the results presented here have practical implications for the development of the USP1 and UAF1 as an anticancer target."

reach
"These results indicate that the two USP1 NLSs identified here mediate the import of the USP1 and UAF1 complex into the nucleus."

reach
"Recent studies have progressively elucidated the immunoregulatory roles of the USP1UAF1 complex."

reach
"USP1-associated factor 1 (UAF1), which is also called WD repeat-containing protein 48, can bind USP1, USP12, and USP46 and stimulate the activity of these ubiquitin-specific proteases (USPs) (6)."

reach
"Nonetheless, both studies revealed a critical role for the UAF1-RAD51AP1 interaction in promoting HR repair, through the DYDL sequence.In summary, this study : 1) dissected the new function of the USP1 and UAF1 complex in HR repair, 2) presents a new mode of regulation of HR repair proteins, and 3) supports the model that UAF1 is a substrate adaptation molecule for USP1."

reach
"Our results are consistent with a model in which USP1 and UAF1 form a complex in the cytoplasm that is subsequently translocated to the nucleus through active nuclear import mediated by the two USP1 NLSs (XREF_FIG)."

reach
"Such a model, obviously, implies that USP1 can bind simultaneously to the nuclear import receptors and to UAF1."

reach
"However, mutation of this sequence to AARAA did not abrogate UAF1 interaction (data not shown), indicating that the VERIV motif is not essential for USP1 and UAF1 complex formation."

reach
"The identification of the UAF1 binding domain in USP1 may be of clinical relevance in the light of the recent observation that interfering with the activity of the USP1 and UAF1 complex sensitizes non small cell lung cancer cells to DNA damaging agents XREF_BIBR."

sparser
"USP1 forms a complex with UAF1 in the cytoplasm, and then the complex translocates to the nucleus utilizing the two nuclear localization signals (NLSs) on USP1 ( xref )."

reach
"Interestingly, inhibition of Usp1, a close homologue of Usp12 has been shown to have promising results in lung cancer systems which was achieved by inhibiting the Usp1 and Uaf-1 complex [XREF_BIBR], [XREF_BIBR]."

reach
"As the USP1 and UAF1 complex is involved in TLS, pimozide and GW7647 may operate by limiting the ability of cancer cells to bypass CDDP induced DNA lesions."

reach
"On one hand, the UAF1 interacting region of USP1 identified here might potentially be targeted to disrupt the USP1 and UAF1 interaction with therapeutic purposes."

reach
"On the other hand, our findings provide the basis for a relocation assay that can be easily implemented in a high throughput setting to search for drugs that may dissociate the USP1 and UAF1 complex and thus interfere with the activity of this complex in tumor cells."

reach
"USP1 NLSs Mediate Nuclear Import of the USP1 and UAF1 Complex."

reach
"We hypothesized that the USP1 NLSs could mediate the nuclear import of the USP1 and UAF1 complex."

reach
"Here, we have used this assay to map the UAF1 binding motif in USP1, but such an assay can also be applied to evaluate the ability of different drug treatments to interfere with the formation of the USP1 and UAF1 complex (XREF_FIG)."

reach
"Indeed, several inhibitors of the USP1 and UAF1 complex have been developed to target the FA pathway."

reach
"Inhibiting the USP1 and UAF1 complex also compromises TLS by increasing levels of monoubiquitinated PCNA, another substrate of USP1, suggesting that inhibition of the USP1 and UAF1 complex can simultaneously target two major steps in the FA pathway."

reach
"This hyper-activation is not observed for the USP1 and UAF1 complex."

sparser
"Whether the UAF1 binding to USP1 is also involved in Snail de-ubiquitination and whether USP1 phosphorylation on S42 and/or S331 affects USP1/UAF interaction have not yet been clarified and certainly need to be addressed in future studies."

reach
"UAF1/USP1 complex activates NLRP3 after deubiquitylation through Lys48 poly-ubiquitin chains and thus increases the level of NLRP3 intracellular levels by protecting it from degradation (Song et al., 2020)."

sparser
"The similar values of substrate K M (within a factor of 2) for USP1 and the USP1-UAF complex, as well as the closely similar Ub-al Ki values for the two enzyme forms, show that the formation of USP1-U[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"In contrast, replacement with aspartic acid, a phosphomimetic residue, promotes formation of a complex with similar kinetic properties as the wild type USP1-UAF1 complex."

reach
"USP1 associates with its activating factor UAF1, and the USP1 and UAF1 complex removes monoubiquitin from FANCD2 to complete the repair (XREF_FIG)."

reach
"Indeed, RAD51AP1 was identified as a ubiquitinated protein in a proteomic study.40 However, we were unable to detect the ubiquitinated species of RAD51AP1, thus we are not able to conclude whether the USP1 and UAF1 complex protects RAD51AP1 from proteasomal degradation by deubiquitination.RAD51AP1 promotes the HR repair by promoting the recombinase activities of RAD51 and its meiotic counterpart DMC1.4,5,19-21 Unlike the RAD51 paralogs (e.g. RAD51C), depletion of RAD51AP1 does not affect the foci formation of RAD51.4,5 Thus it was proposed that RAD51AP1 acts at a later step of the RAD51-ssDNA nucleofilament formation."

sparser
"GSK1904529 (IGF1R inhibitor), Lapatinib (EGFR inhibitor), Linsitinib (IGF1R inhibitor), ML323 (selective USP1UAF1 inhibitor), Dihydrorotenone (mitochondrial inhibitor), OSI − 027 (mTOR inhibitor), TAF1_5496 (TAF1 inhibitor), Tamoxifen (selective estrogen response regulator), Vorinostat (Histone deacetylases inhibitor), Gallibiscoquinazole, OF − 1-1853, Sinularin (natural product from soft coral) were more resistant in high-risk group in GSE39582 and GSE17536 cohorts (Fig.  xref C, xref )."

sparser
"In summary, this study: 1) dissected the new function of the USP1-UAF1 complex in HR repair, 2) presents a new mode of regulation of HR repair proteins, and 3) supports the model that UAF1 is a substrate adaptation molecule for USP1."

reach
"In this study, we define an interaction between UL138 and USP1-associated factor 1 (UAF1) and ubiquitin specific peptidase 1 (USP1)."

reach
"These results validate the interaction between UL138, UAF1, and USP1."

sparser
"BRCA1 recruitment in S/G2 is also regulated by USP1UAF1, which antagonises K63-Ub chains that serve as recruiters for BRCA1."

reach
"Contrary to this finding, we find that inhibition of USP1 with C527 increased TB40/E genome replication in CD34+ HPCs, suggesting that at least the UAF1-USP1 complex is suppressive to viral genome replication, consistent with the role of UL138 in suppressing replication [17]."

reach
"Therefore, it is possible that the UAF1-USP1 complex might deubiquitinate STAT1 to promote its interaction with and subsequent activation by IFNAR."

reach
"In vitro, the USP1 and UAF1 complex can de-ubiquitinate mono-ubiquitinated PCNA more efficiently than USP1 alone [59]."

reach
"ELG1 (enhanced level of genomic instability 1) interacts with the USP1 and UAF1 complex and supports USP1 mediated de-ubiquitination of PCNA [62]."

sparser
"Using HPV-11, Orav et al. demonstrated that this requires interactions between the E1 protein of HPV-11 and a member of the Fanconi anemia DNA repair pathway, the cellular UAF1-USP1 complex [ xref ]."

reach
"In this study, we showed that the previously described interaction between the viral E1 helicase and the cellular UAF1 and USP1 complex is necessary for the completion of bidirectional replication and the subsequent initiation of the unidirectional replication mechanism."

reach
"While the UAF1-USP12 and UAF1-USP46 complexes regulate histone deubiquitination and the cell survival- and growth promoting Akt pathway, the UAF1 and USP1 complex is involved in the Fanconi anemia (FA) DNA repair pathway."

reach
"The FA ID complex is deubiquitinated by the UAF1 and USP1 complex."

sparser
"The interactions of USP1 and UAF1, an essential cofactor of USP1, with TonEBP were confirmed by coimmpunoprecipitation (coIP) ( xref E, xref C, and S1E)."

reach
"Whether the UAF1 binding to USP1 is also involved in Snail de-ubiquitination and whether USP1 phosphorylation on S42 and/or S331 affects USP1/UAF interaction have not yet been clarified and certainly need to be addressed in future studies.Recent studies suggest that the cancer stem cell status is not static but dynamic, that the differentiation into a cancer stem cell is not a monodirectional process, and that bulk cancer cells can also be reprogrammed into stem cells, exploiting plasticity programs (35)."

reach
"ML323, a selective small-molecule inhibitor of the UAF1 and USP1 complex, was used to test the requirement for USP1 activity in the replication of the HPV11 WT and VE mutant genomes."

reach
"The UAF1 and USP1 complex plays a critical role in the FA DNA repair pathway, and it is possible that HPV11 RIs containing two converged theta replication forks are mistaken for ICLs and that the FA pathway is recruited to resolve the structures."

reach
"To our knowledge, these compounds are the first reported inhibitors against USP1-UAF1, and they demonstrate the feasibility of inhibiting the USP1 and UAF1 complex with small molecules."

reach
"Indeed inhibition of the Usp1 and Uaf-1 complex sensitises cells to chemotherapy."

reach
"The results presented in this paper also revealed clues as to how the interaction between E1 and the UAF1 and USP1 complex might lead to the BLM mediated initiation of unidirectional replication."

reach
"By recruiting the UAF1 and USP1 complex to HPV replication forks, HPV11 may promote the deubiquitination of FANCD2 bound to viral genomes, thus promoting the engagement of BLM over the FA effector proteins."

reach
"We ruled out the possibility that ML323 acts as a disruptor of the USP1 and UAF1 complex by native gel analysis of USP1-UAF1 incubated with varied concentrations of ML323 up to 100 times its IC 50 (XREF_SUPPLEMENTARY)."

reach
"We determined the ML323 induced change in deuterium incorporation (DeltaHDX) of the USP1 and UAF1 complex in the presence and absence of ML323."

reach
"ATR-mediated phosphorylation of the USP1-UAF1 complex and of its substrate, ubiquitylated FANCI-D2, has also been shown to regulate the formation of the enzyme-substrate complex for efficient Ub removal.Related to USP1-UAF1, is how some DUBs function in multiple different DDR signalling pathways."

reach
"Notably, ML323 did not inhibit the USP46 and UAF1 complex that contains the same UAF1 subunit as the USP1 and UAF1 complex and demonstrated little inhibition against USP1 alone (IC 50> 200 muM) in a Ub-Rho assay in which USP1-UAF1 and USP1 demonstrated similar K m values."

sparser
"So far there is no structural information available for USP1 alone or the USP1UAF1 complex, but crystal structures of the paralogs with and without the activators have been solved (Yin et al , xref ; Li et al , xref ; Dharadhar et al , xref )."

sparser
"Based on these USP12 and USP46 structures, it is clear that UAF1 binds USP1 on the “finger” sub‐domain of the catalytic domain (Yin et al , xref )."

sparser
"The second possibility is that ELG1 enhances or stabilizes the interaction between USP1 and its activating factor UAF1."

sparser
"Very few USP structures where substrates have been trapped are known to date and include USP30, USP22, and USP1UAF1 complexes with Lys 6 -linked diubiquitin ( xref ), mono-ubiquitinated H2B ( xref ), and mono-ubiquitinated FANCI − FANCD2 ( xref ), respectively."

sparser
"To this end, we employed ML323, a selective, allosteric inhibitor of the USP1-UAF1 de-ubiquitinase complex ( xref )."

sparser
"DNA binding was shown to promote USP1UAF1 activity on FANCD2‐Ub, but in this case, activation is dependent on a DNA‐binding role of UAF1 (Liang et al , xref )."

reach
"Three Usp1 inhibitors explored this strategy, Pimozide [XREF_BIBR], ML323 [XREF_BIBR], [XREF_BIBR], [XREF_BIBR] and GW7647 [XREF_BIBR] each targeting the Usp1 and Uaf-1 complex in a non competitive manner."

reach
"Thus, our study uncovers a positive feedback mechanism of cGAS-dependent antiviral responses and suggests the UAF1-USP1 complex as a potential target for the treatment of diseases caused by aberrant cGAS activation."

sparser
"We first employed ML323, which inhibits the USP1UAF1 interaction."

reach
"UAF1 SLD2 binds directly to a SIM in FANCI, bridging the interaction between USP1 and UAF1 and FANCD2 and FANCI."

reach
"Both USP12 and USP46 were found to individually associate with all 3 WD40 proteins and both phosphatases, while USP1 associated with WDR48 and PHLPP but not WDR20, DMWD, or PHLPPL."

sparser
"USP1 forms a complex with UAF1 where its WD40 domain binds and stimulates the ubiquitin protease activity of USP1 to counteract the monoubiquitination of the FANCD2-FANCI heterodimer [ xref ]."

reach
"The rise in ubiquitin corresponds to a decrease in the abundance of the de and ubiquitinating complex USP1 and WDR48, which targets FANCD2."

sparser
"USP1UAF1 is known to play a role in PCNA‐Ub‐mediated translesion synthesis (Huang et al , xref )."

reach
"Deubiquitination of FANCD2 may be attributed to the key role of USP1 and UAF1 complex in regulating HR repair, however whether USP1-UAF1 promotes HR repair independently of FANCD2 deubiquitination is not known."

reach
"These data support models in which the Usp1 and UAF1 complex is indirectly recruited to PCNA-Ub."

sparser
"Moreover, it has been suggested that USP1UAF1 travels with elongating replication forks where it deubiquitinates its substrates."

reach
"The difference in cross linking pattern between the copurified USP1 and UAF1 complex and individually purified USP1 suggests that Ser313 likely undergoes dephosphorylation when being individually expressed and purified from insect cells."

sparser
"To validate our kinetic modelling, we performed EMSA‐based binding analysis for USP1UAF1) on DNA which confirmed that USP1UAF1 has stronger binding compared to USP1 alone (Fig  xref )."

sparser
"Elg1, a protein related to RFC1, the large subunit of Replication Factor C, also interacts with USP1 and UAF1."

sparser
"Hence, a deubiquitinase complex, USP1 and UAF1 complex, eliminates the K48-connected poly-ubiquitination of TBK1, blocks TBK1 protein degradation, and increases anti-viral responses [73] ."

reach
"The USP1 and UAF1 complex reduces the accumulation of mono-ubiquitinated PCNA during normal cell division, which prevents mutagenesis by error-prone TLS polymerases."

reach
"hELG1 was found in association with the USP1 and UAF1 complex and down regulates PCNA mono-ubiquitination."

reach
"hELG1 's role in PCNA de-ubiquitination is mediated by its interaction with the USP1 and UAF1 complex, which has been shown to work in de-ubiquitination [95]."

reach
"Several studies have demonstrated that hELG1 specifically directs the USP1 and UAF1 complex to PCNA at the damage site and in doing so initiates the switch from the TLS polymerases to the replicative polymerases."

reach
"Interestingly, UAF1 contains a tandem repeat of SUMO like domains at its C-terminus which bind to a SIM (SUMO Interacting Motif) on hELG1 allowing the recruitment of the USP1 and UAF1 complex to the u[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"When FANCD2- Ubi –FANCI- Ubi is dissociated from the DNA, USP1-UAF1 removes ubiquitin from FANCD2- Ubi –FANCI- Ubi [ xref ]."
| PMC

sparser
"Thus, we propose that the interaction with RAD51AP1 at the post-RAD51 foci formation step is an additional mechanism that regulates the HR repair by the USP1-UAF1 complex, independently of FANCD2 deubiquitination."

sparser
"USP1-813 UAF1 deubiquitinase complex stabilizes TBK1 and enhances antiviral responses."
| DOI

sparser
"More importantly, inhibition of USP1 with its inhibitor ML323, a specific USP1-UAF1 inhibitor, significantly inhibited the glycolysis and proliferation of T-ALL cells."

reach
"The USP1 and UAF1 complex deubiquitylates the Fanconi anemia protein FANCD2, which promotes homologous recombination (HR) and DNA cross-link repair."

sparser
"The data in this work suggest that the interaction of Elg1 with USP1UAF1 down-regulates PCNA mono-ubiquitination (see article by K.J. Myung in this issue for further discussion)."

sparser
"For deubiquitination, USP1-UAF1 could efficiently remove ubiquitin from FANCD2- Ubi –FANCI, but not FANCD2- Ubi –FANCI- Ubi [ xref ]."
| PMC

sparser
"Interestingly, we note that not only USP1UAF1 is activated by DNA loading of PCNA, but also USP1 alone, which shows an increase in catalytic efficiency of from 2.3 mM −1  s −1 to 4.8 mM −1  s −1 ."

