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Record W2059347852 · doi:10.14785/lpsn-2015-0005

RelB deficiency causes combined immunodeficiency

2015· article· en· W2059347852 on OpenAlexafffundvenue
Daniele Merico, Nigel Sharfe, Pingzhao Hu, Jo-Anne Herbrick, Chaim M. Roifman

Bibliographic record

VenueLymphoSign Journal · 2015
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicNF-κB Signaling Pathways
Canadian institutionsGeorge & Fay Yee Centre for Healthcare InnovationUniversity of ManitobaSickKids FoundationHospital for Sick ChildrenUniversity of Toronto
FundersOntario Ministry of Research and InnovationHospital for Sick ChildrenGenome CanadaOntario GenomicsOntario Genomics InstituteImmunodeficiency CanadaJeffrey Modell Foundation
KeywordsRELBBiologyImmunodeficiencySignal transductionLYNImmunologyImmune systemPrimary immunodeficiencyHaploinsufficiencyCIITAGeneticsGeneTranscription factorPhenotypeT cellNFKB1Tyrosine kinase

Abstract

fetched live from OpenAlex

Background: Combined immunodeficiency (CID) presents in infancy with severe microbial infections due to either the depletion or dysfunction of lymphocytes. Several mutated genes have been implicated in causing this condition. These encoded proteins are involved in gene recombination, signal transduction from receptors to transcription factors, or they are critical for lymphocyte development. There remain 20%–30% of patients with similar phenotypes but with no known genetic aberration. The objective of this study was to define the molecular basis of CID in a group of patients. Genotyping was performed using linkage panel chips, and the results were analyzed for parametric linkage. Whole genome sequencing was also performed. In vitro mitogen stimulation, flow cytometry, real time PCR, Western blotting, and cytokine ELISA were used to assess immunological status and signal transduction pathways. We identified a homozygous mutation in the gene for the NFκB transcription factor RelB in 3 patients who suffered repeated infection despite the presence of circulating T and B cells. This mutation introduces a premature stop, resulting in an ablation of RelB expression. Evaluation of patient immune systems revealed reduced response to mitogens and an inability to maintain an adequate antibody response to immunizations. Lack of RelB expression results in a clinical presentation of CID. Statement of novelty: We describe RelB deficiency for the first time.

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: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.010

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0010.000
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)0.0030.001

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 head0.026
GPT teacher head0.251
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 designBench or experimental
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".

Quick stats

Citations31
Published2015
Admission routes3
Has abstractyes

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