TY - JOUR TI - Functional annotation of the 2q35 breast cancer risk locus implicates a structural variant in influencing activity of a long-range enhancer element EP - 1203 AV - public Y1 - 2021/07/01/ KW - breast cancer risk KW - functional annotation KW - risk locus ID - discovery10135823 N2 - A combination of genetic and functional approaches has identified three independent breast cancer risk loci at 2q35. A recent fine-scale mapping analysis to refine these associations resulted in 1 (signal 1), 5 (signal 2), and 42 (signal 3) credible causal variants at these loci. We used publicly available in silico DNase I and ChIP-seq data with in vitro reporter gene and CRISPR assays to annotate signals 2 and 3. We identified putative regulatory elements that enhanced cell-type-specific transcription from the IGFBP5 promoter at both signals (30- to 40-fold increased expression by the putative regulatory element at signal 2, 2- to 3-fold by the putative regulatory element at signal 3). We further identified one of the five credible causal variants at signal 2, a 1.4 kb deletion (esv3594306), as the likely causal variant; the deletion allele of this variant was associated with an average additional increase in IGFBP5 expression of 1.3-fold (MCF-7) and 2.2-fold (T-47D). We propose a model in which the deletion allele of esv3594306 juxtaposes two transcription factor binding regions (annotated by estrogen receptor alpha ChIP-seq peaks) to generate a single extended regulatory element. This regulatory element increases cell-type-specific expression of the tumor suppressor gene IGFBP5 and, thereby, reduces risk of estrogen receptor-positive breast cancer (odds ratio = 0.77, 95% CI 0.74?0.81, p = 3.1 в 10?31). PB - CELL PRESS N1 - 2021 The Authors. This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/) IS - 7 VL - 108 SP - 1190 JF - American Journal of Human Genetics A1 - Baxter, JS A1 - Johnson, N A1 - Tomczyk, K A1 - Gillespie, A A1 - Maguire, S A1 - Brough, R A1 - Fachal, L A1 - Michailidou, K A1 - Bolla, MK A1 - Wang, Q A1 - Dennis, J A1 - Ahearn, TU A1 - Andrulis, IL A1 - Anton-Culver, H A1 - Antonenkova, NN A1 - Arndt, V A1 - Aronson, KJ A1 - Augustinsson, A A1 - Becher, H A1 - Beckmann, MW A1 - Behrens, S A1 - Benitez, J A1 - Bermisheva, M A1 - Bogdanova, N A1 - Bojesen, SE A1 - Brenner, H A1 - Brucker, SY A1 - Cai, Q A1 - Campa, D A1 - Canzian, F A1 - Castelao, JE A1 - Chan, TL A1 - Chang-Claude, J A1 - Chanock, SJ A1 - Chenevix-Trench, G A1 - Choi, J-Y A1 - Clarke, CL A1 - Collaborators, N A1 - Colonna, S A1 - Conroy, DM A1 - Couch, FJ A1 - Cox, A A1 - Cross, SS A1 - Czene, K A1 - Daly, MB A1 - Devilee, P A1 - Doerk, T A1 - Dossus, L A1 - Dwek, M A1 - Eccles, DM A1 - Ekici, AB A1 - Eliassen, AH A1 - Engel, C A1 - Fasching, PA A1 - Figueroa, J A1 - Flyger, H A1 - Gago-Dominguez, M A1 - Gao, C A1 - Garcia-Closas, M A1 - Garcia-Saenz, JA A1 - Ghoussaini, M A1 - Giles, GG A1 - Goldberg, MS A1 - Gonzalez-Neira, A A1 - Guenel, P A1 - Guendert, M A1 - Haeberle, L A1 - Hahnen, E A1 - Haiman, CA A1 - Hall, P A1 - Hamann, U A1 - Hartman, M A1 - Hatse, S A1 - Hauke, J A1 - Hollestelle, A A1 - Hoppe, R A1 - Hopper, JL A1 - Hou, M-F A1 - Ito, H A1 - Iwasaki, M A1 - Jager, A A1 - Jakubowska, A A1 - Janni, W A1 - John, EM A1 - Joseph, V A1 - Jung, A A1 - Kaaks, R A1 - Kang, D A1 - Keeman, R A1 - Khusnutdinova, E A1 - Kim, S-W A1 - Kosma, V-M A1 - Kraft, P A1 - Kristensen, VN A1 - Kubelka-Sabit, K A1 - Kurian, AW A1 - Kwong, A A1 - Lacey, J A1 - Lambrechts, D A1 - Larson, NL A1 - Larsson, SC A1 - Le Marchand, L A1 - Lejbkowicz, F A1 - Li, J A1 - Long, J A1 - Lophatananon, A A1 - LubiNski, J A1 - Mannermaa, A A1 - Manoochehri, M A1 - Manoukian, S A1 - Margolin, S A1 - Matsuo, K A1 - Mavroudis, D A1 - Mayes, R A1 - Menon, U A1 - Milne, RL A1 - Taib, NAM A1 - Muir, K A1 - Muranen, TA A1 - Murphy, RA A1 - Nevanlinna, H A1 - O'Brien, KM A1 - Offit, K A1 - Olson, JE A1 - Olsson, H A1 - Park, SK A1 - Park-Simon, T-W A1 - Patel, A A1 - Peterlongo, P A1 - Peto, J A1 - Plaseska-Karanfilska, D A1 - Presneau, N A1 - Pylkas, K A1 - Rack, B A1 - Rennert, G A1 - Romero, A A1 - Ruebner, M A1 - Ruediger, T A1 - Saloustros, E A1 - Sandler, DP A1 - Sawyer, EJ A1 - Schmidt, MK A1 - Schmutzler, RK A1 - Schneeweiss, A A1 - Schoemaker, MJ A1 - Shah, M A1 - Shen, C-Y A1 - Shu, X-O A1 - Simard, J A1 - Southey, MC A1 - Stone, J A1 - Surowy, H A1 - Swerdlow, AJ A1 - Tamimi, RM A1 - Tapper, WJ A1 - Taylor, JA A1 - Teo, SH A1 - Teras, LR A1 - Terry, MB A1 - Toland, AE A1 - Tomlinson, I A1 - Truong, T A1 - Tseng, C-C A1 - Untch, M A1 - Vachon, CM A1 - van den Ouweland, AMW A1 - Wang, SS A1 - Weinberg, CR A1 - Wendt, C A1 - Winham, SJ A1 - Winqvist, R A1 - Wolk, A A1 - Wu, AH A1 - Yamaji, T A1 - Zheng, W A1 - Ziogas, A A1 - Pharoah, PDP A1 - Dunning, AM A1 - Easton, DF A1 - Pettitt, SJ A1 - Lord, CJ A1 - Haider, S A1 - Orr, N A1 - Fletcher, O SN - 1537-6605 UR - https://doi.org/10.1016/j.ajhg.2021.05.013 ER -