sparser
"Thus, the potential roles of the UAF1-USP1 deubiquitinase complex in spermiogenesis remain unclear."

sparser
"UAF1 interacts with USP1 in mouse testes."

sparser
"Additionally, we constructed an HA-tagged Uaf1 knock-in mouse, and co-IP (co-immunoprecipitation) in UAF1-HA mice testes again showed an interaction between UAF1 and USP1 ( xref E)."

reach
"Thus, the USP1/UAF1 complex does not dissociate in response to UV irradiation.In the above experiments, we observed that the exogenously expressed USP1 protein ( Figure 6 A) was more stable after UV i[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"Ubiqutination-linked phosphorylation of S559/S565 occurs predominantly downstream of monoubiquitination, and acts to inhibit FANCD2 de-ubiquitination by USP1-UAF1 and bypassed the need to deubiquitinate FANCD2 to achieve effective ICL repair [ xref ]."
| PMC

sparser
"We verified the association of UAF1 and USP1 in testes by co-IP and immunoblotting, confirming (1) that USP1 was present among the immunoprecipitated proteins from wild-type (WT) testes using an anti-UAF1 antibody and (2) that UAF1 was present among the immunoprecipitated proteins from WT testes using an anti-USP1 antibody ( xref C and 1D)."

sparser
"Building upon our identification of UAF1 and USP1 interaction, we further analyzed their expression dynamics."

sparser
"Subsequent immunoprecipitation assays using testicular extracts from PD3, PD6, and PD8 mice revealed that the interaction between UAF1 and USP1 begins before PD8 and becomes more pronounced by this stage."

reach
"We speculate that the binding of UAF1 to USP1 induces a conformational change of the enzyme resulting in a robust increase in enzymatic activity."

reach
"The active USP1/UAF1 complex ensures that the Fanconi anemia pathway is kept at an adequate state under normal cell growth."

sparser
"As shown in xref , the de-ubiquitination of R1285Q ID Ub by USP1-UAF1 is indeed significantly faster than the wild type ID Ub ."

reach
"When cells experience genotoxic stress, transcription of the USP1 gene is rapidly shut off, leading to a decrease of USP1/UAF1 complex, thereby activating the Fanconi anemia pathway through increased [MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"Co-IP in the ovary confirmed the interaction between UAF1 and USP1 in WT ovaries ( xref D)."

sparser
"We observed that UAF1 and USP1 interact in the ovary."

sparser
"Usp1 forms a heterodimeric complex with Uaf1 ( U sp1- a ssociated f actor 1 ) (Wdr48), a protein containing WD40-type repeats ( xref , xref ; xref )."

sparser
"In both USP1 and USP1UAF1, the enzyme is faster on DNA‐loaded PCNA‐Ub, but the increase in activity due to DNA loading is higher in the presence of UAF1: fivefold increase in USP1UAF1 and a twofold increase in USP1 alone (Table  xref , Fig  xref )."

sparser
"For optimized catalytic activity, USP1 forms a heterodimeric complex with UAF1 ( xref ), a WD40 repeat-containing protein that also stimulates USP46 and USP12 ( xref )."

reach
"For USP1 and USP1/UAF1 complex purification, cell pellets were resuspended in lysis buffer (50 mM Tris-HCl [pH 8.0], 150 mM NaCl, 10 mM BME, 10 mM imidazole, 10% glycerol, and 0.2% Triton X-100) and s[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"USP1-WDR48 deubiquitinase complex enhances TGF-β induced epithelial-mesenchymal transition of TNBC cells via stabilizing TAK1."

sparser
"These drugs included various small molecule inhibitors, including MEK/ERK pathway inhibitors (selumetinib, trametinib, and VX‐11e), a survivin inhibitor (sepantronium bromide), a CDK1 inhibitor (RO‐3306), a BCR‐ABL inhibitor (nilotinib), a USP1UAF1 inhibitor (ML 323), an estrogen receptor antagonist (fulvestrant), a PORCN/Wnt inhibitor (Wnt‐C59), an IDH inhibitor (AGI‐6780), and cisplatin sensitivity (oxaliplatin; Figure  xref )."

sparser
"Finally, the monoubiquitinated ID2 complex is deubiquitinated by USP1-UAF1 (ubiquitin specific protease 1 — USP associated factor 1), which promotes its clearance [ xref ] ( xref , subsections 1 and 2)."

sparser
"The USP1UAF1 complex deubiquitinates several substrates involved in DNA damage response, including monoubiquitinated PCNA and FANCD2 ( xref )."

sparser
"To evaluate USP1 as a potential drug target, we synthesized previously reported inhibitors of the USP1UAF1 complex, ML323 and I-138 ( xref ; refs. xref )."

sparser
"In an Ub-Rho cleavage assay, these compounds significantly inhibited USP1UAF1 ( xref )."

reach
"FANCD2-I proteins are then deubiquitinated by the USP1-UAF1 complex [91,92,93,94,95,96]."

sparser
"We investigated the mode of USP1UAF1 inhibition by I-138 using mutual exclusivity experiments."

sparser
"These results strongly suggest that I-138 binds to USP1UAF1 at an allosteric pocket synergistically with ubiquitin, likely at the same site as ML323 ( xref )."

sparser
"Fifty percent of inhibition of the closely related USP12–UAF1 and USP46–UAF1 complexes only occurred at 10 μmol/L compared with USP1-UAF1 IC 50 of 4.1 nmol/L, reflecting a >2,000-fold selectivity."

reach
"There is evidence that suggests that the USP1 and UAF1 complex, previously discussed above to be involved in the FA pathway and TLS, promotes DSB repair via HR."

sparser
"The SLDs of UAF1 interact with the SIMs of FANCI to promote the recruitment of USP1-UAF1 and de-ubiquitination of ID2 [ xref ]."

sparser
"These data show that I-138 is a potent and selective USP1UAF1 inhibitor (abbreviated as USP1 inhibitor) displaying cellular target engagement."

reach
"Positive regulators like neuregulin receptor degradation protein 1 (Nrdp1), RING finger protein 128 (RNF128), tyrosine kinase Src, Raf kinase inhibitory protein (RKIP), Glycogen Synthase Kinase-3 Beta (GSK3β), DNA methyltransferases 3A (Dnmt3a), histone deacetylase 3 (HDAC3), and tripartite motif protein 9 (TRIM9) could enhance the autophosphorylation or activation, and USP1-UAF1 complex could stabilizes TBK1 to enhance its function."

sparser
"Moreover, H 2 O 2 treatment did not affect the association of Usp1 with UAF1 ( xref ), suggesting that inhibition of USP1 by oxidative stress is not likely caused by ROS-mediated disruption of the Usp1-UAF1 complex."

sparser
"Therefore SIM-SLD interactions support the ubiquitin USP1-UAF1 dependent removal of ID2 and in parallel, SUMO conjugation and ubiquitination provide an alternative route of ID2 extraction from chromatin ( xref , subsection 4)."

reach
"To explain this phenomenon, it has been suggested that HU disrupts the interaction between USP1 and its activating partner protein UAF1 (USP1 associated factor 1) [XREF_BIBR, XREF_BIBR]."

sparser
"D. Model: The USP1-UAF1 complex promotes the HR repair in multiple mechanisms. CELL CYCLE 2643 to the DNA crosslinking agent MMC."

sparser
"Notably, ID1 is a substrate of USP1 [ xref ], and blockade of USP1 by ML323 (a selective inhibitor of the USP1-UAF1 complex) not only promoted CD8 + T-cell infiltration and function but also markedly inhibited the liver metastasis of CRC in an orthotopic liver metastatic syngeneic mouse model [ xref ] ( Table  xref ) ."

sparser
"Lastly, we present evidence that E1 promotes unidirectional replication not only through its association with UAF1-USP1 but also by binding to the Bloom (BLM) helicase."

reach
"This binding event was suggested to protect PCNA-Ub from deubiquitylation by the USP1/UAF1 complex to drive the stress response that is dependent on the ubiquitin signal [71] ."

reach
"Taken together, ELG1 specifically directs USP1 and UAF1 complex for PCNA deubiquitination."

reach
"Last year, great strides were made in understanding the functional significance of ELG1 when it was discovered that the human homolog of ELG1 (which is also called ATAD5) not only interacts with PCNA and colocalizes with the sliding clamp at stalled replication forks with distinct foci structures in the nucleus, but also associates with the USP1 and UAF1 complex."

reach
"The second possibility is that ELG1 enhances or stabilizes the interaction between USP1 and its activating factor UAF1."

sparser
"Deubiquitination of FANCD2 may be attributed to the key role of USP1-UAF1 complex in regulating HR repair, however whether USP1-UAF1 promotes HR repair independently of FANCD2 deubiquitination is not known."

reach
"Equally important as PCNA ubiquitylation is PCNA deubiquitylation, which is carried out by the USP1 and UAF1 complex in conjunction with ELG1."

reach
"For high-throughput screening, we used the recombinant USP1 and UAF1 complex in a 4 muL assay volume to screen several libraries of 9,525 bioactive small molecules in qHTS mode, by testing each compound as a dilution series spanning the concentration range of 0.46 muM to 114 muM (see Experimental Procedures)."

reach
"Additionally, we constructed an HA-tagged Uaf1 knock-in mouse, and co-IP (co-immunoprecipitation) in UAF1-HA mice testes again showed an interaction between UAF1 and USP1 (Figure 1E)."

reach
"The DUB complex USP1/USP1-associated factor 1 (UAF1) deubiquitylates PCNA after the complex is recruited to the substrate by recruitment factor human ELG1 [14] ."

reach
"A final example of negative regulation is the deubiquitylation of monoubiquitylated PCNA (PCNA-Ub) by the USP1/UAF1 complex."

reach
"Likewise, the activity of the deubiquitinating complex USP1 and UAF1 over FANCD2 keeps the FA pathway in check 104, 105."

sparser
"Although we cannot exclude the possibility of a UAF1‐ssDNA binding event promoting ID2 deubiquitination, there is no evidence in support of dsDNA‐UAF1 binding influencing ID2 deubiquitination: cryo‐EM analysis of in vitro assembled USP1UAF1‐ID2 Ub ‐DNA complexes, suggests that formation of a ternary complex is favoured, with both dsDNA and USP1UAF1 preferentially binding ubiquitinated ID2, rather than each other (Rennie et al ,  xref )."

reach
"The WDR48USP1 complex acts as a regulator during DNA damage, especially in translation synthesis and Fanconi anaemia pathway."

reach
"In cluster 1, one of the highly ranked complexes searched against CORUM is the USP1 and UAF1 complex which was reported as a schizophrenic candidate complex, and its inhibitor, Pimozide, is one of the antipsychotics with the neuroleptic property of treating schizophrenic patients [XREF_BIBR]."

reach
"We further studied the molecular mechanism of WDR48‐mediated progression of HCC through the prediction of BioGrid and HitPredict data sets and found that c‐Myc might interact with the WDR48/USP1 complex."

reach
"7 The interaction of the WDR48/USP1 complex with the substrate mainly depended on the C‐terminal domain of the WDR48‐targeting substrate, and WDR48 was distributed in the cytoplasm and nucleus."

sparser
"Moreover, the USP1-WDR48 complex was found to downregulate the polyubiquitination of TAK1 and mediate its in vitro stability."

reach
"It has been postulated that the USP1 and UAF1 complex is associated with pathogenesis for schizophrenia."

sparser
"For example, the CDK1-mediated serine phosphorylation of USP1 disrupts the interaction between USP1 and UAF1."

reach
"incubation on ice, 400 nM of the USP1 and UAF1 complex (Boston Biochem, E-568) was added."

sparser
"For the protection seen on I Ub in the absence of D2, there is a possibility that dsDNA blocks access of USP1UAF1 to FANCI's ubiquitin, either directly or indirectly through altering the conformation of I Ub ."

sparser
"In mice peritoneal macrophages, viral infection triggers the activation of the UAF1-USP1 deubiquitinase complex, which specifically combines with cGAS to cleave its K48-linked polyubiquitination, enhancing its protein level and cGAS-dependent type I IFN responses."

sparser
"In 2014, Liang et al. claimed ML323 (42) ( Fig. 18 ) was a highly potent and selective inhibitor of the USP1UAF1 deubiquitinase complex [ 177 ]."

sparser
"Antiviral responses induced by cGAS are inhibited by a deficiency in Uaf1 and inhibitors of the UAF1-USP1 deubiquitinase complex, thus promoting viral infection [ xref ]."

sparser
"ML323 (42) was a nanomolar inhibitor of USP1UAF1 with IC 50 = 76 nM."

sparser
"12 The USP1-UAF1 complex also deubiquitinates FANCI, which interacts with FANCD2,13 and a replicative polymerase processive factor PCNA."

reach
"Disruption of the USP1 and UAF1 complex leads to DNA repair defects similar to those of mutants in the FA pathway."

reach
"The Usp1 and UAF1 complex is recruited to PCNA-Ub via an interaction of UAF1 with Elg1, an alternative component of the clamp loader."

reach
"ATAD5, in cooperation with the UAF1-USP1 complex, is responsible for this de-ubiquitination."

reach
"It is well established that ATAD5-RLC is responsible for unloading PCNA and that ATAD5 interacts with the UAF1-USP1 complex to facilitate the de-ubiquitination of Ub-PCNA."

sparser
"On the right are the representative images. immunoprecipitation also reversely pulled down both UAF1 and USP1, suggesting that RAD51AP1 associate with the USP1-UAF1 complex (Fig. 2D)."

sparser
"ML323 binding to USP1-UAF1 also induced alterations in deuterium incorporation in the UAF1 subunit."

sparser
"Discussion Here we described a functional interaction between RAD51AP1 and the USP1-UAF1 deubiquitinating enzyme complex."

reach
"The C-terminal SLD (SUMO-likedomain) domain of UAF1 interacts with the SIM (SUMOinteracting motif) of FANCI,23 as well as the SIM of ELG1.28 Thus the SUMO like delivery system was proposed as a mecha-nism for the USP1 and UAF1 complex to recruit its substrates fromthese studies."

sparser
"The USP1-UAF1 protein complex is an important regulator in DNA damage response pathways [ xref ]."

reach
"Thus, human Elg1 regulates PCNA deubiquitination by recruiting the USP1 and UAF1 complex to ubiquitinated PCNA on chromatin."

sparser
"Notably, ML323 did not disrupt the association of the USP1 and UAF1 subunits in the complex."

sparser
"ML323 is a highly effective inhibitor of USP1-UAF1 deubiquitinase complex, blocking the allosteric activation pathway may be another strategy to study and discover USP inhibitors [ xref , xref , xref ]."

sparser
"The FA pathway has at least five steps that are exemplary of the repair-replication interface: 1) collision of replication machinery with a blocking lesion; 2) activation of FA core complex ubiquitin [MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"The USP1 and UAF1 complex purified from insect cells was reduced with 100 mM dithiothreitol, alkylated with 150 mM iodoacetamide, and then digested with trypsin at 37 degreesC overnight."

sparser
"The purified UAF1-USP1 complex was indeed active in the Ub-AFC assay, and the activity was strongly and reversibly inhibited by H 2 O 2 ."

sparser
"Ubiquitination of FANCI, on the other hand, largely protects the ubiquitin on FANCD2 from USP1UAF1 deubiquitination, with key hydrophobic residues of FANCI's ubiquitin being important for this protection."

sparser
"19, 2636–2646 http://dx.doi.org/10.1080/15384101.2016.1209613 REPORT The USP1-UAF1 complex interacts with RAD51AP1 to promote homologous recombination repair Scott Cukrasa, Euiho Leea, Emily Palumboa, Pamela Benavideza, George-Lucian Moldovanb, and Younghoon Keea aDepartment of Cell Biology, Microbiology, and Molecular Biology, College of Arts and Sciences, University of South Florida, Tampa, FL, USA; bDepartment of Biochemistry and Molecular Biology, Pennsylvania State University College of Medicine, Hershey, PA, USA ABSTRACT USP1 deubiquitinating enzyme and its stoichiometric binding partner UAF1 play an essential role in promoting DNA homologous recombination (HR) repair in response to various types of DNA damaging agents."

sparser
"Removal of the ubiquitins, on FANCI and FANCD2, is also critical for the FA pathway, and this deubiquitination step is catalysed by the USP1UAF1 complex xref , xref ."

reach
"Since ATAD5 facilitates Ub-PCNA de-ubiquitination through the UAF1-USP1 complex, the effect of BAZ1B on de-ubiquitination may be reduced without USP1."

reach
"This configuration suggests that the ATAD5-BAZ1B interaction may hinder the binding of ATAD5 to the UAF1-USP1 complex."

