23andMe, Inc. , Mountain View, CA , USA ; Department of Genetics , Stanford University School of Medicine , Stanford, CA , USA.
PeerJ. 2013 Feb 12;1:e8. doi: 10.7717/peerj.8. Print 2013.
Background. Inherited BRCA gene mutations convey a high risk for breast and ovarian cancer, but current guidelines limit BRCA mutation testing to women with early-onset cancer and relatives of mutation-positive cases. Benefits and risks of providing this information directly to consumers are unknown. Methods. To assess and quantify emotional and behavioral reactions of consumers to their 23andMe Personal Genome Service(®) report of three BRCA mutations that are common in Ashkenazi Jews, we invited all 136 BRCA1 and BRCA2 mutation-positive individuals in the 23andMe customer database who had chosen to view their BRCA reports to participate in this IRB-approved study. We also invited 160 mutation-negative customers who were matched for age, sex and ancestry. Semi-structured phone interviews were completed for 32 mutation carriers, 16 women and 16 men, and 31 non-carriers. Questions addressed personal and family history of cancer, decision and timing of viewing the BRCA report, recollection of the result, emotional responses, perception of personal cancer risk, information sharing, and actions taken or planned. Results. Eleven women and 14 men had received the unexpected result that they are carriers of a BRCA1 185delAG or 5382insC, or BRCA2 6174delT mutation. None of them reported extreme anxiety and four experienced moderate anxiety that was transitory. Remarkably, five women and six men described their response as neutral. Most carrier women sought medical advice and four underwent risk-reducing procedures after confirmatory mutation testing. Male carriers realized that their test results implied genetic risk for female relatives, and several of them felt considerably burdened by this fact. Sharing mutation information with family members led to screening of at least 30 relatives and identification of 13 additional carriers. Non-carriers did not report inappropriate actions, such as foregoing cancer screening. All but one of the 32 mutation-positive participants appreciated learning their BRCA mutation status. Conclusions. Direct access to BRCA mutation tests, considered a model for high-risk actionable genetic tests of proven clinical utility, provided clear benefits to participants. The unexpected information demonstrated a cascade effect as relatives of newly identified carriers also sought testing and more mutation carriers were identified. Given the absence of evidence for serious emotional distress or inappropriate actions in this subset of mutation-positive customers who agreed to be interviewed for this study, broader screening of Ashkenazi Jewish women for these three BRCA mutations should be considered.
遗传性 BRCA 基因突变可显著增加乳腺癌和卵巢癌的发病风险,但目前的指南仅将 BRCA 基因突变检测限制于发病年龄较早的女性和突变阳性病例的亲属。向消费者直接提供该信息的获益和风险尚不清楚。
为评估和量化消费者在获知 23andMe 个人基因组服务(®)报告中发现三种常见于 Ashkenazi 犹太人的 BRCA 基因突变时的情绪和行为反应,我们邀请了 23andMe 客户数据库中所有 136 名已选择查看 BRCA 报告的 BRCA1 和 BRCA2 基因突变阳性个体参加这项经机构审查委员会批准的研究。我们还邀请了 160 名年龄、性别和祖源相匹配的 BRCA 基因突变阴性的对照者。我们对 32 名突变携带者(16 名女性和 16 名男性)和 31 名非携带者进行了半结构式电话访谈。访谈问题涉及癌症的个人和家族史、查看 BRCA 报告的决策和时机、对结果的回忆、情绪反应、对个人癌症风险的感知、信息共享,以及已采取或计划采取的行动。
11 名女性和 14 名男性收到了意料之外的结果,他们是 BRCA1 185delAG 或 5382insC 或 BRCA2 6174delT 基因突变的携带者。他们中没有一人报告有极度焦虑,有 4 人经历了短暂的中度焦虑。值得注意的是,有 5 名女性和 6 名男性将其反应描述为中性。大多数女性携带者寻求了医疗建议,并在经过确认性突变检测后进行了降低风险的手术。男性携带者意识到他们的检测结果暗示其女性亲属存在遗传风险,其中一些人为此深感负担。与家庭成员共享突变信息导致对至少 30 名亲属进行了筛查,并发现了 13 名额外的携带者。非携带者没有报告不当行为,例如放弃癌症筛查。在参加这项研究的突变阳性参与者中,除了 1 人以外,其他人都对了解自己的 BRCA 突变状态表示赞赏。
直接获得 BRCA 基因突变检测结果(被认为是具有明确临床效用的高风险可操作遗传检测的模型)为参与者带来了明确的获益。意料之外的信息产生了级联效应,因为新确定的携带者的亲属也寻求检测,从而发现了更多的携带者。鉴于在同意接受这项研究的访谈的这组突变阳性客户中,没有证据表明存在严重的情绪困扰或不当行为,因此应该考虑对 Ashkenazi 犹太女性进行这三种 BRCA 基因突变的广泛筛查。