Human Cancer Genomic Research, Research Center, King Faisal Specialist Hospital and Research Center, 11211, Riyadh, Saudi Arabia.
Department of Pathology and Laboratory Medicine, King Faisal Specialist Hospital and Research Center, 11211, Riyadh, Saudi Arabia.
Sci Rep. 2023 Nov 27;13(1):20924. doi: 10.1038/s41598-023-48231-0.
Breast cancer (BC) is the most prevalent malignancy among women worldwide with germline pathogenic variants/likely pathogenic variants (PVs/LPVs) in BRCA1/2 accounting for a large portion of hereditary cases. Recently, heterozygous PVs/LPVs in the ATM serine/threonine kinase or Ataxia-telangiectasia mutated gene (ATM) has been identified as a moderate susceptibility factor for BC in diverse ethnicities. However, the prevalence of ATM PVs/LPVs in BC susceptibility in Arab populations remains largely unexplored. This study investigated the prevalence of ATM PVs/LPVs among BC patients from Saudi Arabia, employing capture-sequencing technology for ATM PVs/LPVs screening in a cohort of 715 unselected BC patients without BRCA1/2 PVs/LPVs. In addition, founder mutation analysis was conducted using the PHASE program. In our entire cohort, four unique PVs/LPVs in the ATM gene were identified in six cases (0.8%). Notably, one recurrent LPV, c.6115G > A:p.Glu2039Lys was identified in three cases, for which haplotype analysis confirmed as a novel putative founder mutation traced back to 13 generations on average. This founder mutation accounted for half of all identified mutant cases and 0.4% of total screened cases. This study further reveals a significant correlation between the presence of ATM mutation and family history of BC (p = 0.0127). These findings underscore an approximate 0.8% prevalence of ATM germline PVs/LPVs in Arab BC patients without BRCA1/2 PVs/LPVs and suggest a founder effect of specific recurrent ATM mutation. These insights can help in the design of a genetic testing strategy tailored to the local population in Saudi Arabia, thereby, enabling more accurate clinical management and risk prediction.
乳腺癌(BC)是全球女性中最常见的恶性肿瘤,BRCA1/2 种系致病性变异/可能致病性变异(PV/LPV)占遗传性病例的很大一部分。最近,在不同种族中,ATM 丝氨酸/苏氨酸激酶或共济失调毛细血管扩张突变基因(ATM)的杂合性 PV/LPV 已被确定为 BC 的中度易感性因素。然而,ATM PV/LPV 在阿拉伯人群中的 BC 易感性中的患病率在很大程度上仍未得到探索。本研究采用捕获测序技术对 715 例无 BRCA1/2 PV/LPV 的未选择 BC 患者进行 ATM PV/LPV 筛查,调查了沙特阿拉伯 BC 患者中 ATM PV/LPV 的患病率。此外,还使用 PHASE 程序进行了创始人突变分析。在我们的整个队列中,在 6 例(0.8%)患者中发现了 ATM 基因中的 4 个独特的 PV/LPV。值得注意的是,在 3 例患者中发现了一个反复出现的 LPV,c.6115G>A:p.Glu2039Lys,通过单体型分析证实其为一个新的假定创始人突变,可追溯到平均 13 代。该创始人突变占所有鉴定的突变病例的一半和总筛选病例的 0.4%。本研究进一步揭示了 ATM 突变的存在与 BC 家族史之间存在显著相关性(p=0.0127)。这些发现表明,在无 BRCA1/2 PV/LPV 的阿拉伯 BC 患者中,ATM 种系 PV/LPV 的患病率约为 0.8%,并提示存在特定的 ATM 突变的创始人效应。这些发现有助于制定适合沙特阿拉伯当地人群的基因检测策略,从而实现更准确的临床管理和风险预测。