sparser
"Thus, our study uncovers a positive feedback mechanism of cGAS-dependent antiviral responses and suggests the UAF1-USP1 complex as a potential target for the treatment of diseases caused by aberrant cGAS activation."

reach
"ATAD5 (1–603) significantly enhanced Ub-PCNA de-ubiquitination by the UAF1-USP1 complex, thereby supporting the role of ATAD5 in this process."

reach
"The binding of Uaf1 to Usp1 DUB strongly increases its deubiquitylating activity."

reach
"The DUB activity of autocleaved Usp1 can apparently be retained if the N-terminal fragment (denoted as Usp1 Nt) and Gln Usp1 Ct are held together within the cleaved Usp1 and Uaf1 complex."

sparser
"Interestingly, the interaction of USP1 and UAF1 was not affected by H 2 O 2 , suggesting that the inhibition is likely caused by oxidation of the catalytic active site of USP1, which is consistent with other DUBs ( xref ; xref ; xref )."

sparser
"Using phosphomimetic and phosphodead mutants of FANCI we show that S559 and S565 are particularly important for protecting the complex from the activity of the deubiquitinating enzyme USP1:UAF1."

sparser
"We further demonstrate that ubiquitination of FANCI largely protects the ID2 complex from USP1UAF1 deubiquitination, which likely contributes to the maintenance of ubiquitination‐associated ID2‐DNA binding enhancement in the cellular context."

reach
"The Role of the Complex USP1 and WDR48 in Differentiation and Proliferation Processes in Cancer Stem Cells."

reach
"Interestingly, the addition of BAZ1B-SMARCA5 complex inhibited Ub-PCNA de-ubiquitination by ATAD5 (1–603) and the UAF1-USP1 complex (Fig. 6d)."

reach
"We also show that a metabolic stabilization of Gln Usp1 Ct, a change that prolongs DUB activity of the autocleaved Usp1 in the Usp1 and Uaf1 complex, makes cells hypersensitive to UV irradiation."

reach
"Collectively, these findings suggest that the interaction between ATAD5 and BAZ1B-SMARCA5 complex interferes with the ability of ATAD5 to facilitate Ub-PCNA de-ubiquitination by the UAF1-USP1 complex."

reach
"If so, the Arg and N-end rule pathway acts, in this setting, as a regulator that determines the duration of DUB activity of the autocleaved Usp1 in the Usp1 and Uaf1 complex."

reach
"Setting up and exploring an Arg and N-end rule based in vitro ubiquitylation and degradation system, with the Usp1 and Uaf1 complex as its autocleaved substrate, is likely to be a particularly informative approach to mechanistic questions in this arena, and to some physiological questions as well."

reach
"Taken together, our data demonstrate the important regulatory role of the USP1 and UAF1 complex in HR repair through its regulation of the FANCD2-Ub and PCNA-Ub cellular pools."

reach
"Immunoblotting of the immunoprecipitate revealed the presence of endogenous USP1 as well as its cleavage product, confirming the presence of a native USP1/UAF1 complex ( Figure 1 C)."

reach
"ATAD5, in concert with the UAF1-USP1 complex, de-ubiquitinates Ub-PCNA at the final stage of DNA repair synthesis."

reach
"We observed that the majority of USP1 protein in the HeLa cells existed as a protein complex with UAF1 ( Figure 1 D), further underscoring the importance of the USP1/UAF1 complex.Because the majority [MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"In particular, phosphorylation of FANCI provides an on/off switch for the FA pathway activation, as it is required for the monoubiquitination of both FANCI and FANCD2 and also provides a protective mechanism toward the action of the deubiquitinating enzyme USP1:UAF1 ( xref , xref , xref )."

reach
"The USP1/UAF1 complex, on the other hand, exhibited a strong increase in activity compared to USP1 alone."

reach
"The activity of the USP1/UAF1 heterodimer was about 35-fold higher than the activity of USP1 alone ( Figure 3 C)."

reach
"Thus, USP1 appears to be an essentially inactive enzyme, or proenzyme, in the absence of UAF1, depending on its binding for deubiquitinating activity.The above experiments were performed using a prefo[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"To gain further insight into the dynamics of USP1/UAF1 complex formation and USP1 activation, we investigated the kinetics of UAF1 binding and activation of USP1."

sparser
"H 2 O 2 does not affect the interaction of USP1 with its co-activator USP1-associated factor 1 (UAF1), suggesting that the inhibition is likely caused by oxidation of the USP1 active site [ xref , xref ]."

reach
"Such a scenario reveals UAF1 as the regulatory factor of USP1.In order to better understand the enzymatic consequences of the complex formation between UAF1 and USP1, we determined the kinetic paramet[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"In this study, we show that viral infection induces the expression of the UAF1-USP1 deubiquitinase complex in primary peritoneal macrophages (PMs) of C57BL/6J mice."

reach
"These findings imply that BAZ1B regulates the ATAD5-UAF1 interaction.Next, we conducted an in vitro Ub-PCNA de-ubiquitination assay with UAF1-USP1 complex, ATAD5 (1–603), and BAZ1B-SMARCA5 complex (Fig. 6d, Supplementary Fig. 7e)."

sparser
"Concordantly, the UAF1-USP1 deubiquitinase complex enhances cGAS-dependent type I IFN responses in PMs."

sparser
"Uaf1 deficiency and ML323 (a specific inhibitor of UAF1-USP1 deubiquitinase complex) attenuates cGAS-triggered antiviral responses and facilitates viral replication both in vitro and in vivo."

reach
"As a result, the k cat /K M is 35-fold higher for the USP1/UAF1 complex compared to USP1 alone."

reach
"In contrast, the USP1/UAF1 complex readily deubiquitinated the substrate ( Figure 5 B, lane 3)."

reach
"The proper association of UAF1-USP1 complex to ATAD5 is crucial for the de-ubiquitination of Ub-PCNA; any alterations in this interaction by BAZ1B may inhibit the de-ubiquitination."

reach
"Our finding that USP1 is activated by UAF1 prompted us to test whether the USP1/UAF1 complex is capable of deubiquitinating this physiologic substrate."

reach
"We next subjected the purified Ub-FANCD2 protein to an in vitro deubiquitinating reaction, using either USP1 enzyme alone or the USP1/UAF1 complex, both purified from Sf9 cells."

reach
"Our finding that the USP1 enzyme is nearly inactive, unless it is a part of the USP1/UAF1 complex, prompted us to investigate whether the complex dissociates after DNA damage."

reach
"The USP1/UAF1 complex was relatively stable after UV irradiation, and the levels of UAF1 were directly proportional to the levels of USP1 ( Figure 6 A)."

reach
"For the UAF1-USP1 complex, N-terminal 10xHIS-tagged UAF1 and N-terminal 2xStrepII-tagged USP1 were cloned into the pFL multi-gene expression vector and expressed from a single construct."

reach
"In contrast, two other known USP1 serine phosphorylations (Ser42 and Ser67) are dispensable with respect to the activity of the USP1 and UAF1 complex."

sparser
"It has been postulated that the USP1-UAF1 complex is associated with pathogenesis for schizophrenia."

reach
"First, the protease complex USP1 and UAF1 deubiquitinates monoubiquitinated PCNA."

sparser
"The UAF1-USP1 Deubiquitinase Complex Stabilizes cGAS and Facilitates Antiviral Responses."

reach
"In 2001, Chen et al. identified pimozide and GW7647 as the first inhibitors of the human USP1-UAF1 complex with low micromolar affinity."

reach
"These compounds demonstrate the possibility of inhibiting the USP1-UAF1 complex with small molecules and synergize with cisplatin in cisplatin-resistant cells."

sparser
"FANCI ubiquitination protects ID2 Ub complex against USP1UAF1 deubiquitination."

reach
"An S313D phosphomimetic mutation in USP1 can substitute for Ser313 phosphorylation in promoting the formation of the USP1 and UAF1 complex."

reach
"However, the potential role of USP1 and UAF1 complex in the immune system, especially in the innate antiviral immunity, remains unknown."

reach
"Finally, in the last step, the deubiquitinating (DUB) complex USP1 and UAF1 removes monoubiquitin from the ID complex, allowing for pathway regeneration."

reach
"A recent publication features the crystal structure of human USP1-UAF1 complex with and without ubiquitin, and one that is bound to monoubiquitinated FANCI-FANCD2 59 ."

reach
"The pSer313 dependent formation of USP1 and UAF1 complex points to a new approach of inhibiting USP1 activity by disrupting the interaction between the UAF1 's WD40-repeat domain and the Ser313 containing phosphopeptide in USP1."

reach
"To date, how USP1 and UAF1 interact and how the formation of the USP1 and UAF1 complex is regulated remains largely unknown."

reach
"Collectively, these data illustrate the dynamic changes of UAF1 and USP1 expression during viral infection, suggesting that USP1 and UAF1 complex is potentially involved in the regulation of antiviral responses."

reach
"USP1 and UAF1 complex facilitates IFN-beta production."

reach
"We found that Ser313 phosphorylation plays an essential role in regulating the formation of the USP1 and UAF1 complex."

reach
"UAF1 is a WD40-repeat containing protein that also binds to and regulates two other DUBs, USP12 and USP46 [XREF_BIBR], but its best characterized role is as a USP1 cofactor in the context of a USP1 and UAF1 complex."

reach
"UAF1 and USP1 knockdown both dramatically attenuated SeV- and ISD induced Tyr701 phosphorylation of STAT1, indicating the effect of USP1 and UAF1 complex is consistent for IFN-beta-mediated signaling (XREF_FIG)."

reach
"Likewise, the SLD2 domain of UAF1 also binds to the SIM on hELG1 to direct USP1 and UAF1 binding to another important substrate, PCNA-Ub."

sparser
"We hypothesized that since FANCI ubiquitination does not further enhance ID2‐DNA binding, its primary role may be in protecting FANCD2's ubiquitin from USP1UAF1 activity."

sparser
"To examine to what extent the presence of I or I Ub influences D2 Ub deubiquitination, we compared the progress of D2 Ub , ID2 Ub and I Ub D2 Ub deubiquitination by USP1UAF1 (in the presence of dsDNA) in a time course (Fig  xref A)."

reach
"These data indicate that USP1 and UAF1 complex targets molecules upstream of IRF3 to inhibit IFN-beta production."

reach
"Therefore, we speculate that TBK1 may be a target of USP1 and UAF1 complex."

reach
"USP7 and USP11 may associate transiently with the ATAD5–UAF1USP1 complex."

sparser
"To validate a novel role of USP1 along this RSK1-FLT3 axis, we treated MV4-11 and MOLM-13 cell lines with USP1 inhibitor SJB3-019A which reduced expression of FLT3 and USP1-UAF1 complex and suppressed FLT3 and S6 phosphorylation (Fig. xref )."

reach
"As shown in XREF_FIG, three discrete bands corresponding to the USP1 and UAF1 complex and the individual UAF1 and USP1 proteins were detected in native gel electrophoresis and Coomassie blue staining."

reach
"To further clarify the target of USP1 and UAF1 complex, we examined the association between USP1-UAF1 and several adaptors."

reach
"For USP1 (S313A) and UAF1 complex, no band coressponding to the USP1 and UAF1 complex was detected."

reach
"For USP1 (S313D) and UAF1 complex, a band similar to that of the wild-type USP1 and UAF1 complex was detected."

sparser
"According to our data, this may occur because FANCD2's ubiquitin is no longer sufficiently protected against USP1UAF1 deubiquitination."

sparser
"At the same time, phosphorylation can promote the interaction of USP1 and its cofactor UAF1, whereas the binding of OTULIN to LUBAC is blocked by phosphorylation."

reach
"For the wild-type USP1 and UAF1, a single band corresponding to the complex was detected with both antibodies (XREF_FIG, panel B and C), confirming the presence of both USP1 and UAF1 polypeptides in the native gel band assigned as USP1 and UAF1 complex."

reach
"In marked contrast, a band corresponing the USP1 and UAF1 complex was absent for USP1 (S313A) and UAF1 complex in both the Commassie blue stained gel (XREF_FIG, panel A) and the Western blotting gels (XREF_FIG, panel B and C)."

reach
"USP1 and UAF1 complex possesses potent deubiquitinase activity."

sparser
"FANCD2-I proteins are then deubiquitinated by the USP1-UAF1 complex [ xref , xref , xref , xref , xref , xref ]."

reach
"A 227-fold decrease in k cat / K m was evident when compared to the wild-type USP1 and UAF1 complex."

reach
"ML323, a selective USP1-UAF1 inhibitor, was used to inhibit deubiquitinase activity of USP1 and UAF1 complex."

reach
"USP1 and UAF1 complex facilitates IRF3 activation."

reach
"To investigate the physiological function of USP1 and UAF1 complex in IRF3 activation, mouse peritoneal macrophages were transfected with UAF1 or USP1 siRNA."

reach
"Unlike the USP1 and UAF1 complex, USP1 (S313A) exhibited a catalytic efficiency (k cat / K m = 4.8 x 10 3 M -1 s -1) similar to that of the wild-type and USP1 (S313D) (XREF_TABLE)."

reach
"A phosphomimetic USP1 (S313D) mutant can substitute Ser313 phosphorylation in promoting formation of the USP1 and UAF1 complex."

reach
"The USP1 and UAF1 complex also deubiquitinates PCNA-Ub, and deubiquitination requires the PCNA binding protein hELG1."

reach
"The SLD2 sequence of UAF1 binds to a SIM on hELG1, thus targeting the USP1 and UAF1 complex to its PCNA-Ub substrate."

reach
"To further investigate whether USP1 and UAF1 complex could inhibit TBK1 protein degradation, a cycloheximide (CHX) chase experiment was performed."

reach
"This agreed with the native gel electrophoresis study suggesting that mutation of Ser313 of USP1 to Ala prevented formation of the USP1 and UAF1 complex, while the USP1 (S313D) mutant retains normal ability to form a complex with UAF1."

reach
"We then investigated the effects of USP1 and UAF1 complex on viral replication using a vesicular stomatitis virus (VSV, another kind of ssRNA virus recognized by RIG-I) infection model."

reach
"We demonstrated using mass spectrometry that treatment of purified wild-type USP1 and UAF1 complex with PP1 effectively removed the phosphate group from Ser313 and Ser67."

reach
"The catalytic activity of the singly six-His-tagged USP1 and UAF1 complex that contained wild-type or mutant USP1 was assessed."

reach
"As shown in XREF_SUPPLEMENTARY, the steady-state kinetic constants of the USP1 (S42A)/UAF1, USP1 (S42D)/UAF1, USP1 (S67A)/UAF1, USP1 (S67D)/UAF1, and USP1 (S313D)/UAF1 complexes are close to that of the wild-type USP1 and UAF1 complex."

reach
"We next examined the potential functions of USP1 and UAF1 complex in vivo using ML323."

reach
"Their k cat and K m values are also similar to those of the corresponding doubly six-His-tagged USP1 and UAF1 complex (XREF_TABLE)."

reach
"Collectively, these data suggest that USP1/UAF1 complex-mediated METTL3 upregulation contributes to SCI occurrence and development via the METTL3/IGF2BP2-YAP1 axis."

reach
"Phosphatase treatment of the USP1 and UAF1 complex inactivates the enzyme complex."

sparser
"Alternatively, FANCI ubiquitination may directly disrupt USP1UAF1 binding to FANCD2."

sparser
"USP1 interacts with the activating protein partner UAF1 (USP1-associated factor 1) and de-ubiquitylate Ubi-PCNA in the absence of DNA damage ( xref )."

reach
"Given that reversible phosphorylation is a common mechanism of regulating the activity of protein complex, we next asked the question whether dephosphorylation of USP1 can lead to inactivation of USP1 and UAF1 complex."

sparser
"Previous studies have shown that the USP1-UAF1 complex binds to TBK1, enhancing IFN-β secretion by removing K48-linked polyubiquitination [ xref ]."

sparser
"One explanation for FANCI's conjugated ubiquitin facilitating a USP1UAF1 resistant complex is that this ubiquitin interacts with FANCD2."

reach
"Recently, PCNA deubiquitylation by the USP1 and UAF1 complex in conjunction with ELG1, which directs the USP1-UAF1 to ubiquitin-PCNA, has emerged as an important regulatory mechanism of damage bypass."

reach
"The DUB activity of USP1 and UAF1 complex treated with PP1 was measured."

reach
"In a control experiment, the USP1 and UAF1 complex treated with phosphatase buffer did not result in an appreciable decrease in the activity of USP1 and UAF1 complex."

reach
"When USP1 and UAF1 complex was treated with PP1, a large decrease in its activity was observed."

reach
"After a 30 min treatment, 4% of the activity for the treated USP1 and UAF1 complex remain."

reach
"Chemical cross linking of the USP1 and UAF1 complex."

