Evidence map›Paper›PMID 39734035›Full record

ArticleThe Journal of allergy and clinical immunology2025

Thymic and T-cell intrinsic critical roles associated with severe combined immunodeficiency and Omenn syndrome due to a heterozygous variant (G201R) in PSMB10.

Hye Sun Kuehn, Marita Bosticardo, Antonio C Arrieta, Jennifer L Stoddard, Francesca Pala, Julie E Niemela, Agustin A Gil Silva, Paighton L King, Ana Esteve-Sole, Amreen Naveen and 6 more

Abstract read
In one paragraph

Article in The Journal of allergy and clinical immunology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

5 citing papers in PubMed.

  1. Trial
  2. Review
  3. Article
  4. Review
  5. Novel de novo dominantJournal of human immunity · 2026
    Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

16 authors.

Hye Sun KuehnImmunology Service, Department of Laboratory Medicine, National Institutes of Health Clinical Center, Bethesda, Md.
Marita BosticardoLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Antonio C ArrietaDivision of Infectious Disease, Children's Hospital of Orange County, Orange, Calif.
Jennifer L StoddardImmunology Service, Department of Laboratory Medicine, National Institutes of Health Clinical Center, Bethesda, Md.
Francesca PalaLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Julie E NiemelaImmunology Service, Department of Laboratory Medicine, National Institutes of Health Clinical Center, Bethesda, Md.
Agustin A Gil SilvaImmunology Service, Department of Laboratory Medicine, National Institutes of Health Clinical Center, Bethesda, Md.
Paighton L KingDivision of Hematology, Children's Hospital of Orange County, Orange, Calif.
Ana Esteve-SoleImmunology Service, Department of Laboratory Medicine, National Institutes of Health Clinical Center, Bethesda, Md.
Amreen NaveenLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Eduardo AnayaLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Pooi Meng TruongLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
Ottavia M DelmonteLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md.
David K BuchbinderDivision of Hematology, Children's Hospital of Orange County, Orange, Calif. Electronic address: dbuchbinder@choc.org.
Sergio D RosenzweigImmunology Service, Department of Laboratory Medicine, National Institutes of Health Clinical Center, Bethesda, Md. Electronic address: srosenzweig@cc.nih.gov.
Luigi D NotarangeloLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Md. Electronic address: luigi.notarangelo2@nih.gov.

Funding

Studies of Inherited Causes of Fungal SusceptabilityZIAAI001122 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI ROSENZWEIG, SERGIO · 2010 to 2025
$26.0M
Intramural NIH HHS Z99 CL999999Intramural NIH HHS ZIA AI001122
6 · The paper itself

Abstract

backgroundHeterozygous immunoproteasome 20 S subunit beta 10 (PSMB10) mutations can cause severe combined immunodeficiency and Omenn syndrome. Hematopoietic stem cell transplantation in these patients is associated with severe complications and poor immune reconstitution, often resulting in death.

objectiveWe sought to perform immunologic and molecular characterization of an infant with a PSMB10 heterozygous variant.

methodsA heterozygous variant in PSMB10 (p.G201R) was identified in the index patient but not her parents. Detailed immunophenotyping and functional studies, including flow cytometry, immunoblotting, and T-cell development in artificial thymic organoids, were performed.

resultsThe patient presented with severe B-, natural killer-, and T-cell lymphopenia, with a progressive increase in memory CD4

conclusionsThe PSMB10 G201R variant is associated with reduced immunoproteasome expression levels that appear to play vital roles in hematopoietic and extrahematopoietic immune system development and function. PSMB10-associated thymoproteasome dysfunction leads to impaired thymopoiesis and the development of severe combined immunodeficiency and Omenn syndrome, suggesting possible benefit from thymus implantation.

Indexed as

Proteasome Endopeptidase ComplexSevere Combined ImmunodeficiencyThymus GlandT-LymphocytesHeterozygoteHumansMutationProteasome Endopeptidase Complexartificial thymic organoidImmunoproteasomeinborn errors of immunityprimary immunodeficiencythymoproteasome

Identifiers

PMID39734035
PMCPMC11972880

What Socratic holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

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 Socratic graph.