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对首批有亨廷顿病风险的症状前检测个体进行基因检测及结果反馈的观点

Perspectives on Genetic Testing and Return of Results from the First Cohort of Presymptomatically Tested Individuals At Risk of Huntington Disease.

作者信息

Stuttgen K M, Bollinger J M, Dvoskin R L, McCague A, Shpritz B, Brandt J, Mathews Debra J H

机构信息

Berman Institute of Bioethics, Johns Hopkins University, 1809 Ashland Avenue, Room 211, Baltimore, MD, 21205, USA.

Institute of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, USA.

出版信息

J Genet Couns. 2018 Dec;27(6):1428-1437. doi: 10.1007/s10897-018-0274-0. Epub 2018 Jul 2.

Abstract

This qualitative study gathered opinions about genetic testing from people who received presymptomatic testing for Huntington's disease (HD) 20-30 years ago and have lived with the implications of that testing for decades. During the last section of a semi-structured interview, participants were asked open-ended questions about their opinions on the importance of autonomy in the decision to be tested for HD, whether a formal HD testing protocol is necessary, whether physician ordering for HD is acceptable without a formal protocol, whether online direct-to-consumer (DTC) genetic testing for HD is acceptable, and whether incidental/secondary findings should be returned in the context of whole exome/genome sequencing. Most-but not all-participants were in favor of an individual's right to decide whether and when to pursue HD testing, use of a formal HD testing protocol, and returning medically actionable secondary findings. However, the majority of participants were opposed not only to physician ordering and DTC HD testing in the absence of a formal protocol but also to returning a secondary finding of an expanded HD allele. This study presents the opinions of a unique and extremely well-informed cohort on issues that need to be taken into careful consideration by genetic counselors and other medical professionals who are developing genetic testing protocols, making decisions about the availability of genetic tests, and making decisions about whether and how to return incidental findings.

摘要

这项定性研究收集了20至30年前接受亨廷顿舞蹈症(HD)症状前检测、且在数十年间一直受该检测影响的人群对基因检测的看法。在半结构化访谈的最后一部分,参与者被问及一些开放式问题,内容涉及他们对HD检测决策中自主权重要性的看法、是否需要正式的HD检测方案、在没有正式方案的情况下医生开具HD检测是否可接受、是否接受针对HD的在线直接面向消费者(DTC)基因检测,以及在全外显子组/基因组测序的背景下是否应返回偶然/次要发现。大多数(但并非全部)参与者支持个人有权决定是否以及何时进行HD检测、使用正式的HD检测方案,以及返回具有医学可操作性的次要发现。然而,大多数参与者不仅反对在没有正式方案的情况下医生开具检测和DTC HD检测,还反对返回HD等位基因扩展的次要发现。这项研究呈现了一个独特且信息极为丰富的队列对一些问题的看法,这些问题需要遗传咨询师和其他医学专业人员在制定基因检测方案、决定基因检测的可及性,以及决定是否和如何返回偶然发现时予以仔细考虑。

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Recommendations for the predictive genetic test in Huntington's disease.亨廷顿舞蹈病预测性基因检测的建议。
Clin Genet. 2013 Mar;83(3):221-31. doi: 10.1111/j.1399-0004.2012.01900.x. Epub 2012 Jul 30.

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