reach
"Moreover, mutagenesis of equivalent residues in the WDR48 : USP1 system abrogates both USP1 binding to WDR48 and deubiquitination of a USP1 substrate in cells, suggesting that this interface, distinct[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"26 Hence, the significance of Elg1 's affinity for SUMO is not fully understood.Intriguingly, human ATAD5 appears to respond differently from its yeast counterpart to PCNA modifications : whereas no p[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"In addition, Ge et al. reported that METTL3 is up-regulated in reactive astrocytes following SCI, which is further stabilized by the USP1/UAF1 complex that specifically binds to and subsequently removes the K48-linked ubiquitin [67]."

reach
"The mixture was then assayed for DUB activity using Ub-AMC as a substrate, and the measured specific activity was compared to that of the copurified USP1 and UAF1 complex (treated as 100%)."

reach
"The activity of USP1 after treatment with CDK1 for 2 h showed approximately 40% specific activity relative to the copurified USP1 and UAF1 complex."

sparser
"In this way, USP1:UAF1 drives FANCI:FANCD2 complex toward a uniformly di-ubiquitinated state, that can only be de-ubiquitinated post-repair."

reach
"USP1 Ser313 phosphorylation promotes complex formation between USP1 and UAF1."

sparser
"Previous work on UAF1-USP1 binding showed WD40 domain of UAF1 was sufficient to act as an adapter protein and activate the catalytic DUB activity of USP1, which on its own has very little intrinsic catalytic activity in-vitro xref ."

reach
"We observed a slightly lower k cat for USP1 (S313D) and UAF1 complex (0.084 s -1) compared to the wild-type USP1 and UAF1 complex (0.096 s -1)."

sparser
"USP1 associates with UAF1 (USP1-associated factor 1), resulting in the heterodimeric USP1/UAF1 complex that is required for deubiquitinase activity. xref The USP1/UAF1 complex has been shown to regulate the tolerance of DNA damage induced by DNA cross-linking agents through deubiquitination of PCNA (proliferating cell nuclear antigen) xref and FANCD2 (Fanconi anemia complementation group D2), xref which are proteins that function in translesion synthesis and the Fanconi anemia pathway, respectively."

sparser
"Interestingly, the ubiquitin on FANCI was also protected from USP2, USP7 and USP1UAF1 deubiquitination in the I Ub D2 Ub ‐DNA complex."

reach
"Ser313 phosphorylation not only is required for the tight association between USP1 and UAF1 but also plays a role in relaying the long-range allostery induced by the binding of UAF1."

sparser
"In fact, in the context of the I Ub D2 Ub ‐DNA complex, FANCI deubiquitination was far from complete at 40 min, even at the highest USP1UAF1 concentration used (Fig  xref )."

reach
"The USP1 and UAF1 complex is an important player in two major DNA damage response pathways, i.e., translesion DNA synthesis and the Fanconi anemia pathway."

sparser
"However, isolated I Ub was completely deubiquitinated within 30 min by USP1UAF1 (Fig  xref D)."

reach
"We recently demonstrated that selective USP1 and UAF1 complex inhibitors identified through high-throughput screening sensitized non small cell lung cancer cells to killing by a DNA damaging agent cisplatin."

reach
"Our finding of the pSer313- dependent formation of USP1 and UAF1 complex points to a new approach of inhibiting USP1 activity."

sparser
"Moreover, we observed that FANCD2 was deubiquitinated by USP1UAF1 faster than FANCI in the I Ub D2 Ub complex (Fig  xref )."

sparser
"Hence, if such a complex results from USP1UAF1 deubiquitination, it is likely to retain a certain extent of the strong association with DNA observed with I Ub D2 Ub , potentially via partially stabilizing the closed state."

sparser
"To assemble I Ub D2 Ub ‐DNA complex with ubiquitin mutants on FANCI, we added D2 Ub to each ubiquitin‐mutated I Ub and subjected the resulting I Ub D2 Ub ‐DNA complex to deubiquitination treatment with USP1UAF1 (Fig  xref E)."

reach
"Probing the interaction between UAF1 and the USP1 inserted domain (USP1_ID) by a pull-down experiment."

reach
"To determine whether this domain contributes to the interaction between USP1 and UAF1, a truncated USP1 (amino acids 235-408) (USP1_ID) with a N-terminal GST fusion was generated for pull-down assay with UAF1."

sparser
"We tested each I Ub in DUB assays and found that only I Ub‐I44A was also deubiquitinated to the same extent as I Ub‐WT by USP1UAF1."

reach
"This has already been evidenced by the development of therapeutics aimed at the Usp1 and Uaf-1 complex, which have been shown to reverse cisplatin-resistance in non small lung cancer cells [XREF_BIBR, XREF_BIBR]."

sparser
"Previous work has shown that when DNA is removed from the reaction, di‐monoubiquitinated ID2 is no longer resistant to deubiquitination by USP1UAF1 xref , xref , but it is currently not clear how DNA may protect I Ub D2 Ub from USP1UAF1 deubiquitination."

sparser
"One possibility is that in the absence of DNA ubiquitinated FANCD2 dissociates from ubiquitinated FANCI, and in isolation both proteins are known to be susceptible to USP1UAF1 activity xref ."

sparser
"ATAD5 promotes the distinct deubiquitination process of UAF1USP1, USP7, and USP11 for poly-Ub–PCNA."

sparser
"This could be achieved in a cellular context by extraction of ubiquitinated ID2 complexes by the DVC1‐p97 segregase xref and subsequent deubiquitination by USP1UAF1."

sparser
"Further, FANCI monoubiquitination prevents deubiquitination of ID2 complex by USP1-UAF1 deubiquitinating enzyme in vitro ( Rennie et al., 2020b ; van Twest et al., 2017 )."

sparser
"These results suggest that FANCI monoubiquitination creates a physical barrier that prevents USP1-UAF1 from deubiquitinating ID2 before the DNA damage repair steps are completed."

reach
"Furthermore, the USP1 and UAF1 complex can readily, and rapidly, de-ubiquitinate mono-ubiquitinated FANCD2 in an in vitro de-ubiquitination reaction [XREF_BIBR]."

reach
"The USP1 and UAF1 complex is an important de-ubiquitylase for the RSR that can remove ubiquitin from PCNA and the FANCD2/I complex."

sparser
"Surprisingly, the motif on ATAD5 responsible for the binding to UAF1USP1 was found to coincide with a SIM, and a corresponding SUMO-like domain was identified in the C-terminus of UAF1, which is resp[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"It was shown that efficient mono-Ub–PCNA deubiquitination by UAF1USP1 requires an additional factor, ATPase family AAA+ domain-containing protein 5 (ATAD5), the largest subunit of the PCNA-unloading complex ( xref ), while UAF1USP1 deubiquitinates other substrates such as FANCD2–FANCI on its own ( xref xref – xref )."

sparser
"However, the action of USP1UAF1 within the cell will allow a population of ID2 Ub to revert to an open state via the unstable closed state of deubiquitinated ID2."

sparser
"A cryo-EM reconstruction of USP1-UAF1 in complex with monoubiquitinated FANCI-FANCD2 highlights a highly orchestrated deubiquitination process, with USP1-UAF1 driving conformational changes in the substrate."

sparser
"Overall, our data provide molecular details of USP1-UAF1 regulation and substrate recognition."

sparser
"However, it is currently unknown whether there is a functional interaction between UAF1USP1, USP7, USP11, and ATAD5 that leads to Ub–PCNA deubiquitination."

sparser
"Focusing on autophagy that makes a critical effect on tumor development, researchers have successively identified and screened various specific small-molecule inhibitors of autophagy-related DUBs, including USP1-UAF1 complex inhibitors GW7647 and Pimozide, which can successfully reverse NSCLC cell resistance to chemotherapeutic drugs ( xref )."

sparser
"The ATAD5 N-terminal domain (ATAD5-N) interacts with UAF1USP1 ( xref )."

sparser
"One substrate of the USP1-UAF1 complex is the monoubiquitinated FANCI-FANCD2 heterodimer, which is involved in the repair of DNA interstrand crosslinks via the Fanconi anemia pathway."

sparser
"In addition to UAF1USP1, a role of USP7 in the deubiquitination of Ub–PCNA was reported ( xref , xref )."

sparser
"The crystal structures of USP1-UAF1 reveal plasticity in USP1 and key differences to USP12-UAF1 and USP46-UAF1, two related proteases."

reach
"Overall work by these groups has confirmed the druggability of the Usp1 and Uaf -1 complex and the advantages of complex targeting approach."

sparser
"UAF1USP1 preferentially deubiquitinates DNA-loaded Ub–PCNA over free Ub–PCNA."

reach
"We assayed interaction with hsFANCD2 and found that a USP1UAF1 complex can capture the non-ubiquitinated substrate (Fig S7A)."

sparser
"We find that FANCD2 ubiquitination enhances binding of the ID2 complex to dsDNA, while FANCI ubiquitination protects the complex from deubiquitination by USP1UAF1."

sparser
"For example, regarding FANCD2 ubiquitination, the extent of ID2 chromatin association or foci formation could be assessed in cells where FANCI cannot be ubiquitinated and USP1UAF1 is additionally defective."

sparser
"Our biochemical analyses show that ATAD5 recognizes DNA-loaded Ub–PCNA and recruits UAF1USP1 to facilitate Ub–PCNA deubiquitination."

reach
"However, this study revealed that while an interaction between USP1 and p65 was detected, it was the USP1 binding cofactor UAF1, not USP1 itself, that played an essential role in stabilizing p65 protein (Song et al. 2020)."

sparser
"USP1 forms a complex with UAF1 in the cytoplasm, and then it is translocated into the nucleus."

sparser
"ATAD5-N Facilitates Deubiquitination of Ub–PCNA by UAF1USP1."

reach
"We have shown that UL138 interacts with a UAF1-USP1 complex to sustain STAT1 activation and enhance an early ISG response that restricts viral replication (40)."

sparser
"USP12 and USP46 are two close homologues of USP1 that also bind UAF1. xref − xref Both ML323 and KSQ-4279 resulted in the near-complete inhibition of USP1-UAF1."

sparser
"It has been proposed that ATAD5-N is implicated in Ub–PCNA deubiquitination through interactions with UAF1USP1 ( xref )."

reach
"Here we show evidence that the USP1 and UAF1 complex has a FANCD2independent function in promoting HR repair."

sparser
"DNA-loaded Ub–PCNA was then deubiquitinated by UAF1USP1 ( xref and)."

reach
"UAF1 also binds and activates two other DUBs, USP12 and USP46 (19), and studies that reveal how UAF1 binds and activates USP12 and USP46 suggest that UAF1 will bind to USP1 in an analogous manner (20, 21)."

reach
"We find that UAF1 can bind the minimal USP1 catalytic domain (USP1 ) by size exclusion chromatography (SEC) (Fig 1D) and stimulate DUB activity on a fluorescent ubiquitinated-dipeptide substrate, Ub-KG (Fig 1E)."

reach
"The cleavage rates show that the activity of a USP1UAF1 complex is almost identical to that of USP1UAF1."

reach
"The USP1UAF1 complex can deubiquitinate several monoubiquitinated substrates, including human FANCD2-K561Ub (hsFANCD2-Ub) (6), human FANCI-K523Ub (hsFANCI-Ub) (31), and human PCNA-K164Ub (PCNA-Ub) (25)."

reach
"Cell CycleISSN : 1538-4101 (Print) 1551-4005 (Online) Journal homepage : http://www.tandfonline.com/loi/kccy20The USP1 and UAF1 complex interacts with RAD51AP1 to promote homologous recombination repairScott Cukras, Euiho Lee, Emily Palumbo, Pamela Benavidez, George-Lucian Moldovan & Younghoon KeeTo cite this article : Scott Cukras, Euiho Lee, Emily Palumbo, Pamela Benavidez, GeorgeLucian Moldovan & Younghoon Kee (2016) The USP1 and UAF1 complex interacts with RAD51AP1 to promote homologous recombination repair, Cell Cycle, 15:19, 2636-2646, DOI : 10.1080/15384101.2016.1209613 To link to this article : https://doi.org/10.1080/15384101.2016.1209613View supplementary materialAccepted author version posted online : 27 Jul 2016."

reach
"The USP1/UAF1 complex specifically binds to and subsequently removes K48-linked ubiquitination of the METTL3 protein to maintain its stability after SCI."

sparser
"These findings demonstrate that ATAD5-N modulates Ub–PCNA deubiquitination process directly and is crucial for UAF1USP1 to efficiently deubiquitinate Ub–PCNA."

sparser
"Ub–PCNA Deubiquitination Requires the Stable Interaction of ATAD5-N with UAF1USP1."

sparser
"It is known that interaction between ATAD5 and UAF1USP1 plays a crucial role in controlling the level of Ub–PCNA ( xref )."

sparser
"However, it has not yet been determined how ATAD5 binding to UAF1USP1 affects Ub–PCNA deubiquitination at the molecular level."

sparser
"These findings imply that ATAD5 interacts with UAF1USP1 through UAF1."

sparser
"Our findings revealed an intriguing mechanism of regulating USP1 activity that combines phosphorylation of a key serine residue in USP1 and the specific interaction of USP1 with a WD40-repeat protein UAF1."

reach
"USP1 forms a complex with UAF1 in the cytoplasm, and then the complex translocates to the nucleus utilizing the two nuclear localization signals (NLSs) on USP1."

sparser
"In contrast, ATAD5 (1 to 500) had no impact on cleavage of K63-linked diubiquitin or Ub–AMC by UAF1USP1 ()."

sparser
"However, we were unable to obtain reliable structures of USP1-UAF1 alone with the inhibitor."

reach
"Previous studies have shown that the USP1-UAF1 complex binds to TBK1, enhancing IFN-β secretion by removing K48-linked polyubiquitination [10]."

reach
"A recent report has showed that the di-monoubiquitinated frog ID2 complex (both FANCD2 and FANCI monoubiquitinated), generated in the presence of plasmid DNA, remains resistant to deubiquitination by a GST-tagged USP1UAF1 complex and only the removal of DNA permits efficient DUB activity (32)."

reach
"Furthermore, the USP1UAF1 complex has been shown to play critical roles in homologous recombination (10)."

reach
"The m 6 A level increases in injured spinal cord, and METTL3, which is the core subunit of methyltransferase complex, is upregulated in reactive astrocytes, and further stabilized by the USP1/UAF1 complex after SCI."

sparser
"To date, how USP1 and UAF1 interact and how the formation of the USP1/UAF1 complex is regulated remains largely unknown."

sparser
"To determine the potency of both compounds on USP1, the ability of USP1-UAF1 (0.008 nM) to cleave the ubiquitin-rhodamine substrate (100 nM) was determined in the presence or absence of a half-log, eight-point dilution series of each compound."

reach
"C527 (40) was found to inhibit the USP1/UAF1 complex in vitro in a dose-dependent manner with an IC 50 in the high nanomolar range."

reach
"The USP1 and UAF1 complex promotes homologous recombination and DNA cross-link repair via deubiquitylation of two critical DNA repair substrates : Fanconi anaemia protein (FANCD2) [XREF_BIBR] and proliferating cell nuclear antigen (PCNA) [XREF_BIBR, XREF_BIBR]."

reach
"SJB-2043 (41) is an inhibitor of the natural USP1/UAF1 complex through a series of data."

sparser
"These results suggest that USP1 Ser313 phosphorylation is required for formation of the complex between USP1 and UAF1."

sparser
"Finally, KSQ-4279 was added at 2 equiv of USP1-UAF1."

reach
"Furthermore, USP1, as a prototypical deubiquitylating enzyme, must bind with UAF1 for its catalytic action."

sparser
"To confirm that the interaction between USP1 and UAF1 is mediated by USP1 Ser313 phosphorylation, six-His-tagged USP1 was coexpressed with untagged UAF1 in insect cells."

sparser
"For the wild-type six-His-tagged USP1 coexpressed with untagged UAF1, the eluted fractions contained both USP1 and UAF1, suggesting that the wild-type USP1 can form a stable complex with UAF1 when expressed in insect cells."

reach
"Additionally, we report an X-ray co-crystal structure of TNG-6132 (18) bound in the USP1-UAF1 complex, a result that furthered our understanding of the role played by key elements of the pharmacophore of this chemotype as well as its mechanism of inhibition of USP1."

sparser
"Probing the interaction between UAF1 and the USP1 inserted domain (USP1_ID) by a pull-down experiment."

sparser
"To determine whether this domain contributes to the interaction between USP1 and UAF1, a truncated USP1 (amino acids 235-408) (USP1_ID) with a N-terminal GST fusion was generated for pull-down assay with UAF1."

reach
"Reaching these nuclear substrates first requires nuclear import of the USP1 and UAF1 complex, which is mediated by two nuclear localization signals (NLSs) in USP1 [XREF_BIBR]."

