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...

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Main Authors: 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
Format: Article
Language:English
Published: MDPI AG 2025-01-01
Series:International Journal of Neonatal Screening
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Online Access:https://www.mdpi.com/2409-515X/11/1/6
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author 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
author_facet 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
author_sort Claire Klippel
collection DOAJ
description 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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series International Journal of Neonatal Screening
spelling doaj-art-453df06b9cf04ced93c08ff8ac0dd0552025-08-20T01:48:46ZengMDPI AGInternational Journal of Neonatal Screening2409-515X2025-01-01111610.3390/ijns11010006Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of ImmunityClaire Klippel0Jiwoon Park1Sean Sandin2Tara M. L. Winstone3Xue Chen4Dennis Orton5Aranjeet Singh6Jonathan D. Hill7Tareq K. Shahbal8Emily Hamacher9Brandon Officer10John Thompson11Phi Duong12Tim Grotzer13Si Houn Hahn14Key Proteo, Inc., Seattle, WA 98122, USAKey Proteo, Inc., Seattle, WA 98122, USAKey Proteo, Inc., Seattle, WA 98122, USAAlberta Precision Laboratories, Calgary, AB T2L 2K8, CanadaAlberta Precision Laboratories, Calgary, AB T2L 2K8, CanadaAlberta Precision Laboratories, Calgary, AB T2L 2K8, CanadaWashington State Department of Health Newborn Screening Laboratories, Seattle, WA 98155, USAWashington State Department of Health Newborn Screening Laboratories, Seattle, WA 98155, USAWashington State Department of Health Newborn Screening Laboratories, Seattle, WA 98155, USAWashington State Department of Health Newborn Screening Laboratories, Seattle, WA 98155, USAWashington State Department of Health Newborn Screening Laboratories, Seattle, WA 98155, USAWashington State Department of Health Newborn Screening Laboratories, Seattle, WA 98155, USASeattle Children’s Research Institute, Seattle, WA 98105, USASeattle Children’s Research Institute, Seattle, WA 98105, USAKey Proteo, Inc., Seattle, WA 98122, USAFor 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.https://www.mdpi.com/2409-515X/11/1/6Wilson DiseaseWiskott–Aldrich syndromeX-linked agammaglobulinemiaadenosine deaminase deficiencynewborn screeningproteomics
spellingShingle 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
Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity
International Journal of Neonatal Screening
Wilson Disease
Wiskott–Aldrich syndrome
X-linked agammaglobulinemia
adenosine deaminase deficiency
newborn screening
proteomics
title Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity
title_full Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity
title_fullStr Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity
title_full_unstemmed Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity
title_short Advancing Newborn Screening in Washington State: A Novel Multiplexed LC-MS/MS Proteomic Assay for Wilson Disease and Inborn Errors of Immunity
title_sort advancing newborn screening in washington state a novel multiplexed lc ms ms proteomic assay for wilson disease and inborn errors of immunity
topic Wilson Disease
Wiskott–Aldrich syndrome
X-linked agammaglobulinemia
adenosine deaminase deficiency
newborn screening
proteomics
url https://www.mdpi.com/2409-515X/11/1/6
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