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Record W4392338914 · doi:10.1093/brain/awae048

HLA-DQB1*05 subtypes and not DRB1*10:01 mediates risk in anti-IgLON5 disease

2024· article· en· W4392338914 on OpenAlexafffund
Selina Yogeshwar, Sergio Muñiz‐Castrillo, Lídia Sabater, Vicente Peris-Sempere, Vamsee Mallajosyula, Luo Guo, Han Yan, Eric Yu, Jing Zhang, Ling Lin, Flavia Fagundes Bueno, Xuhuai Ji, Géraldine Picard, Véronique Rogemond, Anne Laurie Pinto, Anna Heidbreder, Romana Höftberger, Francesc Graus, Josep Dalmau, Joan Santamaría, Alex Iranzo, Bettina Schreiner, Maria Pia Giannoccaro, Rocco Liguori, Takayoshi Shimohata, Akio Kimura, Yoya Ono, Sophie Binks, Sara Mariotto, Alessandro Dinoto, Michael Bonello, Christian Hartmann, Nicola Tambasco, Pasquale Nigro, Harald Prüß, Andrew McKeon, Mark M. Davis, Sarosh R. Irani, Jérôme Honnorat, Carles Gaig, Carsten Finke, Emmanuel Mignot

Bibliographic record

VenueBrain · 2024
Typearticle
Languageen
FieldImmunology and Microbiology
TopicImmune Cell Function and Interaction
Canadian institutionsInstitute of Infection and Immunity
FundersEuropean Regional Development FundInstituto de Salud Carlos IIINeuromuscular Research Association BaselEinstein Stiftung BerlinEinstein Center for Neurosciences BerlinDeutsche ForschungsgemeinschaftHelmholtz-GemeinschaftNational Institutes of HealthEpilepsy Research UKNational Institute of Neurological Disorders and StrokeBundesministerium für Bildung und ForschungNational Institute for Health and Care ResearchMedical Research Council CanadaDeutscher Akademischer AustauschdienstUS-UK Fulbright CommissionResearch and Innovative Technology AdministrationWellcomeBritish Medical AssociationAgence Nationale de la RechercheWellcome TrustNational Institute of Allergy and Infectious DiseasesMedical Research CouncilNational Institute on Handicapped Research
KeywordsDiseaseHuman leukocyte antigenBiologyMedicineGeneticsPathology

Abstract

fetched live from OpenAlex

Anti-IgLON5 disease is a rare and likely underdiagnosed subtype of autoimmune encephalitis. The disease displays a heterogeneous phenotype that includes sleep, movement and bulbar-associated dysfunction. The presence of IgLON5-antibodies in CSF/serum, together with a strong association with HLA-DRB1*10:01∼DQB1*05:01, supports an autoimmune basis. In this study, a multicentric human leukocyte antigen (HLA) study of 87 anti-IgLON5 patients revealed a stronger association with HLA-DQ than HLA-DR. Specifically, we identified a predisposing rank-wise association with HLA-DQA1*01:05∼DQB1*05:01, HLA-DQA1*01:01∼DQB1*05:01 and HLA-DQA1*01:04∼DQB1*05:03 in 85% of patients. HLA sequences and binding cores for these three DQ heterodimers were similar, unlike those of linked DRB1 alleles, supporting a causal link to HLA-DQ. This association was further reflected in an increasingly later age of onset across each genotype group, with a delay of up to 11 years, while HLA-DQ-dosage dependent effects were also suggested by reduced risk in the presence of non-predisposing DQ1 alleles. The functional relevance of the observed HLA-DQ molecules was studied with competition binding assays. These proof-of-concept experiments revealed preferential binding of IgLON5 in a post-translationally modified, but not native, state to all three risk-associated HLA-DQ receptors. Further, a deamidated peptide from the Ig2-domain of IgLON5 activated T cells in two patients, compared with one control carrying HLA-DQA1*01:05∼DQB1*05:01. Taken together, these data support a HLA-DQ-mediated T-cell response to IgLON5 as a potentially key step in the initiation of autoimmunity in this disease.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesInsufficient payload (model declined to judge)
Consensus categoriesInsufficient payload (model declined to judge)
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Not applicable · Consensus signal: Not applicable
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.364
Threshold uncertainty score0.999

Codex and Gemma teacher scores by category

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)0.0030.002

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 head0.008
GPT teacher head0.232
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; both teacher heads agree on what is shown here.

Study designNot applicable
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

Citations41
Published2024
Admission routes2
Has abstractyes

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