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成人家族性地中海热患者的疾病严重程度和基因型-表型相关性。

Disease severity and genotype-phenotype correlation in adult patients with familial Mediterranean fever.

机构信息

Division of Rheumatology, Department of Internal Medicine, School of Medicine, Kocaeli University, Kocaeli, Turkey.

出版信息

Mod Rheumatol. 2023 Dec 22;34(1):214-219. doi: 10.1093/mr/road007.

Abstract

OBJECTIVES

To assess the relationships of disease severity with genotype and phenotype in adult familial Mediterranean fever patients.

METHODS

Two-hundred seventy-five patients included in the study were divided into four groups according to their mutations: Group 1, M694V homozygous; Group 2, M694V-other; Group 3, other-other; and Group 4, no meaningful gene variants. Disease severity was evaluated using the Pras disease severity score. The association between Pras scores and other possible predictors was assessed by the multiple linear regression analysis.

RESULTS

In this study, 12.4% of all patients were in Group 1, 55.3% were in Group 2, 26.5% were in Group 3, and 5.8% were in Group 4. Pras scores were higher in Group 1 than in Groups 2, 3, and 4 (post hoc pairwise comparisons; P = .001, P < .001, and P = .001, respectively). Age at disease onset and age at diagnosis were found moderately and strongly correlated with Pras scores. Patients with moderate and severe disease were intensely involved in Group 1.

CONCLUSIONS

Higher Pras scores, earlier age of symptoms and diagnosis, more frequent arthritis and erysipelas-like erythema, and higher colchicine dose are closely associated with M694V homozygous familial Mediterranean fever patients. These patients also have mostly moderate and severe disease severity.

摘要

目的

评估成年家族性地中海热患者疾病严重程度与基因型和表型的关系。

方法

根据突变情况,将 275 例纳入研究的患者分为 4 组:组 1,M694V 纯合子;组 2,M694V-其他;组 3,其他-其他;组 4,无意义基因变异。采用 Pras 疾病严重程度评分评估疾病严重程度。采用多元线性回归分析评估 Pras 评分与其他可能预测因素之间的关系。

结果

本研究中,所有患者的 12.4%为组 1,55.3%为组 2,26.5%为组 3,5.8%为组 4。组 1 的 Pras 评分高于组 2、组 3 和组 4(事后两两比较;P = .001,P < .001 和 P = .001)。疾病发病年龄和诊断年龄与 Pras 评分呈中度和高度相关。中重度疾病患者主要集中在组 1。

结论

更高的 Pras 评分、更早的症状和诊断年龄、更频繁的关节炎和丹毒样红斑、更高的秋水仙碱剂量与 M694V 纯合子家族性地中海热患者密切相关。这些患者的疾病严重程度也大多为中重度。

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