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Record W2989421973 · doi:10.1200/jco.19.01907

Cancer Risks Associated With Germline<i>PALB2</i>Pathogenic Variants: An International Study of 524 Families

2019· article· en· W2989421973 on OpenAlexafffund
Xin Yang, Goska Leslie, Alicja Doroszuk, Sandra Schneider, Jamie Allen, Brennan Decker, Alison M. Dunning, James Redman, James A. Scarth, Inga Plaskocinska, Craig Luccarini, Mitul Shah, Karen A. Pooley, Leila Dorling, Andrew Lee, Muriel A. Adank, Julian Adlard, Kristiina Aittomäki, Irene L. Andrulis, Peter Ang, Julian Barwell, Jonine L. Bernstein, Kristie Bobolis, Åke Borg, Carl Blomqvist, Kathleen Claes, Patrick Concannon, Adeline Cuggia, Julie O. Culver, Francesca Damiola, Antoine De Pauw, Orland Dı́ez, Jill S. Dolinsky, Susan M. Domchek, Christoph Engel, D. Gareth Evans, Florentia Fostira, Judy E. Garber, Lisa Golmard, Ellen L. Goode, Stephen B. Gruber, Eric Hahnen, Christopher R. Hake, Tuomas Heikkinen, Judith Hurley, Ramūnas Janavičius, Zdeněk Kleibl, Petra Kleiblová, Irene Konstantopoulou, Anders Kvist, Holly LaDuca, Ann S. G. Lee, Fabienne Lesueur, Eamonn R. Maher, Siranoush Manoukian, Rachel McFarland, Wendy McKinnon, Alfons Meindl, Kelly Metcalfe, Nur Aishah Mohd Taib, Jukka S. Moilanen, Katherine L. Nathanson, Susan L. Neuhausen, Pei Sze Ng, Tú Nguyen‐Dumont, Sarah M. Nielsen, Florian Obermair, Kenneth Offit, Olufunmilayo I. Olopade, Laura Ottini, Judith Penkert, Katri Pylkäs, Paolo Radice, Susan J. Ramus, Vilius Rudaitis, Lucy Side, Rachel Silva‐Smith, Valentina Silvestri, Anne‐Bine Skytte, Thomas Slavin, Jana Soukupová, Carlo Tondini, Alison H. Trainer, Gary Unzeitig, Lydia Usha, Thomas van Overeem Hansen, James Whitworth, Marie Wood, Cheng Har Yip, Sook‐Yee Yoon, Amal Yussuf, George Zogopoulos, David E. Goldgar, John L. Hopper, Georgia Chenevix‐Trench, Paul D.P. Pharoah, Sophia George, Judith Balmañà, Claude Houdayer, Paul A. James, Zaki El-Haffaf, Hans Ehrencrona, Markéta Janatová, Paolo Peterlongo, Heli Nevanlinna, Rita K. Schmutzler, Soo‐Hwang Teo, Mark E. Robson, Tuya Pal, Fergus J. Couch, Jeffrey N. Weitzel, Aaron Elliott, Melissa C. Southey, Robert Winqvist, Douglas F. Easton, William D. Foulkes, Antonis C. Antoniou, Marc Tischkowitz

Bibliographic record

VenueJournal of Clinical Oncology · 2019
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicBRCA gene mutations in cancer
Canadian institutionsMcGill UniversityCentre Hospitalier de l’Université de MontréalUniversity of TorontoMcGill University Health CentreLunenfeld-Tanenbaum Research InstituteMount Sinai Hospital
FundersCongressionally Directed Medical Research ProgramsNational Institute of Environmental Health SciencesCanadian Institutes of Health ResearchVšeobecná Fakultní Nemocnice v PrazeUppsala UniversitetMedical Research Councillékařská fakulta Univerzity KarlovyManchester Biomedical Research CentreNational Institutes of HealthDirection Générale de l’offre de SoinsNational Institute for Health and Care ResearchVlaamse regeringSahlgrenska UniversitetssjukhusetLietuvos Mokslo TarybaNational Health and Medical Research CouncilOulun YliopistoUniversiti MalayaNational Cancer InstituteMinisterstvo Zdravotnictví Ceské RepublikyKuopion Yliopistollinen SairaalaNational Medical Research CouncilNIH Office of the DirectorUniversity of TorontoMinistero dell’Istruzione, dell’Università e della RicercaLunds UniversitetMinistero della SaluteCancer AustraliaNational Breast Cancer FoundationNational Institute on Minority Health and Health DisparitiesEuropean CommissionAkademiska SjukhusetAcademy of FinlandUniverzita Karlova v PrazeBreast Cancer Research FoundationGovernment of CanadaUniversity of CambridgeFondation du cancer du sein du QuébecWellcome TrustCancer Research UKMyriad GeneticsDipartimenti di EccellenzaInstitut National Du CancerYayasan Sime DarbySusan G. KomenAssociazione Italiana per la Ricerca sul CancroGenome CanadaItä-Suomen YliopistoUniversity of Miami
KeywordsPALB2MedicineGermlineCancerGermline mutationGeneticsOncologyInternal medicineMutationGeneBiology

Abstract

fetched live from OpenAlex

PURPOSE To estimate age-specific relative and absolute cancer risks of breast cancer and to estimate risks of ovarian, pancreatic, male breast, prostate, and colorectal cancers associated with germline PALB2 pathogenic variants (PVs) because these risks have not been extensively characterized. METHODS We analyzed data from 524 families with PALB2 PVs from 21 countries. Complex segregation analysis was used to estimate relative risks (RRs; relative to country-specific population incidences) and absolute risks of cancers. The models allowed for residual familial aggregation of breast and ovarian cancer and were adjusted for the family-specific ascertainment schemes. RESULTS We found associations between PALB2 PVs and risk of female breast cancer (RR, 7.18; 95% CI, 5.82 to 8.85; P = 6.5 × 10−76), ovarian cancer (RR, 2.91; 95% CI, 1.40 to 6.04; P = 4.1 × 10−3), pancreatic cancer (RR, 2.37; 95% CI, 1.24 to 4.50; P = 8.7 × 10−3), and male breast cancer (RR, 7.34; 95% CI, 1.28 to 42.18; P = 2.6 × 10−2). There was no evidence for increased risks of prostate or colorectal cancer. The breast cancer RRs declined with age ( P for trend = 2.0 × 10−3). After adjusting for family ascertainment, breast cancer risk estimates on the basis of multiple case families were similar to the estimates from families ascertained through population-based studies ( P for difference = .41). On the basis of the combined data, the estimated risks to age 80 years were 53% (95% CI, 44% to 63%) for female breast cancer, 5% (95% CI, 2% to 10%) for ovarian cancer, 2%-3% (95% CI females, 1% to 4%; 95% CI males, 2% to 5%) for pancreatic cancer, and 1% (95% CI, 0.2% to 5%) for male breast cancer. CONCLUSION These results confirm PALB2 as a major breast cancer susceptibility gene and establish substantial associations between germline PALB2 PVs and ovarian, pancreatic, and male breast cancers. These findings will facilitate incorporation of PALB2 into risk prediction models and optimize the clinical cancer risk management of PALB2 PV carriers.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.003
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.009
Threshold uncertainty score0.018

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.003
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0010.002
Science and technology studies0.0010.001
Scholarly communication0.0010.000
Open science0.0000.001
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0020.000

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 head0.088
GPT teacher head0.463
Teacher spread0.374 · 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 source (direct Gemma or distilled Codex), 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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Citations418
Published2019
Admission routes2
Has abstractyes

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