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Record W2476341356 · doi:10.1136/jmedgenet-2016-103839

<i>PALB2</i>, <i>CHEK2</i> and <i>ATM</i> rare variants and cancer risk: data from COGS

2016· article· en· W2476341356 on OpenAlexafffund
Melissa C. Southey, Robert Winqvist, Katri Pylkäs, Fergus J. Couch, Marc Tischkowitz, William D. Foulkes, Joe Dennis, Kyriaki Michailidou, Elizabeth J. van Rensburg, Tuomas Heikkinen, Heli Nevanlinna, John L. Hopper, Thilo Dörk, Kathleen Claes, Jorge S. Reis‐Filho, Zhi L. Teo, Paolo Radice, Irene Catucci, Paolo Peterlongo, Helen Tsimiklis, Fabrice Odefrey, James G. Dowty, Marjanka K. Schmidt, Annegien Broeks, Frans B.L. Hogervorst, Senno Verhoef, Jane Carpenter, Christine L. Clarke, Rodney J. Scott, Peter A. Fasching, Lothar Haeberle, Arif B. Ekici, Matthias W. Beckmann, Julian Peto, Isabel dos‐Santos‐Silva, Olivia Fletcher, Nichola Johnson, Manjeet K. Bolla, Elinor J. Sawyer, Ian Tomlinson, Michael J. Kerin, Nicola Miller, F. Marmé, Barbara Burwinkel, Rongxi Yang, Pascal Guénel, Thérèse Truong, F. Ménégaux, Stig E. Bojesen, Sune F. Nielsen, Henrik Flyger, Javier Benı́tez, M. Pilar Zamora, José Ignacio Arias Pérez, Primitiva Menéndez, Hoda Anton‐Culver, Susan L. Neuhausen, Argyrios Ziogas, Christina A. Clarke, Hermann Brenner, Volker Arndt, Christa Stegmaier, Hiltrud Brauch, Thomas Brüning, Yon‐Dschun Ko, Taru Muranen, Kristiina Aittomäki, Carl Blomqvist, Natalia Bogdanova, Natalia Antonenkova, Annika Lindblom, Sara Margolin, Vesa Kataja, Veli‐Matti Kosma, Jaana M. Hartikainen, Amanda B. Spurdle, kConFab Investigators, Els Wauters, Dominiek Smeets, Benoit Beuselinck, Giuseppe Floris, Jenny Chang‐Claude, Anja Rudolph, Petra Seibold, Dieter Flesch‐Janys, Janet E. Olson, Celine M. Vachon, V. Shane Pankratz, Catriona McLean, Christopher A. Haiman, Brian E. Henderson, Fredrick R. Schumacher, Loı̈c Le Marchand, Vessela Kristensen, Grethe Grenaker Alnæs, Wei Zheng, David J. Hunter, Sara Lindström, Susan E. Hankinson, Peter Kraft, Irene L. Andrulis, Julia A. Knight, Gord Glendon, Anna Marie Mulligan, Arja Jukkola‐Vuorinen, Mervi Grip, Saila Kauppila, Peter Devilee, Robert A.E.M. Tollenaar, Caroline Seynaeve, Antoinette Hollestelle, Montserrat García‐Closas, Jonine D. Figueroa, Stephen J. Chanock, Jolanta Lissowska, Kamila Czene, Hatef Darabi, Mikael Eriksson, Sajjad Rafiq, William Tapper, Sue Gerty, Maartje J. Hooning, John W.M. Martens, J. Margriet Collée, Madeleine M.A. Tilanus‐Linthorst, Per Hall, Jingmei Li, Judith S. Brand, Keith Humphreys, Angela Cox, Malcolm Reed, Craig Luccarini, Caroline Baynes, Alison M. Dunning, Ute Hamann, Diana Torres, Hans Ulrich Ulmer, Thomas Rüdiger, Anna Jakubowska, Jan Lubiński, Katarzyna Jaworska, Katarzyna Durda, Susan Slager, Amanda E. Toland, Christine B. Ambrosone, Drakoulis Yannoukakos, Anthony J. Swerdlow, Alan Ashworth, Nick Orr, Michael E. Jones, Anna González‐Neira, Guillermo Pita, M. Rosario Alonso, Núria