Shindo Koji, Yu Jun, Suenaga Masaya, Fesharakizadeh Shahriar, Cho Christy, Macgregor-Das Anne, Siddiqui Abdulrehman, Witmer P Dane, Tamura Koji, Song Tae Jun, Navarro Almario Jose Alejandro, Brant Aaron, Borges Michael, Ford Madeline, Barkley Thomas, He Jin, Weiss Matthew J, Wolfgang Christopher L, Roberts Nicholas J, Hruban Ralph H, Klein Alison P, Goggins Michael
All authors: The Sol Goldman Pancreatic Cancer Research Center, Johns Hopkins University, Baltimore, MD.
J Clin Oncol. 2017 Oct 20;35(30):3382-3390. doi: 10.1200/JCO.2017.72.3502. Epub 2017 Aug 2.
Purpose Deleterious germline mutations contribute to pancreatic cancer susceptibility and are well documented in families in which multiple members have had pancreatic cancer. Methods To define the prevalence of these germline mutations in patients with apparently sporadic pancreatic cancer, we sequenced 32 genes, including known pancreatic cancer susceptibility genes, in DNA prepared from normal tissue obtained from 854 patients with pancreatic ductal adenocarcinoma, 288 patients with other pancreatic and periampullary neoplasms, and 51 patients with non-neoplastic diseases who underwent pancreatic resection at Johns Hopkins Hospital between 2000 and 2015. Results Thirty-three (3.9%; 95% CI, 3.0% to 5.8%) of 854 patients with pancreatic cancer had a deleterious germline mutation, 31 (3.5%) of which affected known familial pancreatic cancer susceptibility genes: BRCA2 (12 patients), ATM (10 patients), BRCA1 (3 patients), PALB2 (2 patients), MLH1 (2 patients), CDKN2A (1 patient), and TP53 (1 patient). Patients with these germline mutations were younger than those without (mean ± SD, 60.8 ± 10.6 v 65.1 ± 10.5 years; P = .03). Deleterious germline mutations were also found in BUB1B (1) and BUB3 (1). Only three of these 33 patients had reported a family history of pancreatic cancer, and most did not have a cancer family history to suggest an inherited cancer syndrome. Five (1.7%) of 288 patients with other periampullary neoplasms also had a deleterious germline mutation. Conclusion Germline mutations in pancreatic cancer susceptibility genes are commonly identified in patients with pancreatic cancer without a significant family history of cancer. These deleterious pancreatic cancer susceptibility gene mutations, some of which are therapeutically targetable, will be missed if current family history guidelines are the main criteria used to determine the appropriateness of gene testing.
目的 有害的种系突变会导致胰腺癌易感性,并且在有多名成员患胰腺癌的家族中已有充分记录。方法 为了确定这些种系突变在明显散发型胰腺癌患者中的患病率,我们对854例胰腺导管腺癌患者、288例其他胰腺和壶腹周围肿瘤患者以及51例在2000年至2015年间于约翰霍普金斯医院接受胰腺切除术的非肿瘤性疾病患者的正常组织所制备的DNA中的32个基因进行了测序,这些基因包括已知的胰腺癌易感基因。结果 854例胰腺癌患者中有33例(3.9%;95%CI,3.0%至5.8%)发生了有害的种系突变,其中31例(3.5%)影响已知的家族性胰腺癌易感基因:BRCA2(12例患者)、ATM(10例患者)、BRCA1(3例患者)、PALB2(2例患者)、MLH1(2例患者)、CDKN2A(1例患者)和TP53(1例患者)。发生这些种系突变的患者比未发生突变的患者年龄更小(平均±标准差,60.8±10.6岁对65.1±10.5岁;P = 0.03)。在BUB1B(1例)和BUB3(1例)中也发现了有害的种系突变。这33例患者中只有3例报告有胰腺癌家族史,并且大多数没有癌症家族史提示遗传性癌症综合征。288例其他壶腹周围肿瘤患者中有5例(1.7%)也发生了有害的种系突变。结论 在没有明显癌症家族史的胰腺癌患者中,常见胰腺癌易感基因的种系突变。如果以目前的家族史指南作为确定基因检测适用性的主要标准,这些有害的胰腺癌易感基因突变(其中一些是可治疗靶向的)将会被漏检。