Division of Rheumatology, Department of Internal Medicine, Faculty of Medicine, Kocaeli University, Umuttepe, 42000 Kocaeli, Turkey.
Rheumatol Int. 2013 Jul;33(7):1675-80. doi: 10.1007/s00296-012-2609-8. Epub 2012 Dec 27.
Adult onset Still's disease (ASD) is a systemic inflammatory disorder of unknown etiology. ASD is characterized by fever with unknown etiology, rash, arthritis, and involvement of several organ systems. FMF and TRAPS are two important autoinflammatory diseases which characterized with recurrent inflammatory attacks. We aimed in this study to investigate the MEFV gene and TNFRSF1A gene variations in ASD. Twenty consecutive Turkish ASD patients (14 female and 6 male; mean age 38.45 ± 14; mean disease duration 3.3 ± 2.3; mean age of the disease onset 35.1 ± 14.4) and 103 healthy controls of Turkish origin were analyzed. All ASD patients were genotyped for the 4 MEFV mutations (M694V, E148Q, V726A, M680I) and TNFRSF1A gene exon 2-3 and exon 4-5 by using sequence analysis. The healthy controls are genotyped using PCR-RFLP method for intron 4 variation. The results of MEFV gene mutations screening show an increase in the MEFV mutation rate in ASD group, but it was not significantly different (p = 0.442, OR 1.64, 95 % CI 0.409-6.589). T-C polymorphism (rs1800692) was the only variation in the intron 4 of TNFRSF1A gene that we observed at the ASD patients. The frequency of TT genotype was 15 %, TC: 45 %, and CC: 40 % in ASD patients and the frequencies were 22, 41, and 37 % in healthy controls, respectively. When we analyzed the allele difference between both groups, there was no difference (p = 0.54, OR 1.24, 0.619-2.496-2.654). The variations in MEFV may have role in ASD pathogenesis. Our findings suggest that there is no significant association between ASD and TNFRSF1A variations.
成人Still 病(ASD)是一种病因不明的全身性炎症性疾病。ASD 的特征是发热、皮疹、关节炎和多个器官系统受累。FMF 和 TRAPS 是两种重要的自身炎症性疾病,其特征是反复发作的炎症发作。我们旨在本研究中研究 MEFV 基因和 TNFRSF1A 基因在 ASD 中的变异。连续 20 例土耳其 ASD 患者(14 名女性和 6 名男性;平均年龄 38.45 ± 14;平均病程 3.3 ± 2.3;发病年龄平均为 35.1 ± 14.4)和 103 名土耳其原籍健康对照者进行了分析。所有 ASD 患者均进行了 MEFV 基因突变(M694V、E148Q、V726A、M680I)和 TNFRSF1A 基因外显子 2-3 和外显子 4-5 的测序分析。健康对照组采用 PCR-RFLP 法检测内含子 4 变异。MEFV 基因突变筛查结果显示 ASD 组 MEFV 突变率增加,但差异无统计学意义(p = 0.442,OR 1.64,95%CI 0.409-6.589)。我们在 ASD 患者中仅观察到 TNFRSF1A 基因内含子 4 中的 T-C 多态性(rs1800692)。ASD 患者 TT 基因型频率为 15%,TC 为 45%,CC 为 40%,健康对照组分别为 22%、41%和 37%。当我们分析两组间等位基因差异时,无差异(p = 0.54,OR 1.24,0.619-2.496-2.654)。MEFV 的变异可能在 ASD 的发病机制中起作用。我们的研究结果表明,ASD 与 TNFRSF1A 变异之间无显著相关性。