Literature DB >> 19482345

Development of a routine newborn screening protocol for severe combined immunodeficiency.

Mei W Baker1, William J Grossman, Ronald H Laessig, Gary L Hoffman, Charles D Brokopp, Daniel F Kurtycz, Michael F Cogley, Thomas J Litsheim, Murray L Katcher, John M Routes.   

Abstract

BACKGROUND: Severe combined immunodeficiency (SCID) is characterized by the absence of functional T cells and B cells. Without early diagnosis and treatment, infants with SCID die from severe infections within the first year of life.
OBJECTIVE: To determined the feasibility of detecting SCID in newborns by quantitating T-cell receptor excision circles (TRECs) from dried blood spots (DBSs) on newborn screening (NBS) cards.
METHODS: DNA was extracted from DBSs on deidentified NBS cards, and real-time quantitative PCR (RT-qPCR) was used to determine the number of TRECs. Positive controls consisted of DBS from a 1-week-old T(-)B(-)NK(+) patient with SCID and whole blood specimens selectively depleted of naive T cells.
RESULTS: The mean and median numbers of TRECs from 5766 deidentified DBSs were 827 and 708, respectively, per 3.2-mm punch ( approximately 3 muL whole blood). Ten samples failed to amplify TRECs on initial analysis; all but 1 demonstrated normal TRECs and beta-actin amplification on retesting. No TRECs were detected in either the SCID or naive T-cell-depleted samples, despite the presence of normal levels of beta-actin.
CONCLUSIONS: The use of RT-qPCR to quantitate TRECs from DNA extracted from newborn DBSs is a highly sensitive and specific screening test for SCID. This assay is currently being used in Wisconsin for routine screening infants for SCID.

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Year:  2009        PMID: 19482345     DOI: 10.1016/j.jaci.2009.04.007

Source DB:  PubMed          Journal:  J Allergy Clin Immunol        ISSN: 0091-6749            Impact factor:   10.793


  57 in total

1.  Primary immunodeficiency update and newborn screening.

Authors:  Mercedes C Amado
Journal:  Mo Med       Date:  2011 Sep-Oct

2.  Implementing routine testing for severe combined immunodeficiency within Wisconsin's newborn screening program.

Authors:  Mei Wang Baker; Ronald H Laessig; Murray L Katcher; John M Routes; William J Grossman; James Verbsky; Daniel F Kurtycz; Charles D Brokopp
Journal:  Public Health Rep       Date:  2010 May-Jun       Impact factor: 2.792

3.  Cause of death in neonates with inconclusive or abnormal T-cell receptor excision circle assays on newborn screening.

Authors:  Deborah J Accetta; Charles D Brokopp; Mei W Baker; James Verbsky; John M Routes
Journal:  J Clin Immunol       Date:  2011-09-29       Impact factor: 8.317

4.  Neonatal screening for severe combined immunodeficiency.

Authors:  Jennifer M Puck
Journal:  Curr Opin Pediatr       Date:  2011-12       Impact factor: 2.856

Review 5.  The long quest for neonatal screening for severe combined immunodeficiency.

Authors:  Rebecca H Buckley
Journal:  J Allergy Clin Immunol       Date:  2012-01-24       Impact factor: 10.793

6.  Laboratory technology for population-based screening for severe combined immunodeficiency in neonates: the winner is T-cell receptor excision circles.

Authors:  Jennifer M Puck
Journal:  J Allergy Clin Immunol       Date:  2012-01-29       Impact factor: 10.793

7.  Newborn screening for severe combined immunodeficiency; the Wisconsin experience (2008-2011).

Authors:  James W Verbsky; Mei W Baker; William J Grossman; Mary Hintermeyer; Trivikram Dasu; Benedetta Bonacci; Sreelatha Reddy; David Margolis; James Casper; Miranda Gries; Ken Desantes; Gary L Hoffman; Charles D Brokopp; Christine M Seroogy; John M Routes
Journal:  J Clin Immunol       Date:  2011-11-10       Impact factor: 8.317

Review 8.  Advances in basic and clinical immunology in 2010.

Authors:  Javier Chinen; William T Shearer
Journal:  J Allergy Clin Immunol       Date:  2011-02       Impact factor: 10.793

9.  Newborn screening: from Guthrie to whole genome sequencing.

Authors:  Michele Caggana; Elizabeth A Jones; S I Shahied; Susan Tanksley; Cheryl A Hermerath; Ira M Lubin
Journal:  Public Health Rep       Date:  2013 Sep-Oct       Impact factor: 2.792

10.  Improving cellular therapy for primary immune deficiency diseases: recognition, diagnosis, and management.

Authors:  Linda M Griffith; Morton J Cowan; Luigi D Notarangelo; Jennifer M Puck; Rebecca H Buckley; Fabio Candotti; Mary Ellen Conley; Thomas A Fleisher; H Bobby Gaspar; Donald B Kohn; Hans D Ochs; Richard J O'Reilly; J Douglas Rizzo; Chaim M Roifman; Trudy N Small; William T Shearer
Journal:  J Allergy Clin Immunol       Date:  2009-12       Impact factor: 10.793

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