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Record W4292136237 · doi:10.1016/j.jaci.2022.08.004

Aberrant T-cell exhaustion in severe combined immunodeficiency survivors with poor T-cell reconstitution after transplantation

2022· article· en· W4292136237 on OpenAlexafffund
Roxane Labrosse, Inès Boufaied, Benoîte Bourdin, Saideep Gona, Haley E. Randolph, Brent R. Logan, Sara Bourbonnais, Chloé Berthe, Wendy Chan, Rebecca H. Buckley, Roberta Parrott, Geoff D.E. Cuvelier, Neena Kapoor, Sharat Chandra, Blachy J. Dávila Saldaña, Hesham Eissa, Fred Goldman, Jennifer Heimall, Richard J. O’Reilly, Sonali Chaudhury, E. Anders Kolb, Shalini Shenoy, Linda M. Griffith, Michael A. Pulsipher, Donald B. Kohn, Luigi D. Notarangelo, Sung‐Yun Pai, Morton J. Cowan, Christopher C. Dvorak, Élie Haddad, Jennifer M. Puck, Luis B. Barreiro, Hélène Decaluwe

Bibliographic record

VenueJournal of Allergy and Clinical Immunology · 2022
Typearticle
Languageen
FieldImmunology and Microbiology
TopicImmunodeficiency and Autoimmune Disorders
Canadian institutionsCancerCare ManitobaUniversity of ManitobaCentre Hospitalier Universitaire Sainte-JustineUniversité de Montréal
FundersNational Institute of Neurological Disorders and StrokeNational Institute of Allergy and Infectious DiseasesNational Cancer InstituteNational Heart, Lung, and Blood InstituteNational Center for Advancing Translational SciencesHealth Resources and Services AdministrationNational Institutes of HealthOffice of Naval ResearchJazz PharmaceuticalsRare Diseases Clinical Research NetworkCanadian Institutes of Health ResearchOmeros CorporationAlexion PharmaceuticalsInvitaeSt. Baldrick's FoundationEli Lilly and Company
KeywordsImmunodeficiencyTransplantationSevere combined immunodeficiencyMedicineIntensive care medicineImmunologyBiologyInternal medicineImmune systemGenetics

Abstract

fetched live from OpenAlex

Background Severe combined immunodeficiency (SCID) comprises rare inherited disorders of immunity that require definitive treatment through hematopoietic cell transplantation (HCT) or gene therapy for survival. Despite successes of allogeneic HCT, many SCID patients experience incomplete immune reconstitution, persistent T-cell lymphopenia, and poor long-term outcomes. Objective We hypothesized that CD4 + T-cell lymphopenia could be associated with a state of T-cell exhaustion in previously transplanted SCID patients. Methods We analyzed markers of exhaustion in blood samples from 61 SCID patients at a median of 10.4 years after HCT. Results Compared to post-HCT SCID patients with normal CD4 + T-cell counts, those with poor T-cell reconstitution showed lower frequency of naive CD45RA + /CCR7 + T cells, recent thymic emigrants, and TCR excision circles. They also had a restricted TCR repertoire, increased expression of inhibitory receptors (PD-1, 2B4, CD160, BTLA, CTLA-4), and increased activation markers (HLA-DR, perforin) on their total and naive CD8 + T cells, suggesting T-cell exhaustion and aberrant activation, respectively. The exhaustion score of CD8 + T cells was inversely correlated with CD4 + T-cell count, recent thymic emigrants, TCR excision circles, and TCR diversity. Exhaustion scores were higher among recipients of unconditioned HCT, especially when further in time from HCT. Patients with fewer CD4 + T cells showed a transcriptional signature of exhaustion. Conclusions Recipients of unconditioned HCT for SCID may develop late post-HCT T-cell exhaustion as a result of diminished production of T-lineage cells. Elevated expression of inhibitory receptors on their T cells may be a biomarker of poor long-term T-cell reconstitution.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.001
Threshold uncertainty score0.003

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0010.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.009
GPT teacher head0.234
Teacher spread0.225 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

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Citations7
Published2022
Admission routes2
Has abstractno

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