TY - JOUR AU - Dumont, Martine AU - Weber-Lassalle, Nana AU - Joly-Beauparlant, Charles AU - Ernst, Corinna AU - Droit, Arnaud AU - Feng, Bing-Jian AU - Dubois, Stéphane AU - Collin-Deschesnes, Annie-Claude AU - Soucy, Penny AU - Vallée, Maxime AU - Fournier, Frédéric AU - Lemaçon, Audrey AU - Adank, Muriel A AU - Allen, Jamie AU - Altmüller, Janine AU - Arnold, Norbert AU - Ausems, Margreet G E M AU - Berutti, Riccardo AU - Bolla, Manjeet K AU - Bull, Shelley AU - Carvalho, Sara AU - Cornelissen, Sten AU - Dufault, Michael R AU - Dunning, Alison M AU - Engel, Christoph AU - Gehrig, Andrea AU - Geurts-Giele, Willemina R R AU - Gieger, Christian AU - Green, Jessica AU - Hackmann, Karl AU - Helmy, Mohamed AU - Hentschel, Julia AU - Hogervorst, Frans B L AU - Hollestelle, Antoinette AU - Hooning, Maartje J AU - Horváth, Judit AU - Ikram, M Arfan AU - Kaulfuß, Silke AU - Keeman, Renske AU - Kuang, Da AU - Luccarini, Craig AU - Maier, Wolfgang AU - Martens, John W M AU - Niederacher, Dieter AU - Nürnberg, Peter AU - Ott, Claus-Eric AU - Peters, Annette AU - Pharoah, Paul D P AU - Ramirez, Alfredo AU - Ramser, Juliane AU - Riedel-Heller, Steffi AU - Schmidt, Gunnar AU - Shah, Mitul AU - Scherer, Martin AU - Stäbler, Antje AU - Strom, Tim M AU - Sutter, Christian AU - Thiele, Holger AU - van Asperen, Christi J AU - van der Kolk, Lizet AU - van der Luijt, Rob B AU - Volk, Alexander E AU - Wagner, Michael AU - Waisfisz, Quinten AU - Wang, Qing AU - Wang-Gohrke, Shan AU - Weber, Bernhard H F AU - Genome Of The Netherlands Project AU - Ghs Study Group AU - Devilee, Peter AU - Tavtigian, Sean AU - Bader, Gary D AU - Meindl, Alfons AU - Goldgar, David E AU - Andrulis, Irene L AU - Schmutzler, Rita K AU - Easton, Douglas F AU - Schmidt, Marjanka K AU - Hahnen, Eric AU - Simard, Jacques TI - Uncovering the Contribution of Moderate-Penetrance Susceptibility Genes to Breast Cancer by Whole-Exome Sequencing and Targeted Enrichment Sequencing of Candidate Genes in Women of European Ancestry. JO - Cancers VL - 14 IS - 14 SN - 2072-6694 CY - Basel PB - MDPI M1 - DZNE-2022-01381 SP - 3363 PY - 2022 N1 - CC BY AB - Rare variants in at least 10 genes, including BRCA1, BRCA2, PALB2, ATM, and CHEK2, are associated with increased risk of breast cancer; however, these variants, in combination with common variants identified through genome-wide association studies, explain only a fraction of the familial aggregation of the disease. To identify further susceptibility genes, we performed a two-stage whole-exome sequencing study. In the discovery stage, samples from 1528 breast cancer cases enriched for breast cancer susceptibility and 3733 geographically matched unaffected controls were sequenced. Using five different filtering and gene prioritization strategies, 198 genes were selected for further validation. These genes, and a panel of 32 known or suspected breast cancer susceptibility genes, were assessed in a validation set of 6211 cases and 6019 controls for their association with risk of breast cancer overall, and by estrogen receptor (ER) disease subtypes, using gene burden tests applied to loss-of-function and rare missense variants. Twenty genes showed nominal evidence of association (p-value < 0.05) with either overall or subtype-specific breast cancer. Our study had the statistical power to detect susceptibility genes with effect sizes similar to ATM, CHEK2, and PALB2, however, it was underpowered to identify genes in which susceptibility variants are rarer or confer smaller effect sizes. Larger sample sizes would be required in order to identify such genes. KW - breast cancer (Other) KW - genetic susceptibility (Other) KW - moderate-penetrance genes (Other) KW - whole-exome sequencing (Other) LB - PUB:(DE-HGF)16 C6 - pmid:35884425 C2 - pmc:PMC9317824 DO - DOI:10.3390/cancers14143363 UR - https://pub.dzne.de/record/164977 ER -