Ovarian and Breast Cancer Risks Associated With Pathogenic Variants in RAD51C and RAD51D.


Journal

Journal of the National Cancer Institute
ISSN: 1460-2105
Titre abrégé: J Natl Cancer Inst
Pays: United States
ID NLM: 7503089

Informations de publication

Date de publication:
14 12 2020
Historique:
received: 06 12 2019
revised: 07 02 2020
accepted: 25 02 2020
pubmed: 29 2 2020
medline: 29 4 2021
entrez: 29 2 2020
Statut: ppublish

Résumé

The purpose of this study was to estimate precise age-specific tubo-ovarian carcinoma (TOC) and breast cancer (BC) risks for carriers of pathogenic variants in RAD51C and RAD51D. We analyzed data from 6178 families, 125 with pathogenic variants in RAD51C, and 6690 families, 60 with pathogenic variants in RAD51D. TOC and BC relative and cumulative risks were estimated using complex segregation analysis to model the cancer inheritance patterns in families while adjusting for the mode of ascertainment of each family. All statistical tests were two-sided. Pathogenic variants in both RAD51C and RAD51D were associated with TOC (RAD51C: relative risk [RR] = 7.55, 95% confidence interval [CI] = 5.60 to 10.19; P = 5 × 10-40; RAD51D: RR = 7.60, 95% CI = 5.61 to 10.30; P = 5 × 10-39) and BC (RAD51C: RR = 1.99, 95% CI = 1.39 to 2.85; P = 1.55 × 10-4; RAD51D: RR = 1.83, 95% CI = 1.24 to 2.72; P = .002). For both RAD51C and RAD51D, there was a suggestion that the TOC relative risks increased with age until around age 60 years and decreased thereafter. The estimated cumulative risks of developing TOC to age 80 years were 11% (95% CI = 6% to 21%) for RAD51C and 13% (95% CI = 7% to 23%) for RAD51D pathogenic variant carriers. The estimated cumulative risks of developing BC to 80 years were 21% (95% CI = 15% to 29%) for RAD51C and 20% (95% CI = 14% to 28%) for RAD51D pathogenic variant carriers. Both TOC and BC risks for RAD51C and RAD51D pathogenic variant carriers varied by cancer family history and could be as high as 32-36% for TOC, for carriers with two first-degree relatives diagnosed with TOC, or 44-46% for BC, for carriers with two first-degree relatives diagnosed with BC. These estimates will facilitate the genetic counseling of RAD51C and RAD51D pathogenic variant carriers and justify the incorporation of RAD51C and RAD51D into cancer risk prediction models.

Sections du résumé

BACKGROUND
The purpose of this study was to estimate precise age-specific tubo-ovarian carcinoma (TOC) and breast cancer (BC) risks for carriers of pathogenic variants in RAD51C and RAD51D.
METHODS
We analyzed data from 6178 families, 125 with pathogenic variants in RAD51C, and 6690 families, 60 with pathogenic variants in RAD51D. TOC and BC relative and cumulative risks were estimated using complex segregation analysis to model the cancer inheritance patterns in families while adjusting for the mode of ascertainment of each family. All statistical tests were two-sided.
RESULTS
Pathogenic variants in both RAD51C and RAD51D were associated with TOC (RAD51C: relative risk [RR] = 7.55, 95% confidence interval [CI] = 5.60 to 10.19; P = 5 × 10-40; RAD51D: RR = 7.60, 95% CI = 5.61 to 10.30; P = 5 × 10-39) and BC (RAD51C: RR = 1.99, 95% CI = 1.39 to 2.85; P = 1.55 × 10-4; RAD51D: RR = 1.83, 95% CI = 1.24 to 2.72; P = .002). For both RAD51C and RAD51D, there was a suggestion that the TOC relative risks increased with age until around age 60 years and decreased thereafter. The estimated cumulative risks of developing TOC to age 80 years were 11% (95% CI = 6% to 21%) for RAD51C and 13% (95% CI = 7% to 23%) for RAD51D pathogenic variant carriers. The estimated cumulative risks of developing BC to 80 years were 21% (95% CI = 15% to 29%) for RAD51C and 20% (95% CI = 14% to 28%) for RAD51D pathogenic variant carriers. Both TOC and BC risks for RAD51C and RAD51D pathogenic variant carriers varied by cancer family history and could be as high as 32-36% for TOC, for carriers with two first-degree relatives diagnosed with TOC, or 44-46% for BC, for carriers with two first-degree relatives diagnosed with BC.
CONCLUSIONS
These estimates will facilitate the genetic counseling of RAD51C and RAD51D pathogenic variant carriers and justify the incorporation of RAD51C and RAD51D into cancer risk prediction models.

