Department of Pathology, The Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
The Sol Goldman Pancreatic Cancer Research Center, The Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
Genes Chromosomes Cancer. 2021 Aug;60(8):559-564. doi: 10.1002/gcc.22947. Epub 2021 Apr 1.
Pancreatic ductal adenocarcinoma (PDAC) is a lethal disease with a 5-year survival rate of less than 10%. Individuals with a pathogenic germline variant in a pancreatic cancer susceptibility gene are at an increased risk of developing pancreatic cancer. Understanding the inherited genetic basis of pancreatic tumor development provides a unique opportunity to improve patient care and outcomes. For example, relatives of a patients with PDAC who have a pathogenic germline variant in a pancreatic cancer susceptibility gene are eligible for disease surveillance where cancers may be detected early, and 5-year survival greatly improved. Furthermore, for some patients with PDAC and a pathogenic germline variant in a pancreatic cancer susceptibility gene, their tumors may be susceptible to specific anti-cancer therapies. Recently, RABL3 was identified as a pancreatic cancer susceptibility gene. To validate these findings and inform clinical translation, we determined the prevalence of deleterious RABL3 variants in a large cohort of 1037 patients with PDAC that had undergone either whole genome or whole exome germline sequencing. We identified two synonymous variants and four missense variants classified as variants of unknown significance. We found no pathogenic RABL3 variants, indicating that the maximum prevalence of such variants in patients with PDAC is less than 0.36% (minor allele frequency 0, 97.5% one-sided confidence interval: 0-0.0036). This finding has important implications for germline genetic testing of patients with PDAC.
胰腺导管腺癌 (PDAC) 是一种致命疾病,其 5 年生存率低于 10%。具有胰腺癌易感性基因种系致病性变异的个体患胰腺癌的风险增加。了解胰腺肿瘤发生的遗传基础为改善患者的治疗和预后提供了独特的机会。例如,PDAC 患者的亲属如果有一种胰腺癌易感性基因的种系致病性变异,则有资格进行疾病监测,在这种监测中可能早期发现癌症,5 年生存率大大提高。此外,对于一些具有 PDAC 和胰腺癌易感性基因种系致病性变异的患者,他们的肿瘤可能对特定的抗癌治疗敏感。最近,RABL3 被确定为一种胰腺癌易感性基因。为了验证这些发现并为临床转化提供信息,我们在一个由 1037 名接受全基因组或全外显子种系测序的 PDAC 患者的大型队列中确定了有害 RABL3 变异的患病率。我们发现了两个同义变异和四个错义变异,被归类为意义不明的变异。我们没有发现致病性 RABL3 变异,这表明 PDAC 患者中此类变异的最大患病率低于 0.36%(次要等位基因频率 0,97.5%单侧置信区间:0-0.0036)。这一发现对 PDAC 患者的种系基因检测具有重要意义。