reach
"This view has recently gained experimental support with the identification of two compounds that inhibit the activity of the USP1 and UAF1 complex, and reverse the resistance of NSCLC cells to cisplatin [XREF_BIBR], a DNA damaging drug commonly used in cancer chemotherapy."

reach
"In this study, a high-throughput screening of nearly 10,000 bioactive small-molecules, followed by further validation and characterization of the best candidates, led to the identification of two potent and highly selective reversible inhibitors of the enzymatic activity of the USP1 and UAF1 complex, pimozide and GW7647."

reach
"Besides inhibiting its enzymatic activity, an alternative approach to target USP1 could be interfering with the formation of the USP1 and UAF1 complex, which is not disrupted by pimozide and GW7647 [XREF_BIBR]."

sparser
"The FA pathway is thought to occur in the following steps; (i) collision of replication machinery with an ICL (or other blocking lesion); (ii) activation of FA core complex ubiquitin ligase and monoub[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"It was suggested that phosphorylation of USP1 at Ser313 serves as an interface for the association of USP1 with UAF1."

reach
"Third, and perhaps most importantly, FANCI-monoubiquitination protects the ubiquitin on FANCD2 from being acted upon by the deubiquitinating complex USP1-UAF1 ( Rennie et al., 2020b ; van Twest et al.[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"These biochemical results demonstrate that the interaction between ATAD5 and UAF1 is essential for UAF1USP1 to efficiently deubiquitinate Ub–PCNA."

sparser
"Finally, we show that the substrate must be DNA-bound for ubiquitination to occur, and that, unexpectedly, DNA binding also protects the complex from USP1:UAF1 mediated deubiquitination."

sparser
"The ID2 complex is de-ubiquitinated by USP1-UAF1 and unloaded from the chromatin to allow completion of ICL repair."

sparser
"ATAD5 Bridges Ub–PCNA and UAF1USP1."

sparser
"Under what context does the cell prefer to use FBXL12 instead of the canonical USP1-UAF1 complex to eliminate FANCD2 from the chromatin?"

reach
"Our native gel electrophoresis following the incubation of USP1 and UAF1 with pimozide or GW7647 (up to 10 x IC 50) showed no dissociation of the USP1 and UAF1 complex (XREF_SUPPLEMENTARY)."

reach
"For example, the CDK1-mediated serine phosphorylation of USP1 disrupts the interaction between USP1 and UAF1."

reach
"Using HPV-11, Orav et al. demonstrated that this requires interactions between the E1 protein of HPV-11 and a member of the Fanconi anemia DNA repair pathway, the cellular UAF1-USP1 complex [64]."

sparser
"It has been unclear how ATAD5 links UAF1USP1 and Ub–PCNA for deubiquitination."

reach
"The USP1 and UAF1 complex promotes double-strand break repair through homologous recombination."

sparser
"Finally, given that USP1-UAF1 deubiquitinates not only mono-ubiquitinated FANCD2 but also mono-ubiquitinated PCNA, it would be intriguing to investigate whether FBXL12’s role extends to PCNA degradati[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"Specifically, in human macrophages, the UAF1/USP1 complex binds and stabilizes NLRP3 through deubiquitination, while UAF1 regulates NLRP3 transcription through the stabilization of NF-κb, independent of USP1 function (Song et al. 2020)."

sparser
"To examine whether FANCI deubiquitination activity by USP1:UAF1 is regulated by the 3SQ cluster, we performed in vitro deubiquitination reactions where xFANCI Ub :xFANCD2 Ub was treated with lambda phosphatase after ubiquitination."

reach
"In this regard, a relocation based assay for USP1 and UAF1 binding, which could be used to screen for compounds that disrupt their interaction, has been recently described [XREF_BIBR]."

reach
"This approach would greatly benefit from a knowledge of the three-dimensional structure of the USP1 and UAF1 complex, and from a clarification of the controversy regarding the USP1 region that mediates UAF1 binding [XREF_BIBR, XREF_BIBR]."

sparser
"Therefore, ATAD5-N serves as a molecular platform that bridges UAF1USP1 and Ub–PCNA to facilitate the deubiquitination ()."

sparser
"As shown in xref xref – xref , ATAD5 is an essential factor for the efficient deubiquitination of Ub–PCNA by UAF1USP1."

reach
"Upon formation of a USP1 and UAF1 complex, the catalytic activity of USP1 is greatly stimulated."

sparser
"The UAF1-USP1 association may be necessary for HCMV UL138 to control pSTAT1 for virus genomic latency, according to recent studies, raising the possibility that pSTAT may be an effector in virus infection-induced DNA damage [ xref ]."

sparser
"Intriguingly, UAF1USP1 deubiquitinated DNA-loaded Ub–PCNA more efficiently than free Ub–PCNA which were released from DNA by DNase I treatment ( xref and)."

sparser
"UAF1 interacts with DNA, and DNA enhanced FANCI–FANCD2 deubiquitination by UAF1USP1 ( xref )."

sparser
"The DNA binding-deficient UAF1 3A mutation ( xref ) substantially reduced Ub–PCNA deubiquitination ( xref and), suggesting that DNA is required for the efficient action of UAF1USP1 on Ub–PCNA."

reach
"Furthermore, the ectopic expression of ATAD5 mutant protein (ATAD5 UAF1del) that does not interact with the USP1 and UAF1 complex and results in defective PCNA deubiquitylation reduced chromatin bound PCNA level similar to the ectopic expression of wild-type ATAD5 (XREF_FIG)."

reach
"In this study, we subjected the human USP1 and UAF1 complex to high-throughput screening and identified potent and selective inhibitors against USP1 and UAF1."

reach
"It suggests that ATAD5 UAF1del mutant protein has PCNA unloading activity even though it is defective in recruitment of the USP1 and UAF1 complex to PCNA-Ub to remove ubiquitin from PCNA."

reach
"Once the USP1-UAF1 complex is formed, it translocates into the nucleus where the recruitment of FANCD2 and PCNA substrates using a SUMO-like domain takes a place to regulate DNA damage response [72]."

sparser
"These findings imply that UAF1USP1 deubiquitinates DNA-loaded Ub–PCNA preferentially."

sparser
"FANCD2 monoubiquitination is temporally and spatially controlled at stalled forks by the FA core complex (a RING E3 ligase), USP1:UAF1 (a deubiquitinating enzyme) and ATR kinase ( xref )."

reach
"Given the functional role of USP1 as part of the USP1/UAF1 complex, extensive researches have been conducted to develop inhibitors targeting this complex."

sparser
"ATAD5 (1 to 500) bound to DNA with similar affinity as UAF1USP1."

reach
"In this work we discovered potent, and cell-active inhibitors of the USP1 and UAF1 complex through quantitative high-throughput screening (qHTS)."

reach
"Thus, we propose that the interaction with RAD51AP1 at the post-RAD51 foci formation step is an additional mechanism that regulates the HR repair by the USP1 and UAF1 complex, independently of FANCD2 deubiquitination.Our analysis showed that RAD51AP1 is a mildly unstable protein, and depletion of USP1 or UAF1 greatly decreases the stability."

sparser
"In particular, FANCI itself is a target for monoubiquitination by the FA core complex ( xref ), is a substrate of ATR kinase ( xref ) and contains a USP1:UAF1 binding site necessary for deubiquitination of FANCD2 ( xref )."

reach
"To test this possibility, we purified the UAF1 and USP1 complex under reducing conditions (XREF_FIG)."

reach
"The USP1 and UAF1 complex deubiquitinates the Fanconi anemia protein FANCD2, thereby promoting homologous recombination and DNA cross-link repair."

reach
"Compared to the siRNA approach, the small-molecule inhibitor allowed us to probe the cellular function of USP1 and UAF1 without perturbing the level of USP1 and UAF1 complex in human cells."

reach
"The present identification of selective, noncompetitive inhibitors against the USP1 and UAF1 complex represents the first example of targeting the USP and WD40 repeat protein complex for inhibitor discovery."

reach
"In addition, the nuclear transport signals in one of the subunits may determine the localization of the whole enzymatic complex, as exemplified by the nuclear import of the USP1/UAF1 complex mediated [MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"Additionally, the same study disrupted the NHEJ pathway, via knockout of Ku70 (also known as XRCC6 or X-Ray repair cross complementing 6), in UAF1 -/-/- cells which restored the cellular resistance to camptothecin and the PARP inhibitor suggesting that the USP1 and UAF1 complex promotes HR, at least in part by suppressing NHEJ."

sparser
"UAF1USP1 is a major deubiquitinase for Ub–PCNA."

sparser
"We also predict that a conformational change induced by phosphorylation of the S3Q cluster alters the association of the adjacent SLIM domain of FANCI with USP1:UAF1, as a mechanism for the observed reduction in USP1:UAF1 binding in S3Q phosphomimic proteins."

sparser
"For this purpose, G2-phase synchronized U2OS-ATAD5 AID cells were treated with auxin for 6 h to acutely degrade the ATAD5 protein or treated with ML323, a chemical inhibitor of USP1-UAF1 (hereafter referred to as USP1i) ( xref )."

reach
"Recently, a HTS approach led to the identification of two potent and selective reversible inhibitors of the enzymatic activity of the USP1/UAF1 complex, pimozide and GW7646."
| PMC

reach
"Deubiquitination of FANCD2 is mediated by the USP1/UAF1 complex.18 To gain further mechanistic insight, we decided to test whether ATR phosphorylation controls deubiquitination."

reach
"Once the lesion has been repaired, the ID complex is deubiquitinated by the USP1 and UAF1 complex, which also acts on monoubiquitinated PCNA when associated to the RFC subunit ELG1."

reach
"Binding of hUAF1 stabilizes hUSP1 and promotes interaction with PCNA-Ub and FANCD2-Ub [XREF_BIBR], and the USP1 and UAF1 complex promotes DNA double-strand break repair via homologous recombination [XREF_BIBR]."

reach
"UAF1 and the USP1 and UAF1 complex are also important in viral infection and pathogenesis, particularly in viral genomic integration and replication."

sparser
"Upon completion of DNA repair, the ID2 complex is deubiquitinated by the USP1-UAF1 deubiquitinase complex xref ."

sparser
"The complex of USP1 and UAF1 (USP1-associated factor 1) removes the K48-linked ubiquitin chain after binding to TBK1."

sparser
"To further confirm this, we irradiated cells with UV to induce RAD18-mediated PCNA mono-ubiquitination [ xref – xref ] or treated a ubiquitin-specific protease 1 (USP1) inhibitor (hereafter USP1i, ML323) to block Ub-PCNA deubiquitination by the USP1-USP1-associated factor 1 (UAF1) complex [ xref ]."

sparser
"Purified USP7 and USP11 deubiquitinated Ub–PCNA, but higher enzyme concentrations of USP7 or USP11 were required to fully deubiquitinate Ub–PCNA compared to UAF1USP1 ( xref and)."

sparser
"Intriguingly, USP7 and USP11 exhibited different specificity for free Ub–PCNA compared to UAF1USP1 ( xref and xref )."

reach
"Studies show that the formation of the USP1/UAF1 complex greatly enhances the catalytic activity of USP1, but USP1 alone has a very low level of activity (Cohn et al., 2007; Villamil et al., 2012)."

reach
"A study in which USP1 -/-, UAF1 -/-/-, and USP1 -/- UAF1 -/-/- double-knockout DT40 cells showed hypersensitivity to camptothecin and an inhibitor of poly (ADP-ribose) polymerase (PARP), which suggests that the USP1 and UAF1 complex promotes HR."

reach
"Chen et al. subjected the human USP1 and UAF1 complex to a quantitative high-throughput screening (hHTS) and defined potent and selective inhibitors of USP1 and UAF1."

sparser
"USP7 differed from UAF1USP1 in poly-Ub–PCNA deubiquitination in that it promoted polyubiquitin chain shortening."

sparser
"Moreover, hELG1 (enhanced level of genomic instability 1), an alternative subunit of the RFC clamp loader complex, specifically directs the USP1-UAF1 dimer to PCNA for de-ubiquitination [ xref ]."

sparser
"ML323 is a small molecule inhibitor of USP1-UAF1 and targets two major DNA damage response pathways simultaneously by inhibiting USP1 ( xref ), suggesting its potential against various malignancies in previous researches."

sparser
"It has been shown that USP1 could form a deubiquitinase complex with USP1-associated factor 1 (UAF1) [ xref ], and they participate in the pathogenesis of tumors and enhance the antiviral response together [ xref ]."

sparser
"USP1-UAF1 complex increased TLR3/4 and RIG-I mediated IFN-β production, and the discovered USP1-UAF1 inhibitor ML323 can reduce interferon expression and promote viral replication both in vivo and in vitro ( xref )."

reach
"The similar values of substrate K M (within a factor of 2) for USP1 and the USP1-UAF complex, as well as the closely similar Ub-al Ki values for the two enzyme forms, show that the formation of USP1-U[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"USP1 forms a complex with UAF1 in the cytoplasm, and then it is translocated into the nucleus."

reach
"Protein interaction mapping analysis identified some key residues within RAD51AP1 required for interacting with the USP1 and UAF1 complex."

sparser
"Third, and perhaps most importantly, FANCI-monoubiquitination protects the ubiquitin on FANCD2 from being acted upon by the deubiquitinating complex USP1-UAF1 ( Rennie et al., 2020b ; van Twest et al.[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"The WDR48USP1 complex acts as a regulator during DNA damage, especially in translation synthesis and Fanconi anaemia pathway. xref , xref , xref Recent studies have found that the USP1 complex can downregulate the polyubiquitination of TAK1 and mediate its stability in vitro. xref However, few reports exist on the role and mechanism of WDR48 in HCC."

sparser
"We speculated that the formation of the complex of WDR48 and USP1 affected the ubiquitin degradation of c‐Myc by increasing its deubiquitination activity, which was worthy of further exploration."

sparser
"The addition of UAF1USP1 resulted in the cleavage of the polyubiquitin chain from PCNA ( xref and)."

sparser
"These findings indicate that ATAD5 stimulates UAF1USP1 to cleave the isopeptide bond between the polyubiquitin chain and PCNA."

sparser
"Additionally, ATAD5 facilitates the deubiquitination of PCNA after DNA damage bypass through its interaction with the deubiquitinating enzyme for PCNA, USP1-UAF1 [ xref , xref ]."

reach
"Human ELG1 (which is also called ATAD5) interacts with PCNA, colocalizes with the sliding clamp at stalled replication forks, and also associates with the USP1 and UAF1 complex which removes ubiquitin moieties from PCNA [XREF_BIBR]."

sparser
"The amount of cleaved polyubiquitin chain did not perfectly correlate with poly-Ub–PCNA deubiquitination, possibly due to partial cleavage of polyubiquitin chain in solution by UAF1USP1."

sparser
"It will be important going forward to distinguish roles of UAF1-USP1 in regulating pSTAT1 relative to its role in the DNA damage response in HCMV infection."

reach
"However, other DUBs resulted to be inhibited by this same compound, thus, the effects seen by using it in cells could not be imputed to the specific inhibition of USP1 and UAF1 complex."

sparser
"We showed that UAF1USP1 preferentially deubiquitinates DNA-loaded Ub–PCNA ( xref )."

sparser
"The C-terminal SLD (SUMO-like domain) domain of UAF1 interacts with the SIM (SUMOinteracting motif) of FANCI,23 as well as the SIM of ELG1.28 Thus the SUMO-like delivery system was proposed as a mecha- nism for the USP1-UAF1 complex to recruit its substrates from these studies."

sparser
"We defined an interaction between UL138, a pro-latency HCMV gene, and the host deubiquintase complex, UAF1-USP1."

sparser
"The average length of the polyubiquitin chain cleaved by USP7 is shorter than when poly-Ub–PCNA was incubated with UAF1USP1, but the chain was not completely digested to monoubiquitin."

sparser
"UAF1-USP1 sustains an innate immune response through the phosphorylation and activation of signal transducer and activator of transcription-1 (pSTAT1), as well as regulates the DNA damage response."

reach
"An HTS using USP1 and UAF1 complex identified different inhibitors for this deubiquitinating enzyme complex [XREF_BIBR], and pimozide resulted to be the more efficient chemical against it."

sparser
"To complete the pathway, ubiquitin must be removed by the USP1-UAF1 complex xref – xref ."

sparser
"Once the USP1-UAF1 complex is formed, it translocates into the nucleus where the recruitment of FANCD2 and PCNA substrates using a SUMO-like domain takes a place to regulate DNA damage response [ xref ]."

