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Record W2099399490 · doi:10.1371/journal.pone.0007154

Genetic Variation in the Familial Mediterranean Fever Gene (MEFV) and Risk for Crohn's Disease and Ulcerative Colitis

2009· article· en· W2099399490 on OpenAlexafffundabout
Alexandra–Chloé Villani, Mathieu Lemire, Édouard Louis, Mark S. Silverberg, Catherine Collette, Geneviève Fortin, Elaine R. Nimmo, Yannick Renaud, Sébastien Brunet, Cécile Libioulle, Jacques Bélaïche, Alain Bitton, Daniel Gaudet, Albert Cohen, Diane Langelier, John D. Rioux, Ian Arnott, Gary Wild, Paul Rutgeerts, Jack Satsangi, Séverine Vermeire, Thomas J. Hudson, Denis Franchimont

Bibliographic record

VenuePLoS ONE · 2009
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicInflammasome and immune disorders
Canadian institutionsUniversité de MontréalUniversité du Québec à ChicoutimiMount Sinai HospitalRoyal Victoria HospitalUniversity of TorontoOntario Institute for Cancer ResearchMcGill University and Génome Québec Innovation CentreJewish General HospitalMontreal General HospitalMontreal Heart InstituteCentre Hospitalier Universitaire de SherbrookeMcGill University Health Centre
FundersNational Institute of Allergy and Infectious DiseasesFonds De La Recherche Scientifique - FNRSVlaamse regeringNational Institute of Diabetes and Digestive and Kidney DiseasesCrohn's and Colitis Foundation of CanadaMcGill UniversityMcGill University Health CentreWellcome TrustOntario Institute for Cancer ResearchCrohn's and Colitis FoundationGovernment of OntarioCrohn's and Colitis Foundation of America
KeywordsMEFVFamilial Mediterranean feverHaplotypeSingle-nucleotide polymorphismAllelePyrin domainUlcerative colitisGenotypeInflammatory bowel diseaseAllele frequencyGeneticsInflammasomeCandidate geneSanger sequencingGenotypingBiologyMedicineImmunologyGeneInternal medicineDiseaseGene mutationMutation

Abstract

fetched live from OpenAlex

BACKGROUND AND AIMS: The familial Mediterranean fever (FMF) gene (MEFV) encodes pyrin, a major regulator of the inflammasome platform controlling caspase-1 activation and IL-1beta processing. Pyrin has been shown to interact with the gene product of NLRP3, NALP3/cryopyrin, also an important active member of the inflammasome. The NLRP3 region was recently reported to be associated with Crohn's disease (CD) susceptibility. We therefore sought to evaluate MEFV as an inflammatory bowel disease (IBD) susceptibility gene. METHODOLOGY AND RESULTS: MEFV colonic mucosal gene expression was significantly increased in experimental colitis mice models (TNBS p<0.0003; DSS p<0.006), in biopsies from CD (p<0.02) and severe ulcerative colitis (UC) patients (p<0.008). Comprehensive genetic screening of the MEFV region in the Belgian exploratory sample set (440 CD trios, 137 UC trios, 239 CD cases, 96 UC cases, and 107 healthy controls) identified SNPs located in the MEFV 5' haplotype block that were significantly associated with UC (rs224217; p = 0.003; A allele frequency: 56% cases, 45% controls), while no CD associations were observed. Sequencing and subsequent genotyping of variants located in this associated haplotype block identified three synonymous variants (D102D/rs224225, G138G/rs224224, A165A/rs224223) and one non-synonymous variant (R202Q/rs224222) located in MEFV exon 2 that were significantly associated with UC (rs224222: p = 0.0005; A allele frequency: 32% in cases, 23% in controls). No consistent associations were observed in additional Canadian (256 CD trios, 91 UC trios) and Scottish (495 UC, 370 controls) sample sets. We note that rs224222 showed marginal association (p = 0.012; G allele frequency: 82% in cases, 70% in controls) in the Canadian sample, but with a different risk allele. None of the NLRP3 common variants were associated with UC in the Belgian-Canadian UC samples and no significant interactions were observed between NLRP3 and MEFV that could explain the observed flip-flop of the rs224222 risk allele. CONCLUSION: The differences in association levels observed between the sample sets may be a consequence of distinct founder effects or of the relative small sample size of the cohorts evaluated in this study. However, the results suggest that common variants in the MEFV region do not contribute to CD and UC susceptibility.

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: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.954
Threshold uncertainty score0.297

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 head0.013
GPT teacher head0.217
Teacher spread0.204 · 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

Citations60
Published2009
Admission routes3
Has abstractyes

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