Identifiants

pubmed: 32107557
pii: 5764125
doi: 10.1093/jnci/djaa030
pmc: PMC7735771
doi:

Substances chimiques

DNA-Binding Proteins 0
RAD51C protein, human 0
RAD51D protein, human 0

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

1242-1250

Subventions

Organisme : NCI NIH HHS
ID : R01 CA225662
Pays : United States
Organisme : Cancer Research UK
ID : C1005/A12677
Pays : United Kingdom
Organisme : Cancer Research UK
ID : C490/A10119
Pays : United Kingdom
Organisme : CIHR
ID : FDN-148390
Pays : Canada
Organisme : Medical Research Council
ID : MC_UU_12023/20
Pays : United Kingdom
Organisme : Cancer Research UK
ID : C490/A16561
Pays : United Kingdom
Organisme : Cancer Research UK
ID : C490/A10124
Pays : United Kingdom
Organisme : NCI NIH HHS
ID : P30 CA008748
Pays : United States
Organisme : NCI NIH HHS
ID : P50 CA116201
Pays : United States
Organisme : Cancer Research UK
ID : C12292/A20861
Pays : United Kingdom
Organisme : NCI NIH HHS
ID : R01 CA178535
Pays : United States

Informations de copyright

© The Author(s) 2020. Published by Oxford University Press.

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Auteurs

Xin Yang (X)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

Honglin Song (H)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.
Department of Oncology, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

Goska Leslie (G)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

Christoph Engel (C)

Institute for Medical Informatics, Statistics and Epidemiology, University of Leipzig, Leipzig, Germany.

Eric Hahnen (E)

Faculty of Medicine and University Hospital Cologne, Center for Familial Breast and Ovarian Cancer, University of Cologne, Cologne, Germany.
Faculty of Medicine and University Hospital Cologne, Center for Integrated Oncology, University of Cologne, Cologne, Germany.

Bernd Auber (B)

Institute of Human Genetics, Hannover Medical School, Hannover, Germany.

Judit Horváth (J)

Institute of Human Genetics, University of Münster, Münster, Germany.

Karin Kast (K)

Department of Gynecology and Obstetrics, Medical Faculty and University Hospital Carl Gustav Carus, Technische Universität Dresden, Dresden, Germany.
National Center for Tumor Diseases (NCT), Dresden, Germany: German Cancer Research Center (DKFZ), Heidelberg, Germany; Faculty of Medicine and University Hospital Carl Gustav Carus, Technische Universität Dresden, Dresden, Germany; Helmholtz-Zentrum Dresden-Rossendorf (HZDR), Dresden, Germany.
German Cancer Consortium (DKTK), Dresden and German Cancer Research Center (DKFZ), Heidelberg, Germany.

Dieter Niederacher (D)

Department of Gynecology and Obstetrics, Heinrich-Heine University Düsseldorf, University Hospital Düsseldorf, Düsseldorf, Germany.

Clare Turnbull (C)

Division of Genetics and Epidemiology, The Institute of Cancer Research, London, UK.

Richard Houlston (R)

Division of Genetics and Epidemiology, The Institute of Cancer Research, London, UK.

Helen Hanson (H)

Division of Genetics and Epidemiology, The Institute of Cancer Research, London, UK.

Chey Loveday (C)

Division of Genetics and Epidemiology, The Institute of Cancer Research, London, UK.

Jill S Dolinsky (JS)

Ambry Genetics, Aliso Viejo, Canada.

Holly LaDuca (H)

Ambry Genetics, Aliso Viejo, Canada.