sparser
"The ATR phosphorylation sites are located in a flexible region of FANCI, adjacent to the FANCI ubiquitination site, and the FA core complex, Ube2T and USP1-UAF1 binding sites ( xref ), referred to here as the “phospho-loop” xref , xref , xref , xref ."

sparser
"First, we showed that recombinant USP1:UAF1 can promote immediate deubiquitination activity against FANCD2 within the ID2 complex."

reach
"Deubiquitination of both FANCD2 and FANCI, which is also critical for ICL repair, is achieved by the USP1 and UAF1 complex."

sparser
"Likewise, phosphorylation may affect how other FA proteins interact with ubiquitinated D2-I. For example, USP1-UAF1 interact less efficiently with phosphorylated D2-I xref ."

sparser
"Here, we show with reconstituted biochemical systems, which we developed, that efficient FANCD2 deubiquitination by the USP1-UAF1 complex is dependent on DNA and DNA binding by UAF1."

sparser
"Second, in order to understand how deubiquitination might be influenced by components of the FA core complex, we added USP1:UAF1 to our in vitro ubiquitination reactions."

sparser
"Efficient ubiquitination was prevented or reversed when USP1:UAF1 was added in the presence of BL100, BL100-CEF, or the full FA core complex (BL100-CEF-AG20) either during or after the reaction had re[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"We hypothesized that UL138 may commandeer the UAF1-USP1 interaction to regulate pSTAT1 for latency."

sparser
"These findings suggest that both UAF1 and USP1 interact with DNA to efficiently deubiquitinate DNA-loaded Ub–PCNA."

sparser
"However, this property of UAF1USP1 can be challenged by the release of Ub–PCNA from DNA."

sparser
"We revealed how the ATAD5 N-terminal domain recognizes DNA-loaded Ub–PCNA and recruits UAF1USP1 to coordinate Ub–PCNA deubiquitination with PCNA unloading."

sparser
"These data altogether suggest that the USP1-UAF1 complex promotes HR repair via multiple mechanisms: through FANCD2 deubiquitination, as well as by interacting with RAD51AP1."

sparser
"However, USP1:UAF1 activity against ubiquitinated FANCI within the ID2 complex was considerably slower than for FANCD2 ( Figure 6 B)."

sparser
"ATAD5 and UAF1USP1 appear to form a stable complex in cells and operate as a functional unit."

sparser
"While the role of UL138 modulating the DDR will be addressed in a separate study, we hypothesized that UL138 may interact with and direct UAF1-USP1 to enhance and sustain pY701-STAT1 during HCMV infection in order to restrict virus replication."

sparser
"ATAD5 is not a substrate for UAF1USP1, but ATAD5-N bridges Ub–PCNA and UAF1USP1."

sparser
"To determine if the UAF1-USP1 interaction was involved in UL138-mediated regulation of pY701-STAT1, we inhibited UAF1-USP1 interactions with the chemical inhibitor, C527 [ xref ] in fibroblasts infected with WT or Δ UL138 STOP viruses."

sparser
"USP1-UAF1 also acts on the mono-ubiquitinated form of the DNA polymerase processivity factor PCNA in translesion DNA synthesis (TLS) xref ."

sparser
"ATAD5-N appears to keep UAF1USP1 in position so that it can efficiently access the isopeptide bond between ubiquitin and PCNA K164, analogous to what FANCI does for the deubiquitination of FANCD2."

sparser
"Furthermore, on closer analysis of extended time points, it was apparent that the ubiquitinated FANCD2 form never completely disappeared upon addition of USP1:UAF1 ( Figure 6 A)."

sparser
"However, it was suggested recently that DNA shields ubiquitinated ID2 from the DUB activity of USP1-UAF1, such that the removal of DNA would lead to a marked enhancement of the efficiency of ID2 deubiquitination xref ."

sparser
"We wish to define mechanistically how DNA may affect the efficiency of FANCD2 deubiquitination by USP1-UAF1, and have developed a reconstituted system of highly purified human proteins for this purpose."

sparser
"UAF1USP1 and ATAD5-N should be positioned on the back face of PCNA."

sparser
"Time course experiments showed that, after the addition of USP1:UAF1, FANCD2 was rapidly deubiquitinated, but only until the ratio of ubiquitinated FANCD2: ubiquitinated FANCI approaches 1:1."

sparser
"Surprisingly, and in direct contradiction with the recently published report xref , we find that the deubiquitination of FANCD2 by USP1-UAF1 requires DNA being present."

sparser
"Furthermore, ATAD5 forms a heterotrimeric complex with UAF1USP1 deubiquitinase, facilitating the deubiquitination of DNA-loaded Ub–PCNA."

sparser
"Interestingly, the FANCD2 DUB activity of USP1-UAF1 that harbors a DNA binding defective UAF1 mutant is fully restored upon the incorporation of wild type but not a DNA binding defective form of RAD51AP1."

sparser
"One of the best characterized human DUBs is ubiquitin-specific protease 1 (USP1), which plays an important role in the cellular response to DNA damage. xref Indeed, the complex of USP1 with USP1-associated factor 1 (UAF1) acts in two important DDR pathways, namely, translesion synthesis (TLS) and interstrand cross-links (ICLs), through the deubiquitination of proliferating cell nuclear antigen (PCNA) and Fanconi anemia complementation group 2D (FANCD2)."

sparser
"In 2001, Chen et al. xref identified pimozide and GW7647 as the first inhibitors of the human USP1-UAF1 complex with low micromolar affinity."

sparser
"These compounds demonstrate the possibility of inhibiting the USP1-UAF1 complex with small molecules and synergize with cisplatin in cisplatin-resistant cells. xref However, both compounds are known to bind several DUBs, deSUMOylase, and cysteine proteases, which has limited their utility as USP1-UAF1 chemical probes."

sparser
"This nanomolar inhibitor of the USP1-UAF1 deubiquitinase complex demonstrated remarkable selectivity and was reported to exert its effect through an allosteric mechanism, binding far from the USP1 active site ( xref ). xref "

sparser
"This indicates that the level of FANCI monoubiquitination inversely determines the level of FANCD2 deubiquitination by USP1:UAF1."

reach
"For optimized catalytic activity, USP1 forms a heterodimeric complex with UAF1 (2), a WD40 repeat-containing protein that also stimulates USP46 and USP12 (3)."

reach
"The USP1UAF1 complex deubiquitinates several substrates involved in DNA damage response, including monoubiquitinated PCNA and FANCD2 (2, 4–6)."

reach
"Although the only BRCA2 mutant cell line in this dataset, PEO1, was not sensitive to USP1 depletion, presented but unpublished work from us and others have shown USP1 inhibitor activity in BRCA2 mutant PDX models.To evaluate USP1 as a potential drug target, we synthesized previously reported inhibitors of the USP1UAF1 complex, ML323 and I-138 (Fig. 1C; refs."

sparser
"ATAD5-N forms a stable complex with UAF1USP1, but its interactions with USP7 and USP11 are weak."

reach
"Consistent with this notion, USP1 was identified as a putative ATM and ATR substrate, and the interaction of USP1 and UAF1 with FANCI is regulated by DNA damage."

sparser
"This finding suggested that USP1-UAF1 is a key regulator of DDR and a potential target for overcoming resistance to the platinum-based anticancer drugs. xref "

sparser
"Further studies suggested that I-138 binds to USP1-UAF1 at an allosteric site, thought to be the same ML323 binding site ( xref )."

sparser
"In addition to UAF1USP1, ATAD5-N interacts with USP7 and USP11 to promote Ub–PCNA deubiquitination by these enzymes ( xref )."

sparser
"Wild type and mutant forms of RAD51AP1, the ID2 complex, and the USP1-UAF1 complex were expressed and purified as described xref , xref , xref , xref (Supplementary Fig.  xref )."

sparser
"To test the DUB activity of USP1-UAF1, we first depleted the ATP content (to prevent further FANCD2 ubiquitination) of the above reaction by apyrase treatment."

reach
"Lastly, FANCD2 is deubiquitinated by USP1 and UAF1 complex to terminate the FA pathway."

reach
"ATR may also regulate FANCD2 ubiquitylation by targeting the FANCD2 deubiquitination complex USP1 and UAF1."

sparser
"To ask whether DNA is needed for deubiquitination, we treated the reaction mixture with benzonase to digest the DNA before adding USP1-UAF1 (Fig.  xref , lanes 5, 7, 9, and 11; Supplementary Fig.  xref , lanes 4–7)."

sparser
"We also employed Ub-VS as activity probe xref to establish that neither benzonase nor DNase I affects the intrinsic DUB attribute of USP1-UAF1 (Supplementary Fig.  xref )."

reach
"For example, USP1, USP12 and USP46 can interact with WD40-repeat protein USP1 associated factor 1 (UAF1), and UAF1 stimulates the catalytic activity of human USP1 through the formation of a tight complex [XREF_BIBR]."

reach
"The USP1 and UAF1 complex is required for regulation of the Fanconi anaemia (FA) DNA repair pathway."

reach
"Like PCNA, FANCD2 ubiquitination is reversed by the USP1 and UAF1 complex (Nijman et al., 2005a; Cohn et al., 2007)."

sparser
"Contrary to this finding, we find that inhibition of USP1 with C527 increased TB40/E genome replication in CD34+ HPCs, suggesting that at least the UAF1-USP1 complex is suppressive to viral genome replication, consistent with the role of UL138 in suppressing replication [ xref ]."

sparser
"In cells, unloading of FANCD2 Ub –FANCI Ub may be instigated by DVC1-p97 [ xref ] but removal of DNA from the FANCD2 Ub –FANCI Ub complex in vitro is sufficient to again permit the activity of USP1:UAF1 on the complex [ xref , xref ] highlighting the critical role that DNA plays in stabilizing the structure of the heterodimer."

sparser
"The USP1-UAF1 complex directly assembles on FANCI and its SIM domain for deubiquitination of both FANCI and FANCD2."

sparser
"In HPV infection, viral helicase, E1, promotes UAF1-USP1 activity for viral genome replication [ xref , xref ]."

sparser
"ATAD5, in cooperation with the UAF1-USP1 complex, is responsible for this de-ubiquitination."

sparser
"The high-risk HPV oncogenes, E6 and E7, delay UAF1-USP1 mediated deubiquitination of the Fanconi anemia (FA) DNA crosslink repair, contributing to genomic instability [ xref ]."

sparser
"Through the cooperation of ATAD5, UAF1USP1, USP7, and USP11, poly-Ub–PCNA can be efficiently recycled to unmodified PCNA and ubiquitin monomers."

sparser
"This co-regulation occurs because of FANCI:UAF1 interaction, because FANCD2-Ub alone cannot be bound and deubiquitinated by USP1:UAF1 if FANCI is absent from the reaction ( Figure S7 )."

reach
"Of note, USP1 and UAF1 interactions demonstrated using this assay could be consistently validated by co-IP in the present study, as well as in our previous report [XREF_BIBR]."

sparser
"To ask if UAF1 DNA-binding is relevant for ID2 deubiquitination, we examined the mutant USP1-UAF1 3A and USP1-UAF1 11A complexes (Supplementary Fig.  xref ) in FANCD2 deubiquitination."

sparser
"UAF1-USP1 has been reported to sustain pY70-STAT1 signaling by deubiquitinating and preventing proteasomal degradation of TBK1 [ xref ]."

sparser
"ATAD5-N facilitates the de-ubiquitination of Ub-PCNA through interaction with the de-ubiquitinase UAF1-USP1 xref ."

sparser
"To test this, the pan-nuclease Benzonase was added to the combined ubiquitination and/or deubiquitination reactions together with USP1:UAF1."

reach
"It is, therefore, necessary to clarify these conflicting results, and to determine to what extent these two amino acid motifs, and the S313 phosphorylation site, contribute to USP1 and UAF1 interaction."

sparser
"Therefore, it is possible that the UAF1-USP1 complex might deubiquitinate STAT1 to promote its interaction with and subsequent activation by IFNAR."

sparser
"Since RAD51AP1, which stably associates with UAF1 xref , also possesses DNA binding activity xref , it was of considerable interest to test whether it would restore to USP1-UAF1 3A and USP1-UAF1 11A mutant complexes the ability to deubiquitinate FANCD2 on DNA."

sparser
"The UAF1-binding SIM domain of FANCI is next to serines 559 and 565, so an alternative explanation is that ATR acts on these sites post-monoubiquitination, to prevent USP1:UAF1 binding."

sparser
"Importantly, the addition of RAD51AP1 to reactions containing either USP1-UAF1 3A or USP1-UAF1 11A led to robust FANCD2 deubiquitination (Fig.  xref , lanes 7 and 9; Supplementary Fig.  xref )."

sparser
"Mechanistic interrogation revealed two new functional relationships between WDR48USP1 and LIG1 or FEN1 (LIG1/FEN1) and between FANCM and SMARCAL1."

reach
"Notably, ID1 is a substrate of USP1 [51], and blockade of USP1 by ML323 (a selective inhibitor of the USP1-UAF1 complex) not only promoted CD8 T-cell infiltration and function but also markedly inhibited the liver metastasis of CRC in an orthotopic liver metastatic syngeneic mouse model [19] (Table 1)."

reach
"Deubiquitination of FANCD2 and FANCI by the constitutively active deubiquitinating complex UAF1 and USP1 keeps the pathway turned off unless required [XREF_BIBR]."

sparser
"Following TLS polymerase disengagement, ID2-Ub is deubiquitylated by USP1-UAF1 [ xref ], thereby allowing engagement of HR repair factors, such as RPA, RAD51, and GEN1 or SLX4-MUS81-EME1-SLX1 resolvases, that drive DSB repair."

reach
"Moreover, H 2 O 2 treatment did not affect the association of Usp1 with UAF1 (XREF_FIG), suggesting that inhibition of USP1 by oxidative stress is not likely caused by ROS mediated disruption of the Usp1 and UAF1 complex."

sparser
"To test the hypothesis that USP1:UAF1 cannot act on ubiquitinated FANCI, we demonstrate that bead-immobilized catalytically inactive USP1(C90S):UAF1 can only bind the non-ubiquitinated form of FANCI ([MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"Efficient deubiquitination of FANCD2 occurred upon the addition of USP1-UAF1 (Fig.  xref )."

sparser
"Complexes where only FANCD2, and not FANCI, is ubiquitinated are bound by USP1:UAF1, as is the ID2 complex when neither protein is monoubiquitinated ( Figure 6 D)."

sparser
"UAF1-USP1 in best understood for its role in regulating nucleation of DNA damage responses [ xref , xref , xref ]."

sparser
"Notably, even though RAD51AP1 DM could associate with UAF1 and the UAF1 3A and UAF1 11A mutants just as avidly as its wild type counterpart (Supplementary Fig.  xref and ref. xref .), it failed to restore FANCD2 DUB activity to USP1-UAF1 3A (Fig.  xref , lanes 7 and 11)."

sparser
"Deubiquitination was more nuanced – USP1:UAF1 could efficiently remove ubiquitin from FANCD2- Ub -FANCI but not FANCD2- Ub -FANCI- Ub ."

reach
"As is well known, active transcription foci are distributed in the nucleus in a punctate pattern; therefore, USP1, WDR48, KDM4A, and AR might form a complex to regulate transcription in PC cells."

sparser
"It is well established that ATAD5-RLC is responsible for unloading PCNA and that ATAD5 interacts with the UAF1-USP1 complex to facilitate the de-ubiquitination of Ub-PCNA."

sparser
"Interestingly, whereas, as expected xref , xref , xref , UAF1 deficient cells showed an elevated level of monoubiquitinated FANCD2, but cells expressing either the UAF1 3A or UAF1 11A mutant harbored mostly deubiquitinated FANCD2 (Fig.  xref ), indicating that the UAF1 mutations have little or no impact on the FANCD2-specific DUB activity of the USP1-UAF1 complex in cells."

sparser
"However if FANCD2- Ub -FANCI- Ub is dissociated from DNA, it then becomes a USP1:UAF1 substrate ( xref )."

sparser
"Our biochemical reconstitution studies (Fig.  xref and Supplementary Fig.  xref ) revealed that, via its DNA binding activity, RAD51AP1 targets the DNA binding defective USP1-UAF1 3A or USP1-UAF1 11A complex to deubiquitinate FANCD2 on DNA."

sparser
"However, UAF1 forms a complex with USP1 to activate its deubiquitinating activity."

sparser
"Consistent with prior studies, UAF1-USP1 co-purified with ATAD5, reinforcing the role of ATAD5 in Ub-PCNA de-ubiquitination xref ."

sparser
"The deubiquitination activity of USP1:UAF1 in the absence of DNA also proceeded at a continuous rate toward zero for FANCD2 and at a slower rate against FANCI ( Figure 7 B)."

sparser
"ML323 is known to disrupt the interaction between USP1 and UAF1 [ xref ], resulting in decreased interaction between USP1 and C/EBPβ."