Álvarez, Daniel Herrero, Daniel C. Tessier, Daniel Vincent, François Bacot, Jacques Simard, Martine Dumont, Penny Soucy, Rosalind A. Eeles, Kenneth Muir, Fredrik Wiklund, Henrik Grönberg, Johanna Schleutker, Børge G. Nordestgaard, Maren Weischer, Ruth C. Travis, David E. Neal, Jenny Donovan, Freddie C. Hamdy, Kay‐Tee Khaw, Janet L. Stanford, William J. Blot, Stephen N. Thibodeau, Daniel J. Schaid, Joseph L. Kelley, Christiane Maier, Adam S. Kibel, Cezary Cybulski, Lisa Cannon‐Albright, Katja Butterbach, Jong Moon Park, Radka Kaneva, Jyotsna Batra, Manuel R. Teixeira, Zsofia Kote‐Jarai, Ali Amin Al Olama, Sara Benlloch, Stefan P. Renner, Arndt Hartmann, Alexander Hein, Matthias Ruebner, Diether Lambrechts, Els Van Nieuwenhuysen, Ignace Vergote, Sandrina Lambretchs, Jennifer A. Doherty, Mary Anne Rossing, Stefan Nickels, Ursula Eilber, Shan Wang‐Gohrke, Kunle Odunsi, Lara Sucheston‐Campbell, Grace Friel, Galina Lurie, Jeffrey Killeen, Lynne R. Wilkens, Marc T. Goodman, Ingo B. Runnebaum, Peter Hillemanns, Liisa M. Pelttari, Ralf Bützow, Francesmary Modugno, Robert P. Edwards, Roberta B. Ness, Kirsten B. Moysich, Andreas du Bois, Florian Heitz, Philipp Harter, Stefan Kommoss, Beth Y. Karlan, Christine Walsh, Jenny Lester, Allan Jensen, Susanne K. Kjær, Estrid Høgdall, Bernard Peissel, Bernardo Bonanni, Loris Bernard, Ellen L. Goode, Brooke L. Fridley, Robert A. Vierkant, Julie M. Cunningham, Melissa C. Larson, Zachary C. Fogarty, Kimberly R. Kalli, Dong Liang, Karen H. Lu, Michelle A.T. Hildebrandt, Xifeng Wu, Douglas A. Levine, Fanny Dao, Maria Bisogna, Andrew Berchuck, Edwin S. Iversen, Jeffrey R. Marks, Lucy Akushevich, Daniel W. Cramer, Joellen M. Schildkraut, Kathryn L. Terry, Elizabeth M. Poole, Meir J. Stampfer, Shelley S. Tworoger, Elisa V. Bandera, Irene Orlow, Sara H. Olson, Line Bjørge, Helga B. Salvesen, Anne M. van Altena, Katja K.H. Aben, Lambertus A. Kiemeney, Leon F.A.G. Massuger, Tanja Pejović, Yukie T. Bean, Angela Brooks‐Wilson, Linda E. Kelemen, Linda S. Cook, Nhu D. Le, Bohdan Górski, Jacek Gronwald, Janusz Menkiszak, Claus Høgdall, Lene Lundvall, Lotte Nedergaard, Svend Aage Engelholm, Ed Dicks, Jonathan P. Tyrer, Ian Campbell, Iain A. McNeish, James Paul, Nadeem Siddiqui, Rosalind Glasspool, Alice S. Whittemore, Joseph H. Rothstein, Valerie McGuire, Weiva Sieh, Hui Cai, Xiao‐Ou Shu, Rachel Threet Teten, Rebecca Sutphen, Steven A. Narod, Catherine M. Phelan, Álvaro N.A. Monteiro, David Fenstermacher, Hui‐Yi Lin, Jennifer B. Permuth, Thomas A. Sellers, Y. Ann Chen, Ya-Yu Tsai, Zhihua Chen, Aleksandra Gentry‐Maharaj, Simon A. Gayther, Susan J. Ramus, Usha Menon, Anna H. Wu, Celeste Leigh Pearce, David Van Den Berg, Malcolm C. Pike, Agnieszka Dansonka‐Mieszkowska, Joanna Plisiecka-Hałasa, Joanna Moes-Sosnowska, Jolanta Kupryjańczyk, Paul D.P. Pharoah, Honglin Song, Ingrid Winship, Georgia Chenevix‐Trench, Graham G. Giles, Sean V. Tavtigian, Doug Easton, Roger L. Milne

