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一项关于医疗服务提供者对儿科临床实践中全基因组检测影响的观点的定性研究。

A qualitative study of healthcare providers' perspectives on the implications of genome-wide testing in pediatric clinical practice.

作者信息

Reiff Marian, Mueller Rebecca, Mulchandani Surabhi, Spinner Nancy B, Pyeritz Reed E, Bernhardt Barbara A

机构信息

Center for the Integration of Genetic Health Care Technologies, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA,

出版信息

J Genet Couns. 2014 Aug;23(4):474-88. doi: 10.1007/s10897-013-9653-8. Epub 2013 Sep 14.

Abstract

The utilization of genome-wide chromosomal microarray analysis (CMA) in pediatric clinical practice provides an opportunity to consider how genetic diagnostics is evolving, and to prepare for the clinical integration of genome-wide sequencing technologies. We conducted semi-structured interviews with 15 healthcare providers (7 genetic counselors, 4 medical geneticists, and 4 non-genetics providers) to investigate the impact of CMA on clinical practice, and implications for providers, patients and families. Interviews were analyzed qualitatively using content analysis. Most providers reported that genomic testing enhanced their professional experience and was beneficial to patients, primarily due to the improved diagnostic rate compared with earlier chromosomal studies. Other effects on practice included moving towards genotype-first diagnosis and broadening indications for chromosomal testing. Opinions varied concerning informed consent and disclosure of results. The duty to disclose incidental findings (IFs) was noted; however concerns were raised about potential psychosocial harms of disclosing pre-symptomatic findings. Tensions were revealed between the need for comprehensive informed consent for all families and the challenges of communicating time-consuming and potentially anxiety-provoking information regarding uncertain and incidental findings that may be relevant only in rare cases. Genetic counselors can play an important role in liaising with families, health professionals and testing laboratories, providing education and guidance to non-genetics providers, and enabling families to receive adequate pre-and post-test information and follow-up care.

摘要

全基因组染色体微阵列分析(CMA)在儿科临床实践中的应用为思考基因诊断如何发展以及为全基因组测序技术的临床整合做准备提供了契机。我们对15名医疗服务提供者(7名遗传咨询师、4名医学遗传学家和4名非遗传学提供者)进行了半结构化访谈,以调查CMA对临床实践的影响以及对提供者、患者和家庭的意义。采用内容分析法对访谈进行定性分析。大多数提供者报告称,基因检测丰富了他们的专业经验,对患者有益,这主要是因为与早期染色体研究相比诊断率有所提高。对实践的其他影响包括向基因型优先诊断转变以及扩大染色体检测的适应症。对于知情同意和结果披露,意见不一。提到了披露偶发发现(IFs)的责任;然而,对于披露症状前发现可能带来的潜在心理社会危害也存在担忧。在需要为所有家庭提供全面知情同意与传达关于不确定和偶发发现(可能仅在罕见情况下相关)的耗时且可能引发焦虑的信息所面临的挑战之间,出现了矛盾。遗传咨询师在与家庭、卫生专业人员和检测实验室联络、为非遗传学提供者提供教育和指导以及使家庭能够获得充分的检测前和检测后信息及后续护理方面可以发挥重要作用。

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