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Published on: February 3, 2022
Single-Stranded DNA Gap Accumulation Is a Functional Biomarker for USP1 Inhibitor Sensitivity
Alexandre A da Costa1,2, Ozge Somuncu1,2, Ramya Ravindranathan1,2
1Center for DNA Damage and Repair, Dana-Farber Cancer Institute, Boston, Massachusetts.
Abstract:
Recent studies suggest that PARP and POLQ inhibitors confer synthetic lethality in BRCA1-deficient tumors by accumulation of single-stranded DNA (ssDNA) gaps at replication forks. Loss of USP1, a deubiquitinating enzyme, is also synthetically lethal with BRCA1 deficiency, and USP1 inhibitors are now undergoing clinical development for these cancers. Herein, we show that USP1 inhibitors also promote the accumulation of ssDNA gaps during replication in BRCA1-deficient cells, and this phenotype correlates with drug sensitivity. USP1 inhibition increased monoubiquitinated proliferating cell nuclear antigen at replication forks, mediated by the ubiquitin ligase RAD18, and knockdown of RAD18 caused USP1 inhibitor resistance and suppression of ssDNA gaps. USP1 inhibition overcame PARP inhibitor resistance in a BRCA1-mutated xenograft model and induced ssDNA gaps. Furthermore, USP1 inhibition was synergistic with PARP and POLQ inhibition in BRCA1-mutant cells, with enhanced ssDNA gap accumulation. Finally, in patient-derived ovarian tumor organoids, sensitivity to USP1 inhibition alone or in combination correlated with the accumulation of ssDNA gaps. Assessment of ssDNA gaps in ovarian tumor organoids represents a rapid approach for predicting response to USP1 inhibition in ongoing clinical trials. Significance: USP1 inhibitors kill BRCA1-deficient cells and cause ssDNA gap accumulation, supporting the potential of using ssDNA gap detection as a functional biomarker for clinical trials on USP1 inhibitors.
Insights
USP1 inhibitors kill BRCA1-deficient cancer cells by causing DNA gaps, similar to PARP inhibitors. Detecting these single-stranded DNA (ssDNA) gaps may predict treatment response in clinical trials.
Area of Science:
- Genetics
- Molecular Biology
- Oncology
Background:
- BRCA1 deficiency in tumors creates synthetic lethality vulnerabilities.
- PARP and POLQ inhibitors exploit these vulnerabilities by inducing DNA damage.
- USP1 inhibitors are emerging as a therapeutic strategy for BRCA1-deficient cancers.
Purpose of the Study:
- To investigate the mechanism of USP1 inhibitors in BRCA1-deficient cells.
- To determine if USP1 inhibition induces single-stranded DNA (ssDNA) gaps.
- To evaluate USP1 inhibitors as a predictive biomarker for treatment response.
Main Methods:
- Treatment of BRCA1-deficient cells and xenograft models with USP1 inhibitors.
- Assessment of ssDNA gap accumulation and proliferating cell nuclear antigen (PCNA) ubiquitination.
- Evaluation of drug resistance and synergistic effects with PARP and POLQ inhibitors.
- Analysis of patient-derived ovarian tumor organoids for sensitivity and ssDNA gap correlation.
Main Results:
- USP1 inhibition induced ssDNA gaps in BRCA1-deficient cells, correlating with drug sensitivity.
- USP1 inhibition increased monoubiquitinated PCNA, mediated by RAD18.
- RAD18 knockdown conferred resistance to USP1 inhibitors and reduced ssDNA gaps.
- USP1 inhibition overcame PARP inhibitor resistance and showed synergy with PARP and POLQ inhibitors.
- Sensitivity in ovarian tumor organoids correlated with ssDNA gap accumulation.
Conclusions:
- USP1 inhibitors induce synthetic lethality in BRCA1-deficient cells via ssDNA gap accumulation.
- USP1 inhibition is a promising therapeutic strategy, potentially overcoming resistance to other inhibitors.
- ssDNA gap assessment serves as a predictive biomarker for USP1 inhibitor efficacy in clinical trials.
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