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Record W4415680787 · doi:10.1016/j.breast.2025.104619

Large-scale meta-analysis and precision functional assays identify FANCM regions in which PTVs confer different risks for ER-negative and triple-negative breast cancer

2025· article· en· W4415680787 on OpenAlexafffund
Amandine Billaud, Gisella Figlioli, Irene Casamassima, Violette Azzoni, Jahnavi Srivatsa, Mara Colombo, Laura Caleca, Thomas U. Ahearn, Irene L. Andrulis, Antonis C Antoniou, Matthias W. Beckmann, Sabine Behrens, Marina Bermisheva, Natalia Bogdanova, Manjeet K. Bolla, Bernardo Bonanni, Thomas Brüning, Nicola J. Camp, Archie Campbell, Jose E. Castelao, Melissa H. Cessna, Jenny Chang‐Claude, Kamila Czene, Joe Dennis, Peter Devilee, Thilo Dörk, Alison M. Dunning, Mikael Eriksson, D. Gareth Evans, Peter A. Fasching, Jonine D. Figueroa, Marike Gabrielson, Manuela Gago-Domínguez, Anna González‐Neira, Pascal Guénel, Andreas Hadjisavvas, Eric Hahnen, Ute Hamann, Peter Hillemanns, Antoinette Hollestelle, Maartje J. Hooning, Reiner Hoppe, Anthony Howell, Anna Jakubowska, Vessela N. Kristensen, Jan Lubiński, Michael Lush, Siranoush Manoukian, Dimitrios Mavroudis, Roger L. Milne, Anna Marie Mulligan, William G. Newman, Nadia Obi, Mihalis I. Panayiotidis, Guillermo Pita, Muhammad Usman Rashid, Valerie Rhenius, Emmanouil Saloustros, Elinor J. Sawyer, Rita K. Schmutzler, Mitul Shah, Melissa C. Southey, Amanda B. Spurdle, Ian Tomlinson, Thérèse Truong, Qin Wang, Camilla Wendt, Paul L. Auer, Nicholas Boddicker, Clara Bodelón, Elizabeth S. Burnside, Fei Chen, Fergus J. Couch, Susan M. Domchek, A. Heather Eliassen, Christopher Haiman, James M. Hodge, Chunling Hu, Hongyan Huang, Sara Lindström, Maria Elena Martinez, Katherine L. Nathanson, Susan L. Neuhausen, Katie M. O’Brien, Janet E. Olson, Julie R. Palmer, Alpa V. Patel, Kathryn J. Ruddy, Dale P. Sandler, Lauren R. Teras, Clarice R. Weinberg, Jeffrey N. Weitzel, Stacey J. Winham, Siddhartha Yadav, Song Yao, Gary Zirpoli, Markéta Janatová, Zdeněk Kleibl, Petra Kleiblová, Jana Soukupová, Qihong Zhao, Lisa Devereux, Paul A. James, Ian Campbell, Tú Nguyen‐Dumont, James G. Dowty, Nadine Andrieu, Fabienne Lesueur, Dominique Stoppa-Lyonnet, Miguel de la Hoya, Paolo Radice, Claus Storgaard Sørensen, Paolo Peterlongo

