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VEXAS综合征中的肾脏受累:一例罕见的继发性淀粉样变性病例及肾活检确诊报告的系统评价启示

Kidney involvement in VEXAS syndrome: insights from a rare case of secondary amyloidosis and systematic review of renal biopsy-confirmed reports.

作者信息

Ştefan Gabriel, Niculescu Andreea, Cinca Simona, Chiriac Corina, Zugravu Adrian, Cristina Pavel, Ionescu Razvan Adrian, Pompilian Valer Mihai, Petre Nicoleta, Căpusa Cristina, Stancu Simona

机构信息

University of Medicine and Pharmacy "Carol Davila", Bucharest, Romania.

"Dr. Carol Davila" Teaching Hospital of Nephrology, Bucharest, Romania.

出版信息

Clin Rheumatol. 2025 Jul;44(7):3101-3108. doi: 10.1007/s10067-025-07506-9. Epub 2025 May 30.

DOI:10.1007/s10067-025-07506-9
PMID:40445522
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12234580/
Abstract

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.

摘要

VEXAS综合征是一种最近发现的自身炎症性疾病,由UBA1基因的体细胞突变引起,导致全身炎症和血液学异常。虽然其肾脏受累情况仍知之甚少,但报告的病例显示肾脏病理表现多样。我们报告一名69岁男性,有全身炎症病史,出现肾病综合征且肾功能恶化。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。肾脏活检显示继发性淀粉样变性,提示慢性炎症。基因检测证实存在UBA1突变(c.121A > G,p.Met41Val),从而确诊为VEXAS综合征。尽管使用了皮质类固醇、环孢素和白细胞介素-1阻断剂进行治疗,患者病情仍恶化,最终死于感染性休克。一项对活检证实的VEXAS综合征肾脏受累情况的系统综述确定了23例病例,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、微小病变病和淀粉样变性。治疗反应各不相同,免疫抑制疗法的疗效有限。该病例突出了VEXAS综合征未被充分认识的肾脏表现,展示了其诊断挑战以及提高临床怀疑度的必要性。需要进一步研究来确定最佳管理策略并改善患者预后。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ff83/12234580/7c7b5dd0144c/10067_2025_7506_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ff83/12234580/7c7b5dd0144c/10067_2025_7506_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ff83/12234580/7c7b5dd0144c/10067_2025_7506_Fig1_HTML.jpg

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本文引用的文献

1
A Clinicopathological Description of Kidney Features in VEXAS Syndrome.VEXAS综合征肾脏特征的临床病理描述
Kidney Int Rep. 2024 Oct 28;10(1):260-264. doi: 10.1016/j.ekir.2024.10.026. eCollection 2025 Jan.
2
VEXAS syndrome as a mimicker of ANCA-associated vasculitis.VEXAS综合征作为抗中性粒细胞胞浆抗体相关性血管炎的模仿者。
Rheumatol Adv Pract. 2024 Sep 25;8(4):rkae116. doi: 10.1093/rap/rkae116. eCollection 2024.
3
Acute kidney injury, an underrecognized feature of VEXAS syndrome.急性肾损伤,一种VEXAS综合征未被充分认识的特征。
Rheumatology (Oxford). 2025 Apr 1;64(4):2027-2033. doi: 10.1093/rheumatology/keae465.
4
Antineutrophil cytoplasmic antibody-associated vasculitis and VEXAS syndrome: comment on the article by Muratore et al.抗中性粒细胞胞浆抗体相关性血管炎与VEXAS综合征:对穆拉托雷等人文章的评论
Arthritis Rheumatol. 2023 Aug;75(8):1490-1492. doi: 10.1002/art.42466. Epub 2023 May 17.
5
VEXAS syndrome: a new paradigm for adult-onset monogenic autoinflammatory diseases.VEXAS 综合征:一种新的成人发病的单基因自身炎症性疾病模式。
Intern Emerg Med. 2023 Apr;18(3):711-722. doi: 10.1007/s11739-023-03193-z. Epub 2023 Jan 20.
6
VEXAS Syndrome-A Review of Pathophysiology, Presentation, and Prognosis.VEXAS 综合征——发病机制、临床表现和预后的综述。
J Clin Rheumatol. 2023 Sep 1;29(6):298-306. doi: 10.1097/RHU.0000000000001905. Epub 2022 Oct 17.
7
Acute tubulointerstitial nephritis revealing VEXAS syndrome.急性肾小管间质性肾炎揭示VEXAS综合征。
Kidney Int. 2022 Jun;101(6):1295-1297. doi: 10.1016/j.kint.2022.03.012.
8
Disorders of ubiquitylation: unchained inflammation.