Literature DB >> 25217881

Newborn screening for severe T and B cell lymphopenia identifies a fraction of patients with Wiskott-Aldrich syndrome.

Stephan Borte1, Anders Fasth2, Ulrika von Döbeln3, Jacek Winiarski4, Lennart Hammarström5.   

Abstract

The lack or marked reduction of recently formed T and B cells provides a basis for neonatal screening for severe combined immunodeficiencies (SCID) and X-linked agammaglobulinemia (XLA). Newborns with other conditions are also identified if a severe T or B cell lymphopenia is present at birth. We retrospectively analyzed Guthrie card samples from 11 children with Wiskott-Aldrich syndrome (WAS), a rare disease that requires early diagnosis and treatment, to determine whether combined T-cell receptor excision circle (TREC) and kappa-deleting recombination excision circle (KREC) screening could identify these patients. 4 of 11 patients showed markedly reduced TREC or KREC copy numbers in their DBS as compared to storage-time matched controls and prospectively screened Swedish and German newborns. No correlation was observed between the WAS gene mutations, the clinical severity/course and the result of the screening assay. A diagnosis of WAS should thus be considered in newborns with positive TREC or KREC screening results.
Copyright © 2014 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Kappa-deleting recombination excision circles;; Newborn screening;; Severe combined immunodeficiency; T cell receptor excision circles;; Wiskott–Aldrich syndrome;

Mesh:

Substances:

Year:  2014        PMID: 25217881     DOI: 10.1016/j.clim.2014.09.003

Source DB:  PubMed          Journal:  Clin Immunol        ISSN: 1521-6616            Impact factor:   3.969


  4 in total

1.  Primary immunodeficiencies screening: neonatal screening for T/B cell disorders - a triplex PCR method for quantitation of TRECs and KRECs in newborns.

Authors:  L Hammarström
Journal:  Clin Exp Immunol       Date:  2014-12       Impact factor: 4.330

Review 2.  Clinical Manifestations and Pathophysiological Mechanisms of the Wiskott-Aldrich Syndrome.

Authors:  Fabio Candotti
Journal:  J Clin Immunol       Date:  2017-10-30       Impact factor: 8.542

Review 3.  Clinical Features, Cancer Biology, Transplant Approach and Other Integrated Management Strategies for Wiskott-Aldrich Syndrome.

Authors:  Smitha Hosahalli Vasanna; Maria A Pereda; Jignesh Dalal
Journal:  J Multidiscip Healthc       Date:  2021-12-23

4.  Second-Tier Next Generation Sequencing Integrated in Nationwide Newborn Screening Provides Rapid Molecular Diagnostics of Severe Combined Immunodeficiency.

Authors:  Janne Strand; Kiran Aftab Gul; Hans Christian Erichsen; Emma Lundman; Mona C Berge; Anette K Trømborg; Linda K Sørgjerd; Mari Ytre-Arne; Silje Hogner; Ruth Halsne; Hege Junita Gaup; Liv T Osnes; Grete A B Kro; Hanne S Sorte; Lars Mørkrid; Alexander D Rowe; Trine Tangeraas; Jens V Jørgensen; Charlotte Alme; Trude E H Bjørndalen; Arild E Rønnestad; Astri M Lang; Terje Rootwelt; Jochen Buechner; Torstein Øverland; Tore G Abrahamsen; Rolf D Pettersen; Asbjørg Stray-Pedersen
Journal:  Front Immunol       Date:  2020-07-09       Impact factor: 7.561

  4 in total

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