Susan J Ramus (SJ)

School of Women's and Children's Health, Faculty of Medicine, University of NSW Sydney, Sydney, New South Wales, Australia.
Garvan Institute of Medical Research, The Kinghorn Cancer Centre, Sydney, New South Wales, Australia.
Adult Cancer Program, Lowy Cancer Research Centre, University of NSW Sydney, Sydney, New South Wales, Australia.

Usha Menon (U)

MRC Clinical Trials Unit at UCL, Institute of Clinical Trials and Methodology, University College London, London, UK.

Adam N Rosenthal (AN)

Women's Cancer, Institute for Women's Health, University College London, London, UK.

Ian Jacobs (I)

Women's Cancer, Institute for Women's Health, University College London, London, UK.
University of New South Wales, Sydney, New South Wales, Australia.
University of Manchester, Manchester, UK.

Simon A Gayther (SA)

Center for Bioinformatics and Functional Genomics and the Cedars Sinai Genomics Core, Cedars-Sinai Medical Center, Los Angeles, CA, USA.

Ed Dicks (E)

Department of Oncology, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

Heli Nevanlinna (H)

Department of Obstetrics and Gynecology, Helsinki University Hospital, University of Helsinki, Helsinki, Finland.

Kristiina Aittomäki (K)

Department of Clinical Genetics, Helsinki University Hospital, University of Helsinki, Helsinki, Finland.

Liisa M Pelttari (LM)

Department of Obstetrics and Gynecology, Helsinki University Hospital, University of Helsinki, Helsinki, Finland.

Hans Ehrencrona (H)

Department of Clinical Genetics and Pathology, Laboratory Medicine, Office for Medical Services, Region Skåne, Lund, Sweden.
Division of Clinical Genetics, Department of Laboratory Medicine, Lund University, Lund, Sweden.

Åke Borg (Å)

Division of Oncology and Pathology, Department of Clinical Sciences Lund, Lund University, Lund, Sweden.

Anders Kvist (A)

Division of Oncology and Pathology, Department of Clinical Sciences Lund, Lund University, Lund, Sweden.

Barbara Rivera (B)

Gerald Bronfman Dept Oncology, Jewish General Hospital, McGill University and Lady Davis Institute, Montréal, QC, Canada.
Program in Molecular Mechanisms and Experimental Therapy in Oncology (Oncobell), IDIBELL, Hospitalet de Llobregat, Barcelona, Spain.

Thomas V O Hansen (TVO)

Center for Genomic Medicine, Rigshospitalet, Copenhagen University Hospital, Copenhagen, Denmark.
Department of Clinical Genetics Rigshospitalet, Copenhagen University Hospital, Copenhagen, Denmark.

Malene Djursby (M)

Department of Clinical Genetics Rigshospitalet, Copenhagen University Hospital, Copenhagen, Denmark.

Andrew Lee (A)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

Joe Dennis (J)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

David D Bowtell (DD)

Peter MacCallum Cancer Center, Melbourne, Victoria, Australia.
Sir Peter MacCallum, Department of Oncology, The University of Melbourne, Parkville, Victoria, Australia.

Nadia Traficante (N)

Peter MacCallum Cancer Center, Melbourne, Victoria, Australia.
Sir Peter MacCallum, Department of Oncology, The University of Melbourne, Parkville, Victoria, Australia.

Orland Diez (O)

Oncogenetics Group, Vall dHebron Institute of Oncology, Barcelona, Spain.
Clinical and Molecular Genetics Area, University Hospital Vall dHebron, Barcelona, Spain.

Judith Balmaña (J)

Hereditary Cancer Genetics Group, Vall d'Hebron Institute of Oncology, Barcelona, Spain.
Department of Medical Oncology, University Hospital of Vall d'Hebron, Barcelona, Spain.

Stephen B Gruber (SB)

Department of Preventive Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA.

Georgia Chenevix-Trench (G)

Department of Genetics and Computational Biology, QIMR Berghofer Medical Research Institute, Brisbane, Queensland, Australia.

kConFab Investigators (K)

Kathleen Cuningham Foundation Consortium for research into Familial Breast cancer.

Allan Jensen (A)

Department of Virus, Lifestyle and Genes, Danish Cancer Society Research Center, Copenhagen, Denmark.

