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Record W2394486176 · doi:10.1534/g3.116.030841

Analysis of Plasminogen Genetic Variants in Multiple Sclerosis Patients

2016· article· en· W2394486176 on OpenAlexafffundabout
A. Dessa Sadovnick, Anthony Traboulsee, Cecily Q. Bernales, Jay P. Ross, Amanda L. Forwell, Irene M. Yee, Léna Guillot‐Noël, Bertrand Fontaine, Isabelle Cournu‐Rebeix, Antonio Alcina, Marı́a Fedetz, Guillermo Izquierdo, Fuencisla Matesanz, Kelly Hilven, Bénédicte Dubois, An Goris, Ianire Astobiza, Iraide Alloza, Alfredo Rodríguez Antigüedad, Koen Vandenbroeck, Denis A. Akkad, Orhan Aktaş, Paul Blaschke, Mathias Buttmann, Andrew Chan, Lisa-Ann Gerdes, Antje Kroner, Christian Kubisch, Tania Kümpfel, Peter Lohse, Uwe K. Zettl, Frauke Zipp, Lars Bertram, Christina M. Lill, Óscar Fernández, Patricia Urbaneja, Laura Leyva, José C. Álvarez‐Cermeño, Rafael Arroyo, Aroa M Garagorri, Ángel García-Martínez, Luisa María Villar, Elena Urcelay, Sunny Malhotra, Xavier Montalbán, Manuel Comabella, Thomas Berger, Franz Fazekas, Markus Reindl, M. Schmied, Alexander Zimprich, Carles Vilariño‐Güell

Bibliographic record

VenueG3 Genes Genomes Genetics · 2016
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicProtease and Inhibitor Mechanisms
Canadian institutionsUniversity of British Columbia
FundersInstituto de Salud Carlos IIIVlaamse regeringEMD SeronoVancouver Coastal Health Research InstituteFonds Wetenschappelijk OnderzoekChugai PharmaceuticalAgence Nationale de la RechercheSanofiVancouver FoundationCanadian Institutes of Health ResearchJunta de AndalucíaFondation pour l'Aide à la Recherche sur la Sclérose en PlaquesKU LeuvenTeva Pharmaceutical IndustriesBiogen
KeywordsMissense mutationProbandMultiple sclerosisGenotypingPenetranceDiseaseGeneticsMedicineFamily aggregationInternal medicineBiologyImmunologyGenotypeMutationPhenotypeGene

Abstract

fetched live from OpenAlex

Multiple sclerosis (MS) is a prevalent neurological disease of complex etiology. Here, we describe the characterization of a multi-incident MS family that nominated a rare missense variant (p.G420D) in plasminogen (PLG) as a putative genetic risk factor for MS. Genotyping of PLG p.G420D (rs139071351) in 2160 MS patients, and 886 controls from Canada, identified 10 additional probands, two sporadic patients and one control with the variant. Segregation in families harboring the rs139071351 variant, identified p.G420D in 26 out of 30 family members diagnosed with MS, 14 unaffected parents, and 12 out of 30 family members not diagnosed with disease. Despite considerably reduced penetrance, linkage analysis supports cosegregation of PLG p.G420D and disease. Genotyping of PLG p.G420D in 14446 patients, and 8797 controls from Canada, France, Spain, Germany, Belgium, and Austria failed to identify significant association with disease (P = 0.117), despite an overall higher prevalence in patients (OR = 1.32; 95% CI = 0.93-1.87). To assess whether additional rare variants have an effect on MS risk, we sequenced PLG in 293 probands, and genotyped all rare variants in cases and controls. This analysis identified nine rare missense variants, and although three of them were exclusively observed in MS patients, segregation does not support pathogenicity. PLG is a plausible biological candidate for MS owing to its involvement in immune system response, blood-brain barrier permeability, and myelin degradation. Moreover, components of its activation cascade have been shown to present increased activity or expression in MS patients compared to controls; further studies are needed to clarify whether PLG is involved in MS 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: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.270
Threshold uncertainty score0.874

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 head0.020
GPT teacher head0.212
Teacher spread0.192 · 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

Citations15
Published2016
Admission routes3
Has abstractyes

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