Bibliographic record

VenueJournal of Medical Genetics · 2016
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicBRCA gene mutations in cancer
Canadian institutionsCanada's Michael Smith Genome Sciences CentreUniversité LavalBC Cancer AgencyWomen's College HospitalCentre hospitalier universitaire de QuébecSimon Fraser UniversityMcGill University Health CentreMcGill UniversityMcGill University and Génome Québec Innovation CentrePublic Health OntarioUniversity Health NetworkUniversity of TorontoMount Sinai HospitalLunenfeld-Tanenbaum Research Institute
FundersNational Cancer InstituteMedical Research CouncilCanadian Institutes of Health ResearchDeutsche KrebshilfeNational Institute for Health and Care ResearchNational Health and Medical Research CouncilCancer Research UKFrancis Crick InstituteWellcome TrustNational Center for Research ResourcesGénome QuébecSusan G. KomenMemorial Sloan-Kettering Cancer CenterNational Institutes of HealthOvarian Cancer Research FundBreast Cancer Research FoundationU.S. Department of DefenseMcGill UniversityRoyal Marsden NHS Foundation Trust
KeywordsCHEK2PALB2GeneticsCancerBiologyMutationGeneGermline mutation

Abstract

fetched live from OpenAlex

BACKGROUND: The rarity of mutations in PALB2, CHEK2 and ATM make it difficult to estimate precisely associated cancer risks. Population-based family studies have provided evidence that at least some of these mutations are associated with breast cancer risk as high as those associated with rare BRCA2 mutations. We aimed to estimate the relative risks associated with specific rare variants in PALB2, CHEK2 and ATM via a multicentre case-control study. METHODS: We genotyped 10 rare mutations using the custom iCOGS array: PALB2 c.1592delT, c.2816T>G and c.3113G>A, CHEK2 c.349A>G, c.538C>T, c.715G>A, c.1036C>T, c.1312G>T, and c.1343T>G and ATM c.7271T>G. We assessed associations with breast cancer risk (42 671 cases and 42 164 controls), as well as prostate (22 301 cases and 22 320 controls) and ovarian (14 542 cases and 23 491 controls) cancer risk, for each variant. RESULTS: ) and ATM c.7271T>G OR 11.0 (95% CI 1.42 to 85.7, p=0.0012). We also found evidence of association with breast cancer risk for three variants in CHEK2, c.349A>G OR 2.26 (95% CI 1.29 to 3.95), c.1036C>T OR 5.06 (95% CI 1.09 to 23.5) and c.538C>T OR 1.33 (95% CI 1.05 to 1.67) (p≤0.017). Evidence for prostate cancer risk was observed for CHEK2 c.1343T>G OR 3.03 (95% CI 1.53 to 6.03, p=0.0006) for African men and CHEK2 c.1312G>T OR 2.21 (95% CI 1.06 to 4.63, p=0.030) for European men. No evidence of association with ovarian cancer was found for any of these variants. CONCLUSIONS: This report adds to accumulating evidence that at least some variants in these genes are associated with an increased risk of breast cancer that is clinically important.

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)0.003
metaresearch head score (Gemma)0.006
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 score0.023
Threshold uncertainty score0.046

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0030.006
Meta-epidemiology (narrow)0.0010.000
Meta-epidemiology (broad)0.0010.001
Bibliometrics0.0030.003
Science and technology studies0.0010.000
Scholarly communication0.0010.000
Open science0.0010.001
Research integrity0.0010.000
Insufficient payload (model declined to judge)0.0040.001

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 head0.023
GPT teacher head0.309
Teacher spread0.286 · 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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Citations217
Published2016
Admission routes2
Has abstractyes

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Same venueJournal of Medical GeneticsSame topicBRCA gene mutations in cancerFrench-language works237,207