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Record W3094630161 · doi:10.1002/mds.28338

Shared Genetics of Multiple System Atrophy and Inflammatory Bowel Disease

2020· article· en· W3094630161 on OpenAlexfundno aff
Alexey Shadrin, Sören Mucha, David Ellinghaus, Mary B. Makarious, Cornelis Blauwendraat, Ashwin Ashok Kumar Sreelatha, Antonio Heras‐Garvin, Jinhui Ding, Monia Hammer, Alexandra Foubert‐Samier, Wassilios G. Meissner, Olivier Rascol, Anne Pavy‐Le Traon, Oleksandr Frei, Kevin S. O’Connell, Shahram Bahrami, Stefan Schreiber, Wolfgang Lieb, Martina Müller‐Nurasyid, Ulf Schminke, Georg Homuth, Carsten Oliver Schmidt, Markus M. Nöthen, Per Hoffmann, Christian Gieger, Gregor K. Wenning, J. Raphael Gibbs, Andre Franke, John Hardy, Nadia Stefanova, Thomas Gasser, Andrew Singleton, Henry Houlden, Sonja W. Scholz, Ole A. Andreassen, Manu Sharma

Bibliographic record

VenueMovement Disorders · 2020
Typearticle
Languageen
FieldMedicine
TopicParkinson's Disease Mechanisms and Treatments
Canadian institutionsnot available
FundersNational Institute on AgingHelse Sør-Øst RHFMedical Research CouncilNational Institutes of HealthExzellenzclusters EntzündungsforschungMedical Research Council CanadaUniversitetet i OsloMultiple System Atrophy CoalitionNorges ForskningsrådMichael J. Fox Foundation for Parkinson's ResearchAustrian Science FundThe Research CouncilDeutsche ForschungsgemeinschaftASCRS Research FoundationWellcome TrustNational Institute of Neurological Disorders and StrokeBundesministerium für Bildung und Forschung
KeywordsCandidate geneBiologyGeneticsGenome-wide association studyExome sequencingDiseaseInflammatory bowel diseaseImmunologyGenePhenotypeMedicinePathologyGenotypeSingle-nucleotide polymorphism

Abstract

fetched live from OpenAlex

BACKGROUND: Multiple system atrophy (MSA) is a rare neurodegenerative disease characterized by intracellular accumulations of α-synuclein and nerve cell loss in striatonigral and olivopontocerebellar structures. Epidemiological and clinical studies have reported potential involvement of autoimmune mechanisms in MSA pathogenesis. However, genetic etiology of this interaction remains unknown. We aimed to investigate genetic overlap between MSA and 7 autoimmune diseases and to identify shared genetic loci. METHODS: Genome-wide association study summary statistics of MSA and 7 autoimmune diseases were combined in cross-trait conjunctional false discovery rate analysis to explore overlapping genetic background. Expression of selected candidate genes was compared in transgenic MSA mice and wild-type mice. Genetic variability of candidate genes was further investigated using independent whole-exome genotyping data from large cohorts of MSA and autoimmune disease patients and healthy controls. RESULTS: We observed substantial polygenic overlap between MSA and inflammatory bowel disease and identified 3 shared genetic loci with leading variants upstream of the DENND1B and RSP04 genes, and in intron of the C7 gene. Further, the C7 gene showed significantly dysregulated expression in the degenerating midbrain of transgenic MSA mice compared with wild-type mice and had elevated burden of protein-coding variants in independent MSA and inflammatory bowel disease cohorts. CONCLUSION: Our study provides evidence of shared genetic etiology between MSA and inflammatory bowel disease with an important role of the C7 gene in both phenotypes, with the implication of immune and gut dysfunction in MSA pathophysiology. © 2020 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.

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: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.019
Threshold uncertainty score0.559

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 head0.011
GPT teacher head0.217
Teacher spread0.206 · 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

Citations21
Published2020
Admission routes1
Has abstractyes

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