Rego Shannon, Dagan-Rosenfeld Orit, Bivona Stephanie A, Snyder Michael P, Ormond Kelly E
Department of Genetics, Stanford University School of Medicine, Stanford, California.
Institute for Human Genetics, University of California-San Francisco, San Francisco, California.
J Genet Couns. 2019 Apr;28(2):428-437. doi: 10.1002/jgc4.1096. Epub 2019 Mar 5.
The increasing availability of exome sequencing to the general ("healthy") population raises questions about the implications of genomic testing for individuals without suspected Mendelian diseases. Little is known about this population's motivations for undergoing exome sequencing, their expectations, reactions, and perceptions of utility. In order to address these questions, we conducted in-depth semi-structured interviews with 12 participants recruited from a longitudinal multi-omics profiling study that included exome sequencing. Participants were interviewed after receiving exome results, which included Mendelian disease-associated pathogenic and likely pathogenic variants, pharmacogenetic variants, and risk assessments for multifactorial diseases such as type 2 diabetes. The primary motivation driving participation in exome sequencing was personal curiosity. While they reported feeling validation and relief, participants were frequently underwhelmed by the results and described having expected more from exome sequencing. All participants reported discussing the results with at least some family, friends, and healthcare providers. Participants' recollection of the results returned to them was sometimes incorrect or incomplete, in many cases aligning with their perceptions of their health risks when entering the study. These results underscore the need for different genetic counseling approaches for generally healthy patients undergoing exome sequencing, in particular the need to provide anticipatory guidance to moderate participants' expectations. They also provide a preview of potential challenges clinicians may face as genomic sequencing continues to scale-up in the general population despite a lack of full understanding of its impact.
外显子组测序在普通(“健康”)人群中的可及性日益提高,这引发了关于基因组检测对无孟德尔疾病疑似个体的影响的问题。对于这一人群进行外显子组测序的动机、他们的期望、反应以及对实用性的认知,我们知之甚少。为了解决这些问题,我们对从一项包括外显子组测序的纵向多组学分析研究中招募的12名参与者进行了深入的半结构化访谈。参与者在收到外显子组结果后接受了访谈,这些结果包括与孟德尔疾病相关的致病和可能致病变异、药物遗传学变异以及2型糖尿病等多因素疾病的风险评估。推动参与外显子组测序的主要动机是个人好奇心。虽然他们报告感到得到了验证和宽慰,但参与者对结果常常感到失望,并表示对外显子组测序期望更高。所有参与者都报告至少与一些家人、朋友和医疗服务提供者讨论了结果。参与者对结果的回忆有时不正确或不完整,在许多情况下与他们进入研究时对自身健康风险的认知一致。这些结果强调,对于进行外显子组测序的一般健康患者,需要采用不同的遗传咨询方法,特别是需要提供预期指导以适度调整参与者的期望。它们还预示了尽管对基因组测序在普通人群中的影响缺乏全面了解,但随着其在普通人群中继续扩大规模,临床医生可能面临的潜在挑战。