Susanne K Kjær (SK)

Department of Virus, Lifestyle and Genes, Danish Cancer Society Research Center, Copenhagen, Denmark.
Department of Gynaecology, University of Copenhagen, Rigshospitalet, Copenhagen, Denmark.

Estrid Høgdall (E)

Department of Virus, Lifestyle and Genes, Danish Cancer Society Research Center, Copenhagen, Denmark.
Department of Pathology, Molecular Unit, Herlev Hospital, University of Copenhagen, Copenhagen, Denmark.

Laurent Castéra (L)

Department of Cancer Biology and Genetics, Normandy Centre for Genomic and Personalized Medicine, François Baclesse Center, Inserm U1245, Caen, France.

Judy Garber (J)

Cancer Risk and Prevention Clinic, Dana-Farber Cancer Institute, Boston, MA, USA.

Ramunas Janavicius (R)

Department of Molecular and Regenerative Medicine, Hematology, Oncology and Transfusion Medicine Center, Vilnius University Hospital Santariskiu Clinics, Vilnius, Lithuania.
State Research Institute Centre for Innovative Medicine, Vilnius, Lithuania.

Ana Osorio (A)

Centro de Investigación en Red de Enfermedades Raras, Madrid, Spain.
Human Cancer Genetics Programme, Spanish National Cancer Research Centre, Madrid, Spain.

Lisa Golmard (L)

Institut Curie, Paris Sciences Lettres Research University, Service de Génétique, Paris, France.

Ana Vega (A)

Centro de Investigación en Red de Enfermedades Raras, Madrid, Spain.
Fundación Pública Galega de Medicina Xenómica, Santiago de Compostela, Spain.
Instituto de Investigación Sanitaria de Santiago de Compostela, Complejo Hospitalario Universitario de Santiago, SERGAS, Santiago de Compostela, Spain.

Fergus J Couch (FJ)

Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, USA.

Mark Robson (M)

Department of Medicine, Memorial Sloan Kettering Cancer Center, Clinical Genetics Service, New York, NY, USA.

Jacek Gronwald (J)

Department of Genetics and Pathology, Pomeranian Medical University, Szczecin, Poland.

Susan M Domchek (SM)

Basser Center for BRCA, Abramson Cancer Center, University of Pennsylvania, Philadelphia, PA, USA.

Julie O Culver (JO)

Keck School of Medicine, University of Southern California, Los Angeles, CA, USA.

Miguel de la Hoya (M)

Molecular Oncology Laboratory CIBERONC, Hospital Clinico San Carlos, IdISSC (Instituto de Investigación Sanitaria del Hospital Clínico San Carlos), Madrid, Spain.

Douglas F Easton (DF)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.
Department of Oncology, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

William D Foulkes (WD)

Faculty of Medicine and University Hospital Cologne, Center for Integrated Oncology, University of Cologne, Cologne, Germany.
Department of Gynecology and Obstetrics, Medical Faculty and University Hospital Carl Gustav Carus, Technische Universität Dresden, Dresden, Germany.

Marc Tischkowitz (M)

Faculty of Medicine and University Hospital Cologne, Center for Integrated Oncology, University of Cologne, Cologne, Germany.
National Center for Tumor Diseases (NCT), Dresden, Germany: German Cancer Research Center (DKFZ), Heidelberg, Germany; Faculty of Medicine and University Hospital Carl Gustav Carus, Technische Universität Dresden, Dresden, Germany; Helmholtz-Zentrum Dresden-Rossendorf (HZDR), Dresden, Germany.

Alfons Meindl (A)

Department of Gynecology and Obstetrics, University of Munich, Campus Großhadern, Munich, Germany.

Rita K Schmutzler (RK)

Faculty of Medicine and University Hospital Cologne, Center for Familial Breast and Ovarian Cancer, University of Cologne, Cologne, Germany.
Faculty of Medicine and University Hospital Cologne, Center for Integrated Oncology, University of Cologne, Cologne, Germany.
Faculty of Medicine and University Hospital Cologne, Center for Molecular Medicine Cologne, University of Cologne, Cologne, Germany.

Paul D P Pharoah (PDP)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.
Department of Oncology, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

Antonis C Antoniou (AC)

Department of Public Health and Primary Care, Centre for Cancer Genetic Epidemiology, University of Cambridge, Cambridge, UK.

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