sparser
"Furthermore, unlike in previous experiments with intact DNA, the di-monoubiquitinated ID2 complex became completely deubiquitinated when DNA was degraded, irrespective of the level of FANCI-Ub at the [MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"UAF1 has been regarded as the substrate recognition subunit of the USP1-UAF1 complex xref , xref ."

reach
"These results provide new insights into the function and regulation of the USP1 and UAF1 complex."

sparser
"WDR48USP1 protect from toxic PCNA ubiquitylation."

sparser
"Several findings have emerged, namely: (1) occurrence of USP1-UAF1 mediated-deubiquitination of FANCD2 on DNA; (2) identification of UAF1 amino acid residues important for DNA binding activity; (3) demonstration that UAF1 DNA binding as being required for FANCD2 deubiquitination within the context of the USP1-UAF1 complex; (4) documentation that DNA binding by RAD51AP1 can substitute for that of UAF1 in FANCD2 deubiquitination within the context of the trimeric USP1-UAF1-RAD51AP1 complex; and (5) revelation that UAF1 and RAD51AP1 provide redundant functions for FANCD2 deubiquitination in cells, thus uncovering a role of RAD51AP1 in the FA DNA damage response in addition to its well-documented DNA repair function."

reach
"USP1 associates with UAF1 (USP1 associated factor 1), resulting in the heterodimeric USP1 and UAF1 complex that is required for deubiquitinase activity."

reach
"10 The USP1 and UAF1 complex has been shown to regulate the tolerance of DNA damage induced by DNA cross linking agents through deubiquitination of PCNA (proliferating cell nuclear antigen) 11 and FANCD2 (Fanconi anemia complementation group D2), 12 which are proteins that function in translesion synthesis and the Fanconi anemia pathway, respectively."

sparser
"We have shown that the HCMV latency protein UL138 forms a unique interaction with UAF1-USP1 to control a typically antiviral response and utilize pY701-STAT1 for suppression of viral replication, at least in part, by stimulating of pro-latency gene expression programming."

sparser
"It will be important to investigate the interplay between the UAF1-USP1 regulation of the DNA damage response and the innate immune responses in HCMV infection."

sparser
"In that study xref , the human FA core complex was used to ubiquitinate the FancI-FancD2 complex from Xenopus laveis , and the modified Xenopus ID2 complex was then tested with human USP1-UAF1."

sparser
"USP1 binds to USP1-associated factor 1 (UAF1), which stimulates the deubiquitinase activity of USP1 [ xref ]."

sparser
"Given that human FANCI and FANCD2 share relatively low sequence identity with their Xenopus counterparts (~65% for FANCI and ~55% for FANCD2), it seems plausible that the requirement of DNA for efficient FANCD2 deubiquitination by human USP1-UAF1 becomes relaxed with the Xenopus protein."

sparser
"These data indicate that the aberrant accumulation of a ubiquitylated substrate of the WDR48USP1 complex might underlie its synthetic lethal interactions with FEN1 / LIG1 ."

sparser
"WDR48USP1 counteracts ubiquitylation by the FANCL and RAD18 ubiquitin ligases xref , xref ."

sparser
"For these experiments, the ubiquitination reaction was inhibited prior to USP1:UAF1 addition by depletion of ATP."

sparser
"For the substrate, mechanistic insight might also come from a co-structure of USP1:UAF1 and FANCI–FANCD2 Ub , which would uncover how the important deubiquitination reaction is controlled."

sparser
"To identify RAD18 substrates that might drive LIG1/FEN1 : WDR48USP1 synthetic lethality, we performed unbiased diGly capture proteomics."

sparser
"Recently, Lim et al. xref , reported that USP1 also possesses a DNA binding activity with specificity for forked DNA (i.e. Y-shaped DNA with a duplex arm) and provided evidence that DNA binding exerts a modest stimulation of the DUB activity of USP1-UAF1."

sparser
"Since ATAD5 facilitates Ub-PCNA de-ubiquitination through the UAF1-USP1 complex, the effect of BAZ1B on de-ubiquitination may be reduced without USP1."

sparser
"H 2 O 2 does not affect the interaction of USP1 with UAF1 ( xref ), suggesting that the inhibition is likely caused by oxidation of USP1 active site."

sparser
"Furthermore, BRCA1-deficient cells expressing a truncated variant of USP1 deficient in DNA binding fail to deliver USP1-UAF1 to replication forks to act on the ubiquitinated form of the proliferating cell nuclear antigen (PCNA) and are, consequently, impaired for the ability to stabilize stressed DNA replication forks xref ."

sparser
"27 In addition to BRCA1, DUBs such as USP38 and the USP1UAF1 complex are involved in HR and NHEJ by deubiquitinating other important proteins in DNA repair pathways."

sparser
"Even after 90 min in the presence of the USP1:UAF1 protease, ID2 is stably maintained in a di-monoubiquitinated form, but it is then processed to non-ubiquitinated form in the time following DNA remov[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"Proteins involved in the formation of large protein complexes were also selected, such as UAF1, which is part of the deubiquitinating enzyme heterodimeric complex USP1-UAF1 [ xref ]; The Cyclin-Dependent Kinase 7 (CDK7), a subunit of the transcription/DNA repair factor TFIIH [ xref ], is both an effector kinase which phosphorylates both RNA Polymerase II and other transcription factors and a Cdk Activating Kinase (CAK) for essential CDKs [ xref ]; VPS26A, which is a subunit of the core ‘Retromer’ complex that mediates endosomal protein sorting and trafficking [ xref ]; and Munc18a and Munc18c, which regulate SNARE-complexes that mediate vesicle fusion [ xref ]."

sparser
"28 The USP1UAF1 inhibitor ML323 enhances the cytotoxicity of cisplatin in osteosarcoma and non-small cell lung cancer (NSCLC) cells ( Table 2 )."

sparser
"The experimental systems that we and others xref , xref have developed will be useful for answering mechanistic questions regarding the intracellular functions of the USP1-UAF1 and for establishing the biological impact of cancer-associated mutations in the USP1-UAF1-RAD51AP1 complex."

sparser
"This configuration suggests that the ATAD5-BAZ1B interaction may hinder the binding of ATAD5 to the UAF1-USP1 complex."

sparser
"The kinetics of FANCI deubiquitination is considerably slower than for FANCD2, but the results demonstrate that the USP1:UAF1 protease can act on both members of di-monoubiquitinated-ID2, but only whe[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

sparser
"We defined an interaction between UL138, a pro-latency HCMV gene, and the host deubiquitinating complex, UAF1-USP1."

reach
"[236] In contrast, USP1 forms a complex with UAF1 and enhances TLR3/4 and RIG-I-induced IRF3 activation and subsequent IFN-beta secretion by binding to TBK1 and removing its K48 linked polyubiquitin chains, thus preventing it from proteasomal degradation."

sparser
"UAF1-USP1 sustains an innate immune response through the phosphorylation and activation of signal transducer and activator of transcription-1 (pSTAT1), as well as regulates the DNA damage response."

sparser
"ATAD5 (1–603) significantly enhanced Ub-PCNA de-ubiquitination by the UAF1-USP1 complex, thereby supporting the role of ATAD5 in this process."

sparser
"Next, we conducted an in vitro Ub-PCNA de-ubiquitination assay with UAF1-USP1 complex, ATAD5 (1–603), and BAZ1B-SMARCA5 complex (Fig.  xref , Supplementary Fig.  xref )."

sparser
"The USP1UAF1 complex specifically targets ubiquitinated FANCD2 for deubiquitination utilizing an N‐terminal module of USP1 xref ."

reach
"[237] ML323, a specific inhibitor of USP1 and UAF1 was found in the same study to enhance viral replication both in vitro and in vivo, thus adding evidence for the positive role of the USP1 and UAF1 complex."

sparser
"Deubiquitination of both FANCD2 and FANCI, which is also critical for ICL repair, is achieved by the USP1-UAF1 complex."

reach
"To identify the influence of the complex USP1 and WDR48 during the CSC tumorigenesis process, and whether this complex is a possible therapeutic target."

sparser
"These data pinpointed PCNA as a potential substrate of RAD18 that drives cell death in LIG1/FEN1-deficient cells if hyper-ubiquitylation cannot be reversed by WDR48-USP1."

reach
"A literature search regarding the role of the complex USP1 and WDR48 in inhibiting differentiation and increasing proliferation of CSC was performed, and possible selective molecule inhibitors of these deubiquitinase proteins were investigated."

sparser
"It will be important going forward to distinguish roles of UAF1-USP1 in regulating pSTAT1 relative to its role in the DNA damage response in HCMV infection."

sparser
"However, the resulting di-mono-ubiquitinated complex is highly resistant to USP1-UAF1 deubiquitination."

reach
"There is evidence that USP1 and WDR48 complex promotes stem cell conservation and regulation of DNA damage repair."

sparser
"Interestingly, the addition of BAZ1B-SMARCA5 complex inhibited Ub-PCNA de-ubiquitination by ATAD5 (1–603) and the UAF1-USP1 complex (Fig.  xref )."

sparser
"Collectively, these findings suggest that the interaction between ATAD5 and BAZ1B-SMARCA5 complex interferes with the ability of ATAD5 to facilitate Ub-PCNA de-ubiquitination by the UAF1-USP1 complex."

sparser
"We show that a viral latency protein, UL138, interacts with the deubiquitinating complex, UAF1-USP1 to enhance and sustain phosphorylation and activation of signal transducer and activator of transcription 1 (STAT1)."

reach
"In a follow-up study, Liang et al. screened a larger library with a quantitative high throughput assay to identify additional inhibitors of the USP1/UAF1 complex."

sparser
"We hypothesized that UL138 may commandeer the UAF1-USP1 interaction to regulate pSTAT1 for latency."

sparser
"The inhibition of the deubiquitinase complex USP1-UAF1 or the expression of constitutive fusion of ubiquitin to PCNA provides the missing clue required for DNA polymerase η recruitment and thereafter the introduction of A/T base pair (bp) mutations during the process of IgV gene diversification."

reach
"In this study, we define an interaction between UL138 and USP1-associated factor 1 (UAF1) and ubiquitin specific peptidase 1 (USP1)."

reach
"These results validate the interaction between UL138, UAF1, and USP1."

reach
"The scaffold protein UAF1 forms a complex with USP1, USP12, and USP46 that is essential for their activity."

sparser
"ATAD5, in concert with the UAF1-USP1 complex, de-ubiquitinates Ub-PCNA at the final stage of DNA repair synthesis."

reach
"We find that inhibition of USP1 with C527 increased TB40/E genome replication in CD34+ HPCs, suggesting that at least the UAF1-USP1 complex is suppressive to viral genome replication, consistent with the role of UL138 in suppressing replication [17]."

reach
"Therefore, it is possible that the UAF1-USP1 complex might deubiquitinate STAT1 to promote its recruitment to and subsequent activation by IFNAR."

sparser
"Although this can occur when D2 Ub is in isolation or in complex with FANCI, di‐monoubiquitinated ID2 complexes (I Ub D2 Ub ) bound to DNA remain largely resistant to USP1UAF1 deubiquitination xref , xref ."

reach
"The USP1 and UAF1 complex directly assembles on FANCI and its SIM domain for deubiquitination of both FANCI and FANCD2."

sparser
"Fourth, RAD18 depletion blunted PCNA ubiquitylation and reversed its loss in LIG1-depleted cells (Fig.  xref ), suggesting that PCNA mono-ubiquitylation ultimately leads to its poly-ubiquitylation and degradation if not counteracted by WDR48USP1."

sparser
"The proper association of UAF1-USP1 complex to ATAD5 is crucial for the de-ubiquitination of Ub-PCNA; any alterations in this interaction by BAZ1B may inhibit the de-ubiquitination."

sparser
"RAD18 depletion reversed this shortening, confirming that WDR48USP1 is required to limit post-replicative ssDNA gap accumulation in LIG1-depleted cells."

sparser
"USP1UAF1, at low (25 nM) concentrations, deubiquitinated both D2 Ub and ID2 Ub at similar rates."

reach
"These data altogether suggest that the USP1 and UAF1 complex promotes HR repair via multiple mechanisms : through FANCD2 deubiquitination, as well as by interacting with RAD51AP1.ARTICLE HISTORY Received 15 April 2016 Revised 14 June 2016 Accepted 29 June 2016KEYWORDS deubiquitinating enzyme; homologous recombination repair; RAD51AP1; UAF1; USP1IntroductionDNA double strand breaks (DSBs) are highly lethal LESIONS that must be repaired before cell division ensues."

sparser
"USP1 forms a complex with USP1-associated factor 1 (UAF1), which reduces K48-linked ubiquitination of TBK1 and stabilizes it, resulting in the downstream activation of IRF3 and promotion of IFN-β secretion."

sparser
"The USP1-UAF1 inhibitor ML323 suppresses the expression of IFN-β and enhances virus replication in vitro and in vivo ."

sparser
"Overall, our data support a model in which WDR48USP1 counteract the hyperactive RAD18-mediated ubiquitylation of PCNA in FEN1/LIG1-depleted cells (Fig.  xref )."

sparser
"While the role of UL138 modulating the DDR will be addressed in a separate study, we hypothesized that UL138 may interact with and direct UAF1-USP1 to enhance and sustain pSTAT1 during HCMV infection in order to restrict virus replication."

reach
"D. Model : The USP1 and UAF1 complex promotes theHR repair in multiple mechanisms."

sparser
"To determine if the UAF1-USP1 interaction was involved in UL138-mediated regulation of pSTAT1, we inhibited UAF1-USP1 interactions with the chemical inhibitor, C527 [ xref ] at a low concentration of 0.88 μ M which specifically inhibits only UAF1-USP1 deubiquitinating activity [ xref ], in fibroblasts infected with WT or Δ UL138 STOP viruses ( xref )."

sparser
"Without balancing the WDR48USP1 activity, this accumulates to hyper-ubiquitylation and PCNA degradation."

sparser
"This function of WDR48USP1 in preventing PCNA degradation may also be important for the subset of Okazaki fragments that evade repair by FEN1 and LIG1 (ref.  xref )."

sparser
"UAF1 and USP1 form a deubiquitinase complex, which participates in a variety of biological processes, such as regulation of DNA repair processes and tumor pathogenesis."

sparser
"We have shown that UL138 interacts with a UAF1-USP1 complex to sustain STAT1 activation and enhance an early ISG response that restricts viral replication ( xref )."

sparser
"However, the I Ub D2 Ub substrate remained almost completely resistant to USP1UAF1 activity (Fig  xref A; Left )."

sparser
"ID2 Ub ‐DNA, on the other hand, can be efficiently deubiquitinated by USP1UAF1, unless further ubiquitination on FANCI occurs."

sparser
"At higher concentrations of USP1UAF1 (100 nM), we found that FANCD2 can be deubiquitinated, albeit at slower rate than D2 Ub and ID2 Ub , which are rapidly deubiquitinated in under 10 min (Fig  xref A; Right )."

sparser
"These data suggested that the interaction of UAF1 with USP1 is necessary for the stability of USP1 in vivo."

sparser
"Mechanistically, we focused on two of the strongest synthetic lethal gene pairs identified in our screen: FEN1 / LIG1 : WDR48USP1 and FANCM : SMARCAL1 ."

sparser
"Our study implies a crucial role for WDR48USP1 in preventing DNA gaps from inducing PCNA hyper-ubiquitylation and subsequent cell death during unperturbed DNA replication."

sparser
"Removal of the two ubiquitins, through isopeptide cleavage by the USP1UAF1 complex, is also required for ICL repair and maintenance of genomic stability (Oestergaard et al ,  xref ; Kim et al ,  xref ; Murai et al ,  xref )."

sparser
"However, further ID2 ubiquitination on FANCI results in enhanced protection of FANCD2's ubiquitin from USP1UAF1 action (Rennie et al ,  xref )."

sparser
"On the other hand, UAF1 also binds USP1 to selectively remove the K48-linked polyubiquitylation of NLRP3, contributing to subsequent assembly and activation of NLRP3 inflammasome ( xref )."

sparser
"These data suggest that access to the ubiquitin on FANCD2 for USP1UAF1 is reduced when in complex with ubiquitinated FANCI and DNA."

sparser
"With the present data, we now demonstrate that the N-terminal fragment possesses enzymatic activity in complex with UAF1 and the C-terminal fragment of USP1."

reach
"The USP1/UAF1 complex improves motor function in mice by stabilizing METTL3 after SCI [40]."

reach
"These data indicate that the aberrant accumulation of a ubiquitylated substrate of the WDR48USP1 complex might underlie its synthetic lethal interactions with FEN1/LIG1.WDR48USP1 counteracts ubiquitylation by the FANCL and RAD18 ubiquitin ligases ."

