Thompson Susan D, Moroldo Marta B, Guyer Laura, Ryan Mary, Tombragel Esther M, Shear Edith S, Prahalad Sampath, Sudman Marc, Keddache Mehdi A, Brown W Mark, Giannini Edward H, Langefeld Carl D, Rich Stephen S, Nichols William C, Glass David N
Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.
Arthritis Rheum. 2004 Sep;50(9):2920-30. doi: 10.1002/art.20425.
Juvenile rheumatoid arthritis (JRA) represents a heterogeneous group of disorders with a complex genetic component. A genome scan was performed to detect linkage to JRA in 121 families containing 247 affected children in North America (the JRA Affected Sibpair [ASP] Registry).
Genotype data collected for HLA-DR and 386 microsatellite markers were subjected to multipoint nonparametric linkage analysis. Following analysis of the entire set of families, additional analyses were performed after a priori stratification by disease onset type, age at onset, disease course, and selected HLA-DRB1 alleles.
Linkage of JRA to the HLA region was confirmed (logarithm of odds [LOD] score 2.26). Additional evidence supporting linkage of JRA was observed at 1p36 (D1S214; LOD 1.65), 19p13 (D19S216; LOD 1.72), and 20q13 (D20S100; LOD 1.75). For early-onset polyarticular disease, evidence of linkage was found at chromosome 7q11 (D7S502; LOD 3.47). For pauciarticular disease, evidence supporting linkage was observed on chromosome 19p13 (D19S216; LOD 2.98), the same marker that supported linkage to the "JRA" phenotype. Other regions supporting linkage with JRA disease subtype included 20q13, 4q24, 12q24, and Xp11. Stratification of families based on the presence of the HLA-DR8 allele in affected siblings resulted in significant linkage observed at 2p25 (D2S162/D2S305; LOD 6.0).
These data support the hypothesis that multiple genes, including at least 1 in the HLA region, influence susceptibility to JRA. These findings for JRA are consistent with findings for other autoimmune diseases and support the notion that common genetic regions contribute to an autoimmune phenotype.
青少年类风湿性关节炎(JRA)是一组具有复杂遗传成分的异质性疾病。在北美121个家庭(JRA受累同胞对[ASP]登记处)中对247名患病儿童进行了全基因组扫描,以检测与JRA的连锁关系。
收集的HLA-DR和386个微卫星标记的基因型数据进行多点非参数连锁分析。在对整个家庭组进行分析之后,在先按疾病起病类型、起病年龄、病程和选定的HLA-DRB1等位基因进行分层后,又进行了额外的分析。
证实JRA与HLA区域存在连锁关系(优势对数[LOD]分数为2.26)。在1p36(D1S214;LOD 1.65)、19p13(D19S216;LOD 1.72)和20q13(D20S100;LOD 1.75)观察到支持JRA连锁关系的其他证据。对于早发性多关节疾病,在染色体7q11(D7S502;LOD 3.47)发现了连锁证据。对于少关节疾病,在染色体19p13(D19S216;LOD 2.98)观察到支持连锁关系的证据,该标记也支持与“JRA”表型的连锁关系。其他支持与JRA疾病亚型连锁的区域包括20q13、4q24、12q24和Xp11。根据受累同胞中HLA-DR8等位基因的存在情况对家庭进行分层,结果在2p25(D2S162/D2S305;LOD 6.0)观察到显著连锁。
这些数据支持以下假设,即多个基因,包括HLA区域中至少一个基因,影响对JRA的易感性。JRA 的这些发现与其他自身免疫性疾病的发现一致,并支持共同遗传区域促成自身免疫表型的观点。