Zheng Guoqiao, Catalano Calogerina, Bandapalli Obul Reddy, Paramasivam Nagarajan, Chattopadhyay Subhayan, Schlesner Matthias, Sijmons Rolf, Hemminki Akseli, Dymerska Dagmara, Lubinski Jan, Hemminki Kari, Försti Asta
Division of Molecular Genetic Epidemiology, German Cancer Research Center (DKFZ), Im Neuenheimer Feld 580, D-69120 Heidelberg, Germany.
Department of Internal Medicine V, University of Heidelberg, 69120 Heidelberg, Germany.
Cancers (Basel). 2020 Sep 27;12(10):2770. doi: 10.3390/cancers12102770.
Familial clustering, twin concordance, and identification of high- and low-penetrance cancer predisposition variants support the idea that there are families that are at a high to moderate excess risk of cancer. To what extent there may be families that are protected from cancer is unknown. We wanted to test genetically whether cancer-free families share fewer breast, colorectal, and prostate cancer risk alleles than the population at large. We addressed this question by whole-genome sequencing (WGS) of 51 elderly cancer-free individuals whose numerous (ca. 1000) family members were found to be cancer-free ('cancer-free families', CFFs) based on face-to-face interviews. The average coverage of the 51 samples in the WGS was 42x. We compared cancer risk allele frequencies in cancer-free individuals with those in the general population available in public databases. The CFF members had fewer loss-of-function variants in suggested cancer predisposition genes compared to the ExAC data, and for high-risk cancer predisposition genes, no pathogenic variants were found in CFFs. For common low-penetrance breast, colorectal, and prostate cancer risk alleles, the results were not conclusive. The results suggest that, in line with twin and family studies, random environmental causes are so dominant that a clear demarcation of cancer-free populations using genetic data may not be feasible.
家族聚集性、双胞胎一致性以及高、低外显率癌症易感性变异的鉴定支持这样一种观点,即存在一些患癌风险处于中高度增加水平的家族。尚不清楚在何种程度上可能存在对癌症具有抗性的家族。我们想从基因层面测试无癌家族携带的乳腺癌、结直肠癌和前列腺癌风险等位基因是否比普通人群更少。我们通过对51名无癌老年人进行全基因组测序(WGS)来解决这个问题,这些老年人众多(约1000名)的家庭成员经面对面访谈后被发现均无癌症(“无癌家族”,CFF)。WGS中这51个样本的平均覆盖度为42倍。我们将无癌个体中的癌症风险等位基因频率与公共数据库中普通人群的频率进行了比较。与ExAC数据相比,CFF成员在提示的癌症易感基因中功能缺失变异较少,并且对于高风险癌症易感基因,在CFF中未发现致病变异。对于常见的低外显率乳腺癌、结直肠癌和前列腺癌风险等位基因,结果尚无定论。结果表明,与双胞胎和家族研究一致,随机环境因素占主导地位,以至于利用基因数据明确划分无癌人群可能不可行。