Day T G, Ramanan A V, Hinks A, Lamb R, Packham J, Wise C, Punaro M, Donn R P
University of Manchester, Manchester, UK.
Arthritis Rheum. 2008 Jul;58(7):2142-6. doi: 10.1002/art.23604.
To investigate the association of NLRP3, NOD2, MEFV, and PSTPIP1, genes that cause 4 of the autoinflammatory hereditary periodic fever syndromes (HPFS), with juvenile idiopathic arthritis (JIA).
Fifty-one single-nucleotide polymorphisms (SNPs) across the 4 loci were investigated using MassArray genotyping in 950 Caucasian patients with JIA living in the UK and 728 ethnically matched healthy controls.
Prior to Bonferroni correction for multiple testing, significant genotype associations between 6 SNPs in MEFV and JIA were observed and, in subgroup analysis, associations between 12 SNPs across all 4 loci and the subgroup of patients with psoriatic JIA were found. After Bonferroni correction for multiple testing, 2 genotype associations remained significant in the subgroup of patients with psoriatic JIA (MEFV SNP rs224204 [corrected P = 0.025] and NLRP3 SNP rs3806265 [corrected P = 0.04]).
These findings support the use of monogenic loci as candidates for investigating the genetic component of complex disease and provide preliminary evidence of association between SNPs in autoinflammatory genes and psoriatic JIA. Our findings raise the interesting possibility of a shared disease mechanism between the HPFS and psoriatic JIA, potentially involving abnormal production of interleukin-1beta.
研究导致4种自身炎症性遗传性周期性发热综合征(HPFS)的NLRP3、NOD2、MEFV和PSTPIP1基因与幼年特发性关节炎(JIA)之间的关联。
采用MassArray基因分型技术,对950名居住在英国的白种人JIA患者和728名种族匹配的健康对照进行了这4个基因座上的51个单核苷酸多态性(SNP)研究。
在进行多重检验的Bonferroni校正之前,观察到MEFV基因中6个SNP与JIA之间存在显著的基因型关联,在亚组分析中,发现所有4个基因座上的12个SNP与银屑病性JIA患者亚组之间存在关联。在进行多重检验的Bonferroni校正后,银屑病性JIA患者亚组中有2个基因型关联仍然显著(MEFV SNP rs224204 [校正P = 0.025]和NLRP3 SNP rs3806265 [校正P = 0.04])。
这些发现支持将单基因座作为研究复杂疾病遗传成分的候选基因,并为自身炎症性基因中的SNP与银屑病性JIA之间的关联提供了初步证据。我们的发现提出了一种有趣的可能性,即HPFS和银屑病性JIA之间存在共同的疾病机制,可能涉及白细胞介素-1β的异常产生。