肾脏受累的VEXAS综合征:从一个罕见的继发性淀粉样变病例和肾活检证实报告的系统回顾的见解。

IF 2.9 3区 医学 Q2 RHEUMATOLOGY
Gabriel Ştefan, Andreea Niculescu, Simona Cinca, Corina Chiriac, Adrian Zugravu, Pavel Cristina, Razvan Adrian Ionescu, Valer Mihai Pompilian, Nicoleta Petre, Cristina Căpusa, Simona Stancu
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引用次数: 0

摘要

VEXAS综合征是最近发现的一种由UBA1基因体细胞突变引起的自身炎症性疾病,可导致全身炎症和血液系统异常。虽然其对肾脏的影响仍然知之甚少,但报告的病例表明肾脏病理的多样性。我们报告一位69岁男性,有全身性炎症史,并发肾病综合征及肾功能恶化。他的病程包括反复发热、肺部浸润、皮肤血管炎和血液学异常。肾活检显示继发性淀粉样变,提示慢性炎症。基因检测证实了UBA1突变(c.121A > G, p.Met41Val),确定了VEXAS综合征的诊断。尽管给予皮质类固醇、环孢素和白细胞介素-1阻断治疗,患者病情恶化并最终死于感染性休克。对23例VEXAS综合征活检证实的肾脏受累进行系统回顾,其中间质性肾炎是最常见的组织病理学发现,其次是血管炎、IgA肾病、微小病变和淀粉样变性。治疗反应各不相同,免疫抑制疗法的疗效有限。本病例强调了对VEXAS综合征的未被认识的肾脏表现,表明其诊断挑战和需要加强临床怀疑。需要进一步的研究来确定最佳的管理策略并改善患者的预后。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Kidney involvement in VEXAS syndrome: insights from a rare case of secondary amyloidosis and systematic review of renal biopsy-confirmed reports.

VEXAS syndrome is a recently identified autoinflammatory disorder caused by somatic mutations in the UBA1 gene, leading to systemic inflammation and hematologic abnormalities. While its renal involvement remains poorly understood, reported cases suggest a diverse spectrum of kidney pathology. We present a 69-year-old male with a history of systemic inflammation who developed nephrotic syndrome and worsening kidney function. His disease course included recurrent fevers, pulmonary infiltrates, cutaneous vasculitis, and hematologic abnormalities. A kidney biopsy revealed secondary amyloidosis, indicating chronic inflammation. Genetic testing confirmed a UBA1 mutation (c.121A > G, p.Met41Val), establishing the diagnosis of VEXAS syndrome. Despite treatment with corticosteroids, cyclosporine, and interleukin-1 blockade, the patient deteriorated and ultimately succumbed to septic shock. A systematic review of biopsy-confirmed renal involvement in VEXAS syndrome identified 23 cases, with interstitial nephritis as the most frequent histopathologic finding, followed by vasculitis, IgA nephropathy, minimal change disease, and amyloidosis. Treatment responses varied, with limited efficacy of immunosuppressive therapies. This case highlights the under-recognized renal manifestations of VEXAS syndrome, demonstrating its diagnostic challenges and the need for heightened clinical suspicion. Further research is required to define optimal management strategies and improve patient outcomes.

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来源期刊
Clinical Rheumatology
Clinical Rheumatology 医学-风湿病学
CiteScore
6.90
自引率
2.90%
发文量
441
审稿时长
3 months
期刊介绍: Clinical Rheumatology is an international English-language journal devoted to publishing original clinical investigation and research in the general field of rheumatology with accent on clinical aspects at postgraduate level. The journal succeeds Acta Rheumatologica Belgica, originally founded in 1945 as the official journal of the Belgian Rheumatology Society. Clinical Rheumatology aims to cover all modern trends in clinical and experimental research as well as the management and evaluation of diagnostic and treatment procedures connected with the inflammatory, immunologic, metabolic, genetic and degenerative soft and hard connective tissue diseases.
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