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Record W4401345763 · doi:10.1186/s12943-024-02048-1

BRCA1 secondary splice-site mutations drive exon-skipping and PARP inhibitor resistance

2024· letter· en· W4401345763 on OpenAlexaff
Ksenija Nešić, John J. Krais, Yifan Wang, Cassandra J. Vandenberg, Pooja Patel, Kathy Q. Cai, Tanya T. Kwan, Elizabeth Lieschke, Gwo‐Yaw Ho, Holly E. Barker, Justin Bedő, Silvia Casadei, Andrew Farrell, Marc R. Radke, Kristy Shield‐Artin, Jocelyn Sietsma Penington, Franziska Geissler, Elizabeth L. Kyran, Robert Betsch, Lijun Xu, Fan Zhang, Alexander Dobrovic, Inger Olesen, Rebecca Kristeleit, Amit M. Oza, Iain A. McNeish, Gayanie Ratnayake, Nadia Traficante, Georgia Chenevix‐Trench, A Green, Penelope M. Webb, Dorota M. Gertig, Sián Fereday, Suzanne Moore, Jillian A. Hung, K.R. Harrap, T. Sadkowsky, Nirmala Pandeya, M. Malt, A. Mellon, R. Paul Robertson, T. Vanden Bergh, Milissa U. Jones, P. Mackenzie, J. Maidens, K. Nattress, Yoke-Eng Chiew, Annie Stenlake, Harold C. Sullivan, Brian M. Alexander, P. Ashover, Stephen M. Brown, T. Corrish, L. Green, L. M. Jackman, Kaltin Ferguson, Karla Martin, A. Martyn, B. Ranieri, J. White, V. Jayde, Pam Mamers, Leanne Bowes, Laura Galletta, Daniel A. Giles, Joy Hendley, Thomas Schmidt, H. Shirley, C. Ball, Christian D. Young, S. Viduka, Hang Tran, Sanela Bilic, Lydia Glavinas, Julia Brooks, R. Stuart‐Harris, Fred Kirsten, J Rutovitz, P. Clingan, Akisha Glasgow, Anthony Proietto, Stephen Braye, Geoffrey Otton, Jenny Shannon, Tony Bonaventura, James Stewart, Stephen Begbie, Michael Friedländer, Debra Bell, Sally Baron‐Hay, A. Ferrier, G. Gard, David Nevell, Nick Pavlakis, Susan Valmadre, Bruce Young, C. Camaris, R. Crouch, L. Edwards, Neville F. Hacker, Donald E. Marsden, Greg Robertson, Philip Beale, Jane Beith, Jonathan Carter, C. Dalrymple, R. Houghton, Prudence A. Russell, Matthew Links, John J. Grygiel, Jane Hill, Alison H. Brand, Karen Byth, Richard Jaworski, Paul R. Harnett, R. Sharma, Gerard Wain, B. Ward, D. Papadimos, A. Crandon, Michael P. Cummings, K. Horwood, Andreas Obermair, Lewis Perrin, David Wyld, James Nicklin, Marcus Davy, Martin K. Oehler, Cathrine Hall, Tom Dodd, Timothy M. Healy, Keir Pittman, Deborah J. Henderson, Jessica A. Miller, J. Pierdes, Penny Blomfield, D. Challis, Rachel McIntosh, Alyssa Parker, Robert Brown, Robert Rome, D. Allen, Peter Grant, Simon Hyde, R. Laurie, Melissa Robbie, David Healy, Tom Jobling, T. Manolitsas, J. McNealage, Peter A. W. Rogers, B. Susil, E. Sumithran, Ian Simpson, Kelly‐Anne Phillips, Danny Rischin, Stephen B. Fox, Debi Johnson, Stephen Lade, Maurice B. Loughrey, N. O’Callaghan, William K. Murray, Paul Waring, Virginia Billson, Jan Pyman, Deborah Neesham, Michael Quinn, Craig Underhill, Rachel Bell, L. F. Ng, Robert Blum, Vinod Ganju, Ian Hammond, Yee Leung, Anthony J. McCartney, Martin Buck, I. Haviv, D. Purdie, David C. Whiteman, Nikolajs Zeps, Anna DeFazio, David D.L. Bowtell, Thomas C. Harding, Kevin Lin, Elizabeth M. Swisher, Olga Kondrashova, Clare L. Scott, Neil Johnson, Matthew J. Wakefield

Bibliographic record

VenueMolecular Cancer · 2024
Typeletter
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicCRISPR and Genetic Engineering
Canadian institutionsPrincess Margaret Cancer Centre
FundersNational Health and Medical Research CouncilNational Cancer InstituteMedical Research CouncilClovis OncologyQIMR Berghofer Medical Research Institute
KeywordsBiologyPARP inhibitorExon skippingExonGeneticsspliceSplice site mutationMutationCancer researchPoly ADP ribose polymeraseMolecular biologyAlternative splicingDNAGenePolymerase

Abstract

fetched live from OpenAlex

PARP inhibitor (PARPi) therapy has transformed outcomes for patients with homologous recombination DNA repair (HRR) deficient ovarian cancers, for example those with BRCA1 or BRCA2 gene defects. Unfortunately, PARPi resistance is common. Multiple resistance mechanisms have been described, including secondary mutations that restore the HR gene reading frame. BRCA1 splice isoforms △11 and △11q can contribute to PARPi resistance by splicing out the mutation-containing exon, producing truncated, partially functional proteins. However, the clinical impacts and underlying drivers of BRCA1 exon skipping are not fully understood.We analyzed nine ovarian and breast cancer patient derived xenografts (PDX) with BRCA1 exon 11 frameshift mutations for exon skipping and therapy response, including a matched PDX pair derived from a patient pre- and post-chemotherapy/PARPi. BRCA1 exon 11 skipping was elevated in PARPi resistant PDX tumors. Two independent PDX models acquired secondary BRCA1 splice site mutations (SSMs) that drive exon skipping, confirmed using qRT-PCR, RNA sequencing, immunoblotting and minigene modelling. CRISPR/Cas9-mediated disruption of splicing functionally validated exon skipping as a mechanism of PARPi resistance. SSMs were also enriched in post-PARPi ovarian cancer patient cohorts from the ARIEL2 and ARIEL4 clinical trials.Few PARPi resistance mechanisms have been confirmed in the clinical setting. While secondary/reversion mutations typically restore a gene's reading frame, we have identified secondary mutations in patient cohorts that hijack splice sites to enhance mutation-containing exon skipping, resulting in the overexpression of BRCA1 hypomorphs, which in turn promote PARPi resistance. Thus, BRCA1 SSMs can and should be clinically monitored, along with frame-restoring secondary mutations.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesMeta-epidemiology (narrow)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: Not applicable
GenreCandidate signal: Commentary · Consensus signal: none
Teacher disagreement score0.460
Threshold uncertainty score1.000

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.005
GPT teacher head0.267
Teacher spread0.261 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

Study designNot applicable
Domainnot available
GenreCommentary

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations28
Published2024
Admission routes1
Has abstractyes

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