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儿童免疫球蛋白A血管炎:地中海热基因变异对临床病程和炎症标志物的影响。

Pediatric Immunoglobulin A Vasculitis: Impact of Mediterranean Fever Gene Variants on Clinical Course and Inflammatory Markers.

作者信息

Tunce Eray, Turunç Salih, Atamyıldız Uçar Sıla, Sözeri Betül

机构信息

Department of Pediatric Rheumatology, Ümraniye Training and Research Hospital, University of Health Sciences, İstanbul, Türkiye.

Department of Pediatrics, Ümraniye Training and Research Hospital, University of Health Sciences, İstanbul, Türkiye.

出版信息

Turk Arch Pediatr. 2025 Jun 30;60(4):412-418. doi: 10.5152/TurkArchPediatr.2025.25129.

Abstract

Objective: The aim of the study is to evaluate the clinical and laboratory features of pediatric patients with immunoglobulin A vasculitis (IgAV) according to Mediterranean fever (MEFV) gene variant status and to identify possible clinical indicators that may guide selective genetic testing. Materials and Methods: A total of 176 pediatric IgAV patients who underwent MEFV gene analysis were retrospectively reviewed. Patients were classified into 2 groups based on the presence or absence of MEFV variants. Demographic, clinical, and laboratory parameters assessed at diagnosis were compared between groups. Results: Among the 176 patients, 75 (42.6%) carried at least 1 MEFV variant. Compared to those without MEFV variants, patients with MEFV variants had a significantly higher rate of family history of Familial Mediterranean fever (FMF) (42.6% vs. 5.9%, P < .001). In addition, arthralgia (76% vs. 58%, P= .02) and arthritis (36% vs. 20.8%, P= .03) were more frequently observed in patients with MEFV variants. Serum amyloid A levels were significantly elevated in patients with MEFV variants (median 15 [IQR 4-150] vs. 5 [2-33] mg/L, P < .001). No significant differences were observed in renal or gastrointestinal involvement between groups. C-reactive protein and erythrocyte sedimentation rate values were higher in patients with MEFV variants but did not reach statistical significance. Conclusion: The presence of MEFV variant in pediatric IgAV patients is associated with increased systemic inflammation and higher rates of musculoskeletal symptoms. Selective MEFV genetic testing and SAA measurement may be beneficial in IgAV patients presenting with marked inflammatory responses, joint involvement, or positive family history of FMF.

摘要

目的

本研究旨在根据地中海热(MEFV)基因变异状态评估儿童免疫球蛋白A血管炎(IgAV)患者的临床和实验室特征,并确定可能指导选择性基因检测的临床指标。材料与方法:回顾性分析176例接受MEFV基因分析的儿童IgAV患者。根据是否存在MEFV变异将患者分为两组。比较两组在诊断时评估的人口统计学、临床和实验室参数。结果:176例患者中,75例(42.6%)携带至少1种MEFV变异。与无MEFV变异的患者相比,有MEFV变异的患者家族性地中海热(FMF)家族史发生率显著更高(42.6%对5.9%,P <.001)。此外,有MEFV变异的患者关节痛(76%对58%,P =.02)和关节炎(36%对20.8%,P =.03)更为常见。有MEFV变异的患者血清淀粉样蛋白A水平显著升高(中位数15 [四分位间距4 - 150]对5 [2 - 33] mg/L,P <.001)。两组在肾脏或胃肠道受累方面未观察到显著差异。有MEFV变异的患者C反应蛋白和红细胞沉降率值较高,但未达到统计学意义。结论:儿童IgAV患者中MEFV变异的存在与全身炎症增加和肌肉骨骼症状发生率较高相关。对于出现明显炎症反应、关节受累或FMF家族史阳性的IgAV患者,选择性MEFV基因检测和血清淀粉样蛋白A测量可能有益。

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本文引用的文献

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Turk Arch Pediatr. 2024 Nov 1;59(6):527-534. doi: 10.5152/TurkArchPediatr.2024.24188.
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Vasculitis in Children.儿童血管炎
Turk Arch Pediatr. 2024 Nov 1;59(6):517-526. doi: 10.5152/TurkArchPediatr.2024.24181.
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Turk Arch Pediatr. 2021 Nov;56(6):610-617. doi: 10.5152/TurkArchPediatr.2021.21147.
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Rheumatology (Oxford). 2020 May 1;59(Suppl 3):iii95-iii100. doi: 10.1093/rheumatology/kez599.
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Classification criteria for autoinflammatory recurrent fevers.自身炎症性反复发作性发热的分类标准。
Ann Rheum Dis. 2019 Aug;78(8):1025-1032. doi: 10.1136/annrheumdis-2019-215048. Epub 2019 Apr 24.

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