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Record W4415497984 · doi:10.1136/gutjnl-2024-334498

First genome-wide association study reveals immune-mediated aetiopathology in idiopathic achalasia

2025· article· en· W4415497984 on OpenAlexaff
Sandeep Grover, Ines Gockel, Anna Latiano, Anna Mokrowiecka, Pouria Dasmeh, Mira M. Wouters, Zuzana Vacková, Stephan Haas, Tania Triantafyllou, Nicole Kreuser, Jessica Trautmann, Stefan Niebisch, Timo Hess, René Thieme, Jessica Bigge, Hubert Louis, Eric Quertinmont, Aline Meirhaeghe, Manon Muntaner, Philippe Amouyel, Guillaume Gourcerol, Stanislas Bruley des Varannes, François Mion, Michael Vieth, Nikolaos Scarmeas, Orazio Palmieri, Francesca Tavano, Roberto De Giorgio, Daniela Galimberti, Andrea Arighi, Beatrice Arosio, Marco J. Bruno, Justyna Wasielica-Berger, Magdalena Gawron-Kiszka, Maria Janiak, Magdalena Siepsiak, Krystian Adrych, Tomasz Marek, A Dâbrowski, Marek Majewski, Piotr Gietka, Maciej Gonciarz, Laisy Zacarías Martínez, Vilmantas Giedraitis, Lena Kilander, Laura Fratiglioni, Luís Miguel Real, Julius Špičák, Jan Tack, Stefanie Heilmann-Heimbach, Markus M. Nöthen, Martin Ingelsson, Caroline Graff, Agustín Ruiz, Jean‐Charles Lambert, Alfredo Ramı́rez, Alexander J. Eckardt, Michaela Müller, Michael Knapp, Thaddäus T Wissinowski, Jutta Keller, Christiane J. Bruns, Christian Gerges, Horst Neuhaus, Thomas Rösch, Britta Siegmund, Brigitte Schumacher, Marino Venerito, Antonio Ruiz de León, Riccardo Rosati, Vito Annese, Uberto Fumagalli, Luigi Laghi, Elena Urcelay, Fabienne Vavasseur, Sabine Roman, Ping‐Hong Zhou, Quan‐Lin Li, Zuqiang Liu, Burkhard H.A. von Rahden, Dimitris Theodorou, Ewa Małecka‐Panas, Carlo Maj, A. Vigo, Jan Martínek, Guy E. Boeckxstaens, Johannes Schumacher

Bibliographic record

VenueGut · 2025
Typearticle
Languageen
FieldMedicine
TopicGastroesophageal reflux and treatments
Canadian institutionsOntario Brain InstituteUniversity of TorontoUniversity Health Network
FundersDeutsche ForschungsgemeinschaftEU Joint Programme – Neurodegenerative Disease ResearchAlzheimer's Association
KeywordsGenome-wide association studyAssociation (psychology)Identification (biology)AchalasiaGenetic association

Abstract

fetched live from OpenAlex

Background Idiopathic achalasia (IA) is characterised by the degeneration of neurons in the myenteric plexus leading to an irreversible impaired oesophageal function. Although immune-mediated mechanisms have been proposed, the underlying aetiopathology of IA remains poorly understood. Objective This study aimed to uncover the genetic risk architecture of IA. Design We carried out the first genome-wide association study (GWAS) on 4602 European patients with IA and 10 766 ethnically-matched controls. Results A single nucleotide polymorphism (SNP) in HLA-DQB1 leading to an 8-amino acid insertion on the protein level conferred strongest IA risk (PQGPPPAG: p=3.27×10 –68 , OR=2.45). Conditional analyses within the HLA locus revealed a complex genetic risk architecture. Three additional amino acid positions showed independent IA association (Omnibus p<5×10 −8 ). These refer to positions 41 and 130 in HLA-DQα1, position 45 in HLA-DQβ1 and position 86 in HLA-DRβ1. Together, these findings highlight the pivotal role of class II HLA genetic variation in IA pathogenesis. Outside HLA, three independent variants showed IA association (p<5×10 −8 ). One leads to an amino acid substitution with functional effect in PTPN22. Another risk variant leads to a downregulated expression of TNFSF8 , TNFSF15 and TNC in immune cells. The third risk SNP is located near ZNF365 , but the exact underlying cellular mechanism remains unknown. Beyond the single marker level, polygenic risk scores revealed that patients with IA can be stratified based on their genetic risk. In addition, IA shows a shared aetiopathology with Crohn’s disease (r g =0.335). Integrating GWAS and single-cell RNA-sequencing data from the myenteric plexus showed that the memory T-cell type FOS + Tc4 + CD8 + plays a central role in IA development (p=2.50×10 −19 ). Conclusion This GWAS led to the identification of SNPs, cellular mechanisms and cell types that are involved in IA aetiopathology.

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)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation 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 score0.004
Threshold uncertainty score0.560

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
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 head0.009
GPT teacher head0.275
Teacher spread0.265 · 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 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".

Quick stats

Citations2
Published2025
Admission routes1
Has abstractyes

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