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扩大儿科全基因组测序的应用:来自SeqFirst供应商的见解,以促进公平获得精确的基因诊断。

Expanding implementation of pediatric whole-genome sequencing: Insights from SeqFirst providers to inform equitable access to a precise genetic diagnosis.

作者信息

Yu Joon-Ho, MacDuffie Katherine E, Sommerland Olivia, Theoryn Tesla, Murali Priyanka, Anderson Kailyn, Sikes Megan, Kruidenier Lukas, Gildersleeve Heidi I S, Scott Abbey, Buckingham Kati J, McWalter Kirsty, Kruszka Paul, Keefe Alexandra C, Chong Jessica X, Veenstra David L, Dipple Katrina M, Wenger Tara, Doherty Dan, Bamshad Michael J

机构信息

University of Washington, Department of Pediatrics, Division of Genetic Medicine, Seattle, WA, USA; University of Washington, Institute for Public Health Genetics, Seattle, WA USA; Department of Pediatrics, Division of Bioethics and Palliative Care, University of Washington, Seattle, WA, USA; Seattle Children's Hospital and Research Institute, Seattle, WA, USA.

Department of Pediatrics, Division of Bioethics and Palliative Care, University of Washington, Seattle, WA, USA; Seattle Children's Hospital and Research Institute, Seattle, WA, USA.

出版信息

HGG Adv. 2025 Jun 3;6(4):100464. doi: 10.1016/j.xhgg.2025.100464.

Abstract

Whole-genome sequencing (WGS) as a diagnostic test offers children suspected of having a rare genetic condition and their families the best direct path toward securing a precise genetic diagnosis (PrGD). Yet, a limited supply and inequitable access to genetic services are impediments to realizing the benefits of a PrGD. Such access disparities might be due to a range of structural and social determinants that manifest in interactions, or the lack thereof, between families, providers, and institutions. Semi-structured key informant interviews (n = 19) were conducted with neonatologists and neurodevelopmental clinic providers (NDV providers) who referred families to the SeqFirst study to identify barriers and inform strategies to improve equitable access to a PrGD via WGS. Overall, neonatologists and NDV providers were enthusiastic about offering WGS to their patients and families despite different contexts of medical care. Providers cited several considerations that influenced their introduction of WGS and genetic testing to families, including their perceptions of families' capacity, readiness, and distrust and the establishment of sufficient provider-family rapport. These considerations influenced providers' timing and introduction of genetic testing and WGS to families. Together, these findings suggest that providers' perceptions of families may result in delayed introduction of WGS.‬ Despite enthusiasm for early WGS across medical subspecialties, providers' perceptions of families and their social contexts highlight both challenges and opportunities in the implementation of WGS to promote and maximize equitable access.

摘要

全基因组测序(WGS)作为一种诊断测试,为疑似患有罕见遗传病的儿童及其家庭提供了获得精确基因诊断(PrGD)的最佳直接途径。然而,基因服务供应有限且获取不均,阻碍了人们实现精确基因诊断的益处。这种获取差异可能是由于一系列结构和社会决定因素造成的,这些因素体现在家庭、医疗服务提供者和机构之间的互动(或缺乏互动)中。我们对新生儿科医生和神经发育诊所的医疗服务提供者(NDV提供者)进行了半结构化关键信息人访谈(n = 19),这些人曾将家庭转介到SeqFirst研究中,以确定障碍并为改善通过WGS实现公平获取精确基因诊断的策略提供信息。总体而言,尽管医疗护理背景不同,新生儿科医生和NDV提供者仍热衷于为患者及其家庭提供WGS。医疗服务提供者列举了一些影响他们向家庭介绍WGS和基因检测的因素,包括他们对家庭能力、意愿和不信任的看法,以及建立足够的医患融洽关系。这些因素影响了医疗服务提供者向家庭介绍基因检测和WGS的时机。这些发现共同表明,医疗服务提供者对家庭的看法可能导致WGS的引入延迟。尽管各医学亚专业对早期WGS充满热情,但医疗服务提供者对家庭及其社会背景的看法凸显了在实施WGS以促进和最大化公平获取方面的挑战和机遇。

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