在华盛顿州推进新生儿筛查:一种新的多路LC-MS/MS蛋白质组学检测威尔逊病和先天性免疫错误。

IF 4 Q1 GENETICS & HEREDITY
Claire Klippel, Jiwoon Park, Sean Sandin, Tara M L Winstone, Xue Chen, Dennis Orton, Aranjeet Singh, Jonathan D Hill, Tareq K Shahbal, Emily Hamacher, Brandon Officer, John Thompson, Phi Duong, Tim Grotzer, Si Houn Hahn
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引用次数: 0

摘要

对于许多遗传疾病,没有特定的代谢生物标志物,也没有适合新生儿群体筛查的分析方法,即使有非常有效的先发制人的治疗方法。直接测量特征肽作为干血斑(DBSs)中蛋白质的替代标记物,已被证明可以成功识别威尔逊病(WD)和三种危及生命的先天性免疫缺陷患者,即x -连锁无球蛋白血症(XLA)、Wiskott-Aldrich综合征(WAS)和腺苷脱氨酶缺乏症(ADAD)。利用高通量LC-MS/MS开发并验证了一种新的基于蛋白质组学的多重检测方法,用于从DBS中检测这四种情况。临床验证结果表明,该方法可以准确地从对照组中识别出目标疾病患者。此外,从2022年到2024年,华盛顿州卫生部新生儿筛查实验室对30024例新生儿DBS样本进行了筛查。发现1例真实推定阳性WD病例,同时发现3例假阳性病例。检测到5例WAS假阳性,但他们都是早产儿和/或低出生体重婴儿,其中4例没有足够的DNA进行确认。该初步研究证明了利用这种多重蛋白质组分析进行新生儿筛查的可行性和有效性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity.

Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity.

Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity.

Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity.

For many genetic disorders, there are no specific metabolic biomarkers nor analytical methods suitable for newborn population screening, even where highly effective preemptive treatments are available. The direct measurement of signature peptides as a surrogate marker for the protein in dried blood spots (DBSs) has been shown to successfully identify patients with Wilson Disease (WD) and three life-threatening inborn errors of immunity, X-linked agammaglobulinemia (XLA), Wiskott-Aldrich syndrome (WAS), and adenosine deaminase deficiency (ADAD). A novel proteomic-based multiplex assay to detect these four conditions from DBS using high-throughput LC-MS/MS was developed and validated. The clinical validation results showed that the assay can accurately identify patients of targeted disorders from controls. Additionally, 30,024 newborn DBS samples from the Washington State Department of Health Newborn Screening Laboratory have been screened from 2022 to 2024. One true presumptive positive case of WD was found along with three false positive cases. Five false positives for WAS were detected, but all of them were premature and/or low-birth-weight babies and four of them had insufficient DNA for confirmation. The pilot study demonstrates the feasibility and effectiveness of utilizing this multiplexed proteomic assay for newborn screening.

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来源期刊
International Journal of Neonatal Screening
International Journal of Neonatal Screening Medicine-Pediatrics, Perinatology and Child Health
CiteScore
6.70
自引率
20.00%
发文量
56
审稿时长
11 weeks
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