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Record W4399552536 · doi:10.1136/gutjnl-2024-332043

Patient-derived organoid biobank identifies epigenetic dysregulation of intestinal epithelial MHC-I as a novel mechanism in severe Crohn’s Disease

2024· article· en· W4399552536 on OpenAlexaff
Thomas W. Dennison, Rachel D. Edgar, Felicity Payne, Komal Nayak, Alexander Ross, Aurélie Cenier, Claire Glemas, Federica Giachero, April Foster, Rebecca Harris, Judith Kraiczy, Camilla Salvestrini, Georgia Stavrou, Franco Torrente, Kimberley Brook, Claire Trayers, Rasa Elmentaite, Gehad Youssef, Bálint Tél, Douglas J. Winton, Nefeli Skoufou-Papoutsaki, Sam Adler, Philip Bufler, Aline Azabdaftari, Andreas Jenke, G Natasha, Natasha Thomas, Erasmo Miele, Abdulrahman Al-Mohammad, Greta Guarda, Subra Kugathasan, Suresh Venkateswaran, Menna R. Clatworthy, Tomas Castro‐Dopico, Ondřej Suchánek, Caterina Strisciuglio, Marco Gasparetto, Seokjun Lee, Xingze Xu, Erica Bello, Namshik Han, Daniel R. Zerbino, Sarah A. Teichmann, Josquin Nys, Robert Heuschkel, Francesca Perrone, Matthias Zilbauer

Bibliographic record

VenueGut · 2024
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicInflammatory Bowel Disease
Canadian institutionsUniversity Health Network
FundersCancer Research UK Cambridge Institute, University of CambridgeNIHR Cambridge Biomedical Research CentreBiomedical Research CouncilMedical Research CouncilBerlin Institute of HealthSchweizerischer Nationalfonds zur Förderung der Wissenschaftlichen ForschungDeutsche ForschungsgemeinschaftCambridge Institute for Medical Research, University of CambridgeUniversité de LausanneNational Institute for Health and Care ResearchGreat Ormond Street Hospital CharityAstraZenecaEuropean Molecular Biology LaboratoryWellcome TrustLeona M. and Harry B. Helmsley Charitable TrustCancer Research UKNational Science Foundation
KeywordsBiologyEpigeneticsOrganoidMHC class IImmunologyHaploinsufficiencyCancer researchMajor histocompatibility complexPhenotypeCell biologyGeneticsGeneImmune system

Abstract

fetched live from OpenAlex

OBJECTIVE: Epigenetic mechanisms, including DNA methylation (DNAm), have been proposed to play a key role in Crohn's disease (CD) pathogenesis. However, the specific cell types and pathways affected as well as their potential impact on disease phenotype and outcome remain unknown. We set out to investigate the role of intestinal epithelial DNAm in CD pathogenesis. DESIGN: We generated 312 intestinal epithelial organoids (IEOs) from mucosal biopsies of 168 patients with CD (n=72), UC (n=23) and healthy controls (n=73). We performed genome-wide molecular profiling including DNAm, bulk as well as single-cell RNA sequencing. Organoids were subjected to gene editing and the functional consequences of DNAm changes evaluated using an organoid-lymphocyte coculture and a nucleotide-binding oligomerisation domain, leucine-rich repeat and CARD domain containing 5 (NLRC5) dextran sulphate sodium (DSS) colitis knock-out mouse model. RESULTS: and cognate gene upregulation. Single-cell RNA sequencing of primary mucosal tissue and IEOs confirmed the role of NLRC5 as transcriptional transactivator in the intestinal epithelium. Increased mucosal MHC-I and NLRC5 expression in adult and paediatric patients with CD was validated in additional cohorts and the functional role of MHC-I highlighted by demonstrating a relative protection from DSS-mediated mucosal inflammation in NLRC5-deficient mice. MHC-I DNAm in IEOs showed a significant correlation with CD disease phenotype and outcomes. Application of machine learning approaches enabled the development of a disease prognostic epigenetic molecular signature. CONCLUSIONS: Our study has identified epigenetically regulated intestinal epithelial MHC-I as a novel mechanism in CD pathogenesis.

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 categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.349
Threshold uncertainty score0.736

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)0.0000.000

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 head0.007
GPT teacher head0.228
Teacher spread0.222 · 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 teacher head, 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

Citations35
Published2024
Admission routes1
Has abstractyes

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