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一种适用于资源受限环境中罕见遗传疾病的可行分子诊断策略。

A feasible molecular diagnostic strategy for rare genetic disorders within resource-constrained environments.

作者信息

Mudau Maria Mabyalwa, Seymour Heather, Nevondwe Patracia, Kerr Robyn, Spencer Careni, Feben Candice, Lombard Zané, Honey Engela, Krause Amanda, Carstens Nadia

机构信息

Division of Human Genetics, National Health Laboratory Service and School of Pathology, Faculty of Health Sciences, University of the Witwatersrand, Johannesburg, South Africa.

Division of Human Genetics, Faculty of Health Sciences, University of Cape Town, Cape Town, South Africa.

出版信息

J Community Genet. 2024 Feb;15(1):39-48. doi: 10.1007/s12687-023-00674-8. Epub 2023 Oct 10.

Abstract

Timely and accurate diagnosis of rare genetic disorders is critical, as it enables improved patient management and prognosis. In a resource-constrained environment such as the South African State healthcare system, the challenge is to design appropriate and cost-effective assays that will enable accurate genetic diagnostic services in patients of African ancestry across a broad disease spectrum. Next-generation sequencing (NGS) has transformed testing approaches for many Mendelian disorders, but this technology is still relatively new in our setting and requires cost-effective ways to implement. As a proof of concept, we describe a feasible diagnostic strategy for genetic disorders frequently seen in our genetics clinics (RASopathies, Cornelia de Lange syndrome, Treacher Collins syndrome, and CHARGE syndrome). The custom-designed targeted NGS gene panel enabled concurrent variant screening for these disorders. Samples were batched during sequencing and analyzed selectively based on the clinical phenotype. The strategy employed in the current study was cost-effective, with sequencing and analysis done at USD849.68 per sample and achieving an overall detection rate of 54.5%. The strategy employed is cost-effective as it allows batching of samples from patients with different diseases in a single run, an approach that can be utilized with rare and less frequently ordered molecular diagnostic tests. The subsequent selective analysis pipeline allowed for timeous reporting back of patients results. This is feasible with a reasonable yield and can be employed for the molecular diagnosis of a wide range of rare monogenic disorders in a resource-constrained environment.

摘要

及时准确地诊断罕见遗传病至关重要,因为这有助于改善患者管理和预后。在南非国家医疗系统这样资源有限的环境中,挑战在于设计合适且具有成本效益的检测方法,以便能为广大疾病谱的非洲裔患者提供准确的基因诊断服务。下一代测序(NGS)已经改变了许多孟德尔疾病的检测方法,但这项技术在我们的环境中仍相对较新,需要以具有成本效益的方式来实施。作为概念验证,我们描述了一种在我们的遗传学诊所中常见的遗传病(RASopathies综合征、科妮莉亚·德·朗格综合征、特雷彻·柯林斯综合征和CHARGE综合征)的可行诊断策略。定制设计的靶向NGS基因 panel 能够同时对这些疾病进行变异筛查。样本在测序过程中进行分批,并根据临床表型进行选择性分析。本研究采用的策略具有成本效益,测序和分析每个样本的费用为849.68美元,总体检测率达到54.5%。所采用的策略具有成本效益,因为它允许在一次运行中对来自不同疾病患者的样本进行分批,这种方法可用于罕见和较少订购的分子诊断测试。随后的选择性分析流程能够及时反馈患者结果。这在有合理产出的情况下是可行的,并且可用于在资源有限的环境中对广泛的罕见单基因疾病进行分子诊断。

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