| Literature DB >> 26598658 |
Michael J Ombrello1, Elaine F Remmers2, Ioanna Tachmazidou3, Alexei Grom4, Dirk Foell5, Johannes-Peter Haas6, Alberto Martini7, Marco Gattorno8, Seza Özen9, Sampath Prahalad10, Andrew S Zeft11, John F Bohnsack12, Elizabeth D Mellins13, Norman T Ilowite14, Ricardo Russo15, Claudio Len16, Maria Odete E Hilario16, Sheila Oliveira17, Rae S M Yeung18, Alan Rosenberg19, Lucy R Wedderburn20, Jordi Anton21, Tobias Schwarz22, Anne Hinks23, Yelda Bilginer9, Jane Park13, Joanna Cobb24, Colleen L Satorius2, Buhm Han25, Elizabeth Baskin26, Sara Signa27, Richard H Duerr28, J P Achkar29, M Ilyas Kamboh30, Kenneth M Kaufman4, Leah C Kottyan4, Dalila Pinto31, Stephen W Scherer32, Marta E Alarcón-Riquelme33, Elisa Docampo34, Xavier Estivill35, Ahmet Gül36, Paul I W de Bakker37, Soumya Raychaudhuri38, Carl D Langefeld39, Susan Thompson4, Eleftheria Zeggini3, Wendy Thomson24, Daniel L Kastner40, Patricia Woo41.
Abstract
Systemic juvenile idiopathic arthritis (sJIA) is an often severe, potentially life-threatening childhood inflammatory disease, the pathophysiology of which is poorly understood. To determine whether genetic variation within the MHC locus on chromosome 6 influences sJIA susceptibility, we performed an association study of 982 children with sJIA and 8,010 healthy control subjects from nine countries. Using meta-analysis of directly observed and imputed SNP genotypes and imputed classic HLA types, we identified the MHC locus as a bona fide susceptibility locus with effects on sJIA risk that transcended geographically defined strata. The strongest sJIA-associated SNP, rs151043342 [P = 2.8 × 10(-17), odds ratio (OR) 2.6 (2.1, 3.3)], was part of a cluster of 482 sJIA-associated SNPs that spanned a 400-kb region and included the class II HLA region. Conditional analysis controlling for the effect of rs151043342 found that rs12722051 independently influenced sJIA risk [P = 1.0 × 10(-5), OR 0.7 (0.6, 0.8)]. Meta-analysis of imputed classic HLA-type associations in six study populations of Western European ancestry revealed that HLA-DRB1*11 and its defining amino acid residue, glutamate 58, were strongly associated with sJIA [P = 2.7 × 10(-16), OR 2.3 (1.9, 2.8)], as was the HLA-DRB1*11-HLA-DQA1*05-HLA-DQB1*03 haplotype [6.4 × 10(-17), OR 2.3 (1.9, 2.9)]. By examining the MHC locus in the largest collection of sJIA patients assembled to date, this study solidifies the relationship between the class II HLA region and sJIA, implicating adaptive immune molecules in the pathogenesis of sJIA.Entities:
Keywords: Still’s disease; autoinflammation; human leukocyte antigen; systemic juvenile idiopathic arthritis
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Year: 2015 PMID: 26598658 PMCID: PMC4702958 DOI: 10.1073/pnas.1520779112
Source DB: PubMed Journal: Proc Natl Acad Sci U S A ISSN: 0027-8424 Impact factor: 11.205