Bibliographic record

VenueThe Breast · 2025
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicBRCA gene mutations in cancer
Canadian institutionsUniversity Health NetworkLunenfeld-Tanenbaum Research Institute
FundersMedical Research and Materiel CommandDivision of Graduate EducationNIHR Cambridge Biomedical Research CentreServicio Gallego de SaludInstituto de Salud Carlos IIICancer Council TasmaniaNational Health and Medical Research CouncilMedical Research CouncilCanadian Institutes of Health ResearchProgramme Grants for Applied ResearchManchester Biomedical Research CentreU.S. ArmyNational Institutes of HealthCancer Council VictoriaMinistry of Science and Higher Education of the Russian FederationXunta de GaliciaAgence Nationale de Sécurité Sanitaire de l’Alimentation, de l’Environnement et du TravailInstitut National Du CancerDeutsche KrebshilfeMedizinischen Hochschule HannoverAssociazione Italiana per la Ricerca sul CancroFondazione Umberto VeronesiUniversity of UtahNIHR Biomedical Research Centre, Royal Marsden NHS Foundation Trust/Institute of Cancer ResearchFondation de FranceMinistero della SaluteUniversity of CreteKuopion Yliopistollinen SairaalaUniversity of Wisconsin-MadisonKarolinska InstitutetDeutsche ForschungsgemeinschaftNederlandse Organisatie voor Wetenschappelijk OnderzoekDeutsche Gesetzliche UnfallversicherungChief Scientist Office, Scottish Government Health and Social Care DirectorateBundesministerium für Bildung und ForschungMinisterio de Economía y CompetitividadCancer AustraliaAgence Nationale de la RechercheRobert Bosch StiftungCancerfondenNational Cancer InstituteCancer Institute NSWNational Breast Cancer FoundationEuropean CommissionUniversity of CambridgeMinisterio de Sanidad, Servicios Sociales e IgualdadGovernment of CanadaWellcome TrustFondation du cancer du sein du QuébecKWF KankerbestrijdingCancer Research UKItä-Suomen YliopistoGenome CanadaDeutsches KrebsforschungszentrumFondazione AIRC per la ricerca sul cancro ETSNational Institute for Health and Care ResearchScottish GovernmentDivision of Cancer Prevention, National Cancer InstituteDepartment of Health and Social CareFreistaat SachsenHuntsman Cancer InstituteAgency for Science, Technology and ResearchUniversity of PennsylvaniaScottish Funding CouncilKreftforeningenCancer Council South AustraliaAmerican Cancer SocietyMinisterie van Volksgezondheid, Welzijn en SportU.S. Department of Health and Human ServicesBreast Cancer Research FoundationCancer Council NSWSusan G. Komen for the CureResearch Promotion FoundationEuropean Regional Development FundKing's College London
KeywordsBreast cancerGermlineDiseaseCancerMutationGenetic testing

Abstract

fetched live from OpenAlex

The breast cancer risk conferred by germline protein truncating variants (PTVs) in known and putative breast cancer genes has been extensively investigated. However, the effect of FANCM PTVs on breast cancer risk remains unclear. Our previous clinical, genetic and functional results on the N-terminal p.Arg658∗ and the two C-terminal p.Gln1701∗ and p.Gly1906Alafs∗12 variants suggested that FANCM PTVs may confer different risks for ER-negative (ER-neg) and triple-negative (TN) breast cancer subtypes. Here, we performed meta-analyses of seven studies totaling 144 681 breast cancer cases and 123 632 controls. FANCM PTVs were tested for association with breast cancer risk overall and the disease clinical subtypes by single variant and burden analyses. Two CRISPR-Cas9-based functional assays were also conducted to test the fitness of cells after knock-in of the p.Arg658∗, p.Gln1701∗ and p.Gly1906Alafs∗12 PTVs and the sensitivity of different FANCM regions to genome editing. Our results suggest that the N-terminal FANCM region upstream of p.Tyr725 harbors essential functions, whereas downstream regions appear dispensable. This is supported by our genetic data which indicate that all FANCM PTVs, excluding the two C-terminal p.Gln1701∗ and p.Gly1906Alafs∗12, are associated with an increased risk of ER-neg (OR = 1.41, P = 0.023) and TN (OR = 1.64, P = 0.0023). Notably, PTVs upstream of AA position 670 are associated with a moderate risk of developing TN breast cancer, and that even when the p.Arg658∗ carriers were excluded from the analysis. Importantly, our results confirm previous data indicating that p.Arg658∗ carriers are at moderate risk of developing ER-neg (OR = 2.08, P = 0.030) and TN (OR = 3.26; P = 0.0034), whereas carriers of p.Gln1701∗ and p.Gly1906Alafs∗12 should not be considered at increased risk. Our data are useful for counseling carriers of FANCM PTVs, but further analyses are warranted to obtain more precise risk estimates.

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 categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.656
Threshold uncertainty score0.686

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 integrity0.0000.000
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 head0.045
GPT teacher head0.343
Teacher spread0.298 · 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.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

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".

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Citations1
Published2025
Admission routes2
Has abstractyes

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