The cost-benefit of newborn screening for X-linked agammaglobulinemia and related B-cell lymphopenia.

Eyal Grunebaum, Vanessa Tenembaum, Jessica Quinn, Fred Modell, Raz Somech

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Review in Immunologic research, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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5 · Who and what money

Authors and funding

5 authors.

Eyal GrunebaumThe Division of Immunology and Allergy, The Hospital for Sick Children, Toronto, ON, Canada.

Vanessa TenembaumJeffrey Modell Foundation, 780 Third Avenue, 47th Floor, New York City, NY, 10017, USA.

Jessica QuinnJeffrey Modell Foundation, 780 Third Avenue, 47th Floor, New York City, NY, 10017, USA.

Fred ModellJeffrey Modell Foundation, 780 Third Avenue, 47th Floor, New York City, NY, 10017, USA.

Raz SomechPediatric Department and the Immunology Service, Jeffrey Modell Foundation Center, "Edmond and Lily Safra" Children's Hospital, Sheba Medical Center, Tel Hashomer, 52621, Israel. raz.somech@sheba.health.gov.il.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Early detection of X-linked agammaglobulinemia (XLA) and other profound B cell lymphopenias (BCL) in newborns is crucial for timely intervention and improved clinical outcomes. The kappa light chain rearrangement excision circles (KREC) assay has emerged as an effective specific and sensitive tool for newborn screening (NBS) of XLA and other BCL. Large-scale studies have demonstrated that KREC-based NBS identifies affected infants before symptom onset, enabling prompt initiation of immunoglobulin replacement therapy (IGRT) and prophylactic antibiotics, thereby preventing recurrent infections, chronic organ damage, and reducing healthcare burden. Here, we evaluate the possible cost-benefit of NBS for XLA and other BCL. Economic evaluation shows that adding KREC screening to existing NBS programs is likely to be cost-effective and fulfills established criteria for NBS inclusion, supporting its adoption in universal NBS protocols.

Indexed as

AgammaglobulinemiaB-LymphocytesGenetic Diseases, X-LinkedLymphopeniaNeonatal ScreeningAgammaglobulinaemia Tyrosine KinaseCost-Benefit AnalysisHumansImmunoglobulin kappa-ChainsInfant, NewbornAgammaglobulinaemia Tyrosine KinaseImmunoglobulin kappa-ChainsB cell lymphopeniaBTKCost-benefitsKRECNewborn screeningX-linked agammaglobulinemia

Identifiers

PMID42742808

DOI10.1007/s12026-026-09842-7

PMCPMC13577962

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.