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Genetic architecture distinguishes systemic juvenile idiopathic arthritis from other forms of juvenile idiopathic arthritis: clinical and therapeutic implications

2016· article· en· W2560661426 on OpenAlexafffund
Michael J. Ombrello, Victoria Arthur, Elaine F. Remmers, Anne Hinks, Ioanna Tachmazidou, Alexei A. Grom, Dirk Foell, Alberto Martini, Marco Gattorno, Seza Özen, Sampath Prahalad, Andrew Zeft, John F. Bohnsack, Norman T. Ilowite, Elizabeth Mellins, Ricardo Russo, Cláudio Arnaldo Len, Maria Odete Esteves Hilário, Sheila Oliveira, Rae S. M. Yeung, Alan Rosenberg, Lucy R. Wedderburn, Jordi Antón, Johannes‐Peter Haas, Angela Rösen‐Wolff, Kirsten Minden, Klaus Tenbrock, Erkan Demirkaya, Joanna Cobb, Elizabeth Baskin, Sara Signa, Emily G. Shuldiner, Richard H. Duerr, Jean–Paul Achkar, M. Ilyas Kamboh, Kenneth M. Kaufman, Leah C. Kottyan, Dalila Pinto, Stephen W. Scherer, Marta E. Alarcón‐Riquelme, Elisa Docampo, Xavier Estivill, Ahmet Gül, Carl D. Langefeld, Susan D. Thompson, Eleftheria Zeggini, Daniel L. Kastner, Patricia Woo, Wendy Thomson

Bibliographic record

VenueAnnals of the Rheumatic Diseases · 2016
Typearticle
Languageen
FieldMedicine
TopicAutoimmune and Inflammatory Disorders Research
Canadian institutionsSickKids FoundationHospital for Sick ChildrenUniversity of SaskatchewanUniversity of Toronto
FundersSanofiNational Institute of Arthritis and Musculoskeletal and Skin DiseasesNational Institute of Diabetes and Digestive and Kidney DiseasesNHS Blood and TransplantNational Institute for Health and Care ResearchNational Institute on AgingCanadian Institutes of Health ResearchNational Institutes of HealthSwedish Orphan BiovitrumServierBundesministerium für Bildung und ForschungArthritis SocietyCincinnati Children's Hospital Medical CenterMarcus FoundationSparksVersus ArthritisF. Hoffmann-La RocheMedical Research CouncilCelgeneCanadian Arthritis NetworkNational Human Genome Research InstituteWellcome TrustGlaxoSmithKlinePfizer
KeywordsMedicineArthritisGenome-wide association studyGenetic architectureSingle-nucleotide polymorphismJuvenileDiseaseBioinformaticsGeneticsImmunologyGenotypePopulationBiologyInternal medicineGeneQuantitative trait locus

Abstract

fetched live from OpenAlex

OBJECTIVES: Juvenile idiopathic arthritis (JIA) is a heterogeneous group of conditions unified by the presence of chronic childhood arthritis without an identifiable cause. Systemic JIA (sJIA) is a rare form of JIA characterised by systemic inflammation. sJIA is distinguished from other forms of JIA by unique clinical features and treatment responses that are similar to autoinflammatory diseases. However, approximately half of children with sJIA develop destructive, long-standing arthritis that appears similar to other forms of JIA. Using genomic approaches, we sought to gain novel insights into the pathophysiology of sJIA and its relationship with other forms of JIA. METHODS: We performed a genome-wide association study of 770 children with sJIA collected in nine countries by the International Childhood Arthritis Genetics Consortium. Single nucleotide polymorphisms were tested for association with sJIA. Weighted genetic risk scores were used to compare the genetic architecture of sJIA with other JIA subtypes. RESULTS: The major histocompatibility complex locus and a locus on chromosome 1 each showed association with sJIA exceeding the threshold for genome-wide significance, while 23 other novel loci were suggestive of association with sJIA. Using a combination of genetic and statistical approaches, we found no evidence of shared genetic architecture between sJIA and other common JIA subtypes. CONCLUSIONS: The lack of shared genetic risk factors between sJIA and other JIA subtypes supports the hypothesis that sJIA is a unique disease process and argues for a different classification framework. Research to improve sJIA therapy should target its unique genetics and specific pathophysiological pathways.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.002
Version: metacan-v3-hybrid-931329e0061cValidation 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 score0.002
Threshold uncertainty score0.003

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0010.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.001
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0010.000

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 head0.040
GPT teacher head0.321
Teacher spread0.282 · 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 source (direct Gemma or distilled Codex), 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".

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Citations164
Published2016
Admission routes2
Has abstractyes

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