sparser
"Therefore, we speculated that the activation of the USP1 enzyme might result from a direct binding of UAF1 to newly synthesized USP1 in the in vitro reaction."

sparser
"Since USP1UAF1 is a specific deubiquitinase (DUB) for FANCD2 xref , xref , we wanted to assess whether alternative DUBs, that lack FANCD2 specificity (i.e. target structurally diverse substrates), are also able to access the ubiquitin on FANCD2 when this is in complex with I or I Ub ."

sparser
"One such USP1UAF1 inhibitor is currently in Phase I clinical trials, for treatment of advanced solid tumours (KSQ Therapeutics Inc,  xref )."

sparser
"To assess the difference between FANCD2 and FANCI deubiquitination, we assayed I Ub D2 Ub complex deubiquitination by USP1UAF1 in a time‐course."

sparser
"We speculate that the binding of UAF1 to USP1 induces a conformational change of the enzyme resulting in a robust increase in enzymatic activity."

reach
"The USP1 and UAF1 complex is emerging as a novel target in cancer therapy, but several aspects of its function and regulation remain to be further clarified."

sparser
"Positive regulators like neuregulin receptor degradation protein 1 (Nrdp1), RING finger protein 128 (RNF128), tyrosine kinase Src, Raf kinase inhibitory protein (RKIP), Glycogen Synthase Kinase-3 Beta (GSK3β), DNA methyltransferases 3A (Dnmt3a), histone deacetylase 3 (HDAC3), and tripartite motif protein 9 (TRIM9) could enhance the autophosphorylation or activation, and USP1-UAF1 complex could stabilizes TBK1 to enhance its function."

reach
"The USP1 and UAF1 complex deubiquitylates FANCD2 and FANCI."

reach
"Importantly, several inhibitors of the USP1 and UAF1 complex have been recently shown to act synergistically with cisplatin in cancer derived cell lines [XREF_BIBR, XREF_BIBR, XREF_BIBR], suggesting that this complex may represent a valid therapeutic target in cancer."

reach
"Under what context does the cell prefer to use FBXL12 instead of the canonical USP1-UAF1 complex to eliminate FANCD2 from the chromatin?"

sparser
"In particular, we have discovered that the USP1:UAF1 complex has a reciprocal substrate preference to the FA core complex ( Figure 7 D)."

reach
"Our results indicate that the S313 phosphorylation site is not critical for USP1 binding to UAF1 or for PCNA deubiquitination in a cellular environment."

reach
"UAF1 binds and stabilizes a non phosphorylatable S313A mutant version of USP1 in cell based assays."

reach
"Since only in vitro evidence for S313 phosphorylation regulated USP1 and UAF1 interaction has been provided, we decided to evaluate the role that this phosphorylation site may have in regulating USP1 binding to UAF1 in a cellular context."

reach
"The USP1 and UAF1 complex is emerging as a novel target for cancer treatment, and inhibitors of USP1 catalytic activity have been reported to reverse the resistance to platinum based chemotherapeutic drugs in NSCLC cells [XREF_BIBR, XREF_BIBR] and to inhibit the growth of leukemic cell lines [XREF_BIBR]."

sparser
"H. Model: UAF1 mediates the interaction between USP1 and RAD51AP1. of USP12 and USP46, 2 DUBs that also associate with UAF1,26 did not significantly affect the RAD51AP1 stability (Fig. S5), suggesting that the effect is specific to the USP1-UAF1 complex."

reach
"We have previously described a cellular relocation assay to evaluate USP1 and UAF1 interaction [XREF_BIBR]."

reach
"The finding that the non phosphorylatable S313A mutant version of USP1 binds and is stabilized by UAF1 in our cell based assays indicates that phosphorylation at S313 is not critical for USP1 and UAF1 complex formation in a cellular context."

reach
"It is important to note that none of these mutations disrupted USP1 and UAF1 interaction, according to the results of a nuclear relocation assay (Additional file 5B)."

sparser
"We find that inhibition of USP1 with C527 increased TB40/E genome replication in CD34+ HPCs, suggesting that at least the UAF1-USP1 complex is suppressive to viral genome replication, consistent with the role of UL138 in suppressing replication [ xref ]."

sparser
"In HPV infection, viral helicase, E1, promotes UAF1-USP1 activity for viral genome replication [ xref , xref ] and the high-risk HPV oncogenes, E6 and E7, delay UAF1-USP1 mediated deubiquitination of the Fanconi anemia (FA) DNA crosslink repair, contributing to genomic instability [ xref ]."

sparser
"Our data point to two roles for the USP1:UAF1 complex in the FA pathway: (1) fast removal of ubiquitin from ubiquitinated-FANCD2 when FANCI has not been also ubiquitinated and (2) removal of ubiquitin[MISSING/INVALID CREDENTIALS: limited to 200 char for Elsevier]"

reach
"Building upon our identification of UAF1 and USP1 interaction, we further analyzed their expression dynamics."

reach
"Subsequent immunoprecipitation assays using testicular extracts from PD3, PD6, and PD8 mice revealed that the interaction between UAF1 and USP1 begins before PD8 and becomes more pronounced by this stage."

reach
"Co-IP in the ovary confirmed the interaction between UAF1 and USP1 in WT ovaries (Figure S4D)."

reach
"Previous reports show that USP1 and UAF1 form a complex and function together, and UAF1 significantly enhances USP1 activity by stabilizing its protein levels and mediating its access to substrates.14 20 USP1 and UAF1 form a complex in the cytoplasm that is subsequently translocated to the nucleus through active nuclear import mediated by the two USP1 NLSs."

reach
"USP1 is stabilized and activated by UAF1 in vivo and in vitro.14 Previous studies have shown that USP1 and UAF1 form a complex in the cytoplasm that is subsequently translocated to the nucleus through active nuclear import mediated by the two USP1 NLSs.30 Conditional knockout of Uaf1 in the testes results in reduced activity and impaired nuclear-cytoplasmic translocation in round spermatids, thus compromising their function."

reach
"We observed an interaction and colocalization between UAF1 and USP1 in the testes."

reach
"The USP1 and UAF1 complex is also recruited to mono-ubiquitylated PCNA via an interaction between UAF1 and the protein ELG1, a protein involved in replication complexes and in loading PCNA onto DNA for efficient replication, and independently identified as a factor required to suppress genomic instability [XREF_BIBR, XREF_BIBR]."

sparser
"Previous work has shown that DNA is required for efficient protection of both FANCI and FANCD2 against USP1UAF1 mediated deubiquitination (van Twest et al ,  xref ; Arkinson et al ,  xref )."

reach
"The USP1/UAF1 complex is a key regulator of the TLS and FA pathways, both of which play key roles in DNA repair processes (21–23)."

reach
"The deubiquitinating enzyme heterodimeric complex USP1 and UAF1 regulates the Fanconi anemia (FA) DNA repair pathway."

sparser
"These data suggest that USP1UAF1 may be capable of modulating the ID2 Ub ‐DNA complex in order to gain access to FANCD2's conjugated ubiquitin."

sparser
"Alternatively, the presence of simply dsDNA, or FANCD2 (irrespective of ubiquitination status) and dsDNA, may hinder USP1UAF1 from targeting FANCI's ubiquitin."

sparser
"To test those possibilities, we performed USP1UAF1 deubiquitination assays with either isolated FANCI/FANCD2 proteins (I Ub or D2 Ub ) or differentially ubiquitinated ID2 complexes (I Ub D2, I Ub D2 Ub or ID2 Ub ), in the presence or absence of dsDNA (Fig  xref )."

sparser
"Here we show evidence that the USP1-UAF1 complex has a FANCD2independent function in promoting HR repair."

sparser
"This suggests that both DNA and FANCD2 are required for maximal protection of I Ub against USP1UAF1 deubiquitination."

sparser
"Therefore, it is possible that the UAF1-USP1 complex might deubiquitinate STAT1 to promote its recruitment to and subsequent activation by IFNAR."

sparser
"Moreover, the presence of either D2 or D2 Ub provides I Ub ‐deubiquitination protection from USP1∆N‐UAF1 (Fig  xref ), similarly to what observed with wild‐type USP1UAF1 complex (Fig  xref )."

sparser
"Whereas the FANCI interaction with the ubiquitin conjugated to FANCD2 has no protective role against USP1UAF1 mediated FANCD2 deubiquitination, the interaction of FANCI's ubiquitin with FANCD2 (in I Ub D2‐DNA and I Ub D2 Ub ‐DNA complexes) efficiently protects against FANCI deubiquitination."

sparser
"Then, we evaluated antitumor effect of USP1 inhibitor I-138 in vivo , which is a novel USP1USP1-associated factor 1 inhibitor, structurally related to ML323 ( xref )."

sparser
"To test whether this extended interface is involved in protecting FANCI from deubiquitination, we mutated H209, V243 and P244 of FANCD2 to alanine residues, and assessed I Ub ‐DNA deubiquitination by USP1UAF1 in the presence of wild‐type (D2 WT ) or H209A/V243A/P244A mutated (D2 H209A,VP243AA ) FANCD2."

sparser
"For these assays, FANCI was ubiquitinated with DyLight‐680 labelled ubiquitin, mixed with DNA and treated with USP1UAF1, in the presence of increasing concentrations of FANCD2."

sparser
"These results demonstrate that the extended interface of FANCD2 with the ubiquitin of FANCI is involved in protecting I Ub from extensive USP1UAF1 deubiquitination."

sparser
"Nevertheless, our work here shows that this ability of USP1UAF1 is compromised when FANCI is also ubiquitinated."

reach
"Interactions between USP1 and WDR 48 were confirmed using co-immunoprecipitation."

reach
"Among these ten genes, the high expression of seven genes was significantly related to poor OS (BPTF, SETD2, SMARCC1, UBXN7, SMC3, PBRM1 and SF3B1) (Figure 9C), while the other three genes showed no significance (ATRX, SIN3A and USP34) (Figure S3).3.8 Validation of the interaction between USP1 and WDR48 in HCC cell lines."

reach
"In addition, we confirmed the interaction between USP1 and WDR48 in HCC cell lines using co‐immunoprecipitation, and the results showed that USP1 interacted with WDR48 in MHCC97H and SK‐Hep‐1 cells (Figure 10H).3.9 SiRNA-USP1 transfection or ML-323 treatment decreased the proliferation of HCC cells."

reach
"142 Liang et al. 143 investigated the binding effects of inhibitor ML323 on the human deubiquitinase complex USP1 and UAF1 using HDX MS and showed four moderately protected and two strongly protected peptides located outside the USP1 catalytic site, supporting the idea that ML323 is an allosteric inhibitor of USP1-UAF1."

reach
"Our results do not definitely exclude the possibility that S313 phosphorylation may modulate the activity of the USP1 and UAF1 complex."

reach
"If USP1 (235-408) S313D interacts weakly with UAF1, a negative result in the nuclear relocation assay may be due to competition by endogenous partner (s)."

sparser
"However, USP1UAF1 is apparently able to circumvent any protection formed from a locked ID2 Ub ‐DNA complex."

sparser
"UAF1-USP1 in best understood for its role in regulating nucleation of DNA damage responses [ xref , xref , xref ]."

reach
"Yu et al reported that the USP1WDR48 complex stabilized TANK‐binding kinase 1 by moving its K48‐linked polyubiquitination, and this process could be attenuated using the inhibitor ML‐323."

reach
"Hence, we hypothesized that the USP1WDR48 complex played critical roles in HCC by stabilizing the activity of their substrates."

reach
"This finding suggested that the USP1WDR48 complex played a tumour‐promoting role in HCC by stabilizing and deubiquitinating these hub genes."

reach
"64 , 65 These agents inhibit the activity of USP1WDR48 complex in a non‐competitive manner."

sparser
"Our reactions also support a dominance of USP1:UAF1 deubiquitination reaction over the FA core complex monoubiquitination reaction ( Figures 6 and 7 )."

sparser
"The effect of FANCI's ubiquitination may be on rendering the doubly ubiquitinated ID2 complex unamenable to USP1UAF1 modulation, for example by stabilizing an ID2 conformational change induced by FANCD2 ubiquitination or inducing further minor conformational changes that stabilize the complex."

sparser
"This points to either an additional factor or a particular DNA substrate that would continue to drive and/or protect ubiquitination activity in the presence of USP1:UAF1."

reach
"These three clones showed similar sensitivities to chemical cross linking agents, to a topoisomerase poison, camptothecin, and to an inhibitor of poly (ADP-ribose) polymerase (PARP), indicating that the USP1 and UAF1 complex is a regulator of the cellular response to DNA damage."

reach
"The hypersensitivity to both camptothecin and a PARP inhibitor suggests that the USP1 and UAF1 complex promotes homologous recombination (HR)-mediated double-strand break (DSB) repair."

reach
"To gain insight into the mechanism of the USP1 and UAF1 complex in HR, we inactivated the nonhomologous end joining (NHEJ) pathway in UAF1 deficient cells."

sparser
"The clamping on DNA of I Ub D2 and I Ub D2 Ub complexes ensures that maximum protection against USP1UAF1 activity is achieved for both conjugated ubiquitins, and therefore ubiquitinated ID2 cannot easily revert to a non‐ubiquitinated state."

sparser
"In essence, FANCI ubiquitination, via maintaining FANCD2 ubiquitination, commits the ID2 Ub complex for FA repair, since without FANCI ubiquitination such complex would be rapidly transformed to a non‐ubiquitinated ID2 complex, through the action of USP1UAF1."

reach
"The USP1/UAF1 complex also modulates PCNA monoubiquitination via UAF1 and ELG1 interaction [234]."

reach
"USP1 inhibitor screening showed ML323 as a potential USP1/UAF1 complex inhibitor that potentiates cisplatin cytotoxicity along with increased PCNA and FANCD2 monoubiquitination [237,238]."

sparser
"Our data support a model in which USP1 and UAF1 form a complex in the cytoplasm that subsequently translocates to the nucleus through import mediated by USP1 NLSs."

reach
"Mutation of Ser313 to Ala on USP1 abolished the complex formation between USP1 and UAF1, thus prevented the activation of USP1 by UAF1."

sparser
"Our findings indicate that these NLSs mediate the nuclear localization of the USP1/UAF1 complex, supporting a model in which USP1 and UAF1 form a complex in the cytoplasm that subsequently relocates to the nucleus through active nuclear import mediated by USP1 NLSs."

sparser
"Our results are consistent with a model in which USP1 and UAF1 form a complex in the cytoplasm that is subsequently translocated to the nucleus through active nuclear import mediated by the two USP1 NLSs ( xref )."

reach
"As RAD51AP1 amplification is associated with multiple types of cancers,18,43-45 therapeutic targeting of the USP1 and UAF1 complex may be beneficial in suppressing these tumors.Materials and methodsCell lines, plasmids and chemical reagentsHeLa, 293T, and U2OS cells were cultured in Dulbecco 's Modified Eagle 's Medium (DMEM), and HEY cells (a gift from Dr. Meera Nanjundan) were grown in RPMI media."

sparser
"Lastly, FANCD2 deubiquitination, which also involves a conformational transition step, whereby ID2 Ub ‐DNA complex opens‐up a bit upon USP1UAF1 binding (Rennie et al ,  xref ), also progresses much faster when the initial closed ID2 Ub ‐DNA state is compromised into a more open‐state via the FANCI R1285A mutation (Wang et al ,  xref )."

reach
"Our results indicate that the USP1 and UAF1 complex promotes HR, at least in part by suppressing NHEJ."

sparser
"27 Therefore we attempted to test whether the potential ubiquitination might regulate the UAF1 interaction, however, the K139 mutation to Arg (K139R) did not affect the interactions with UAF1 and USP1 (Fig. 4E)."

sparser
"In fact, our deubiquitination assays indicate that I Ub sensitivity to USP1UAF1 action is conferred only by the absence of DNA, while enhanced protection is achieved when both FANCD2 and DNA are present (Fig  xref )."

reach
"For example, USP1 interaction with UAF1 is necessary for DUB activity in vitro and deubiquitination of cellular substrates PCNA and FANCD2."

reach
"Herein, we review the regulation, catalysis, and inhibition of the USP1 and UAF1 complex."

sparser
"To determine the effect of inhibiting the UAF1-USP1 interaction on HPV11 RIs, 2D N/N AGE analysis of RIs generated from the WT genome in the presence of 20 µM ML323 was performed ( xref )."

sparser
"The effect of inhibiting the UAF1-USP1 interaction was very similar to that of inhibiting the E1-UAF1 interaction, confirming that the loss of USP1 activity is essential to the replication defect caused by abrogated UAF1 binding."

reach
"The biological functions of USP1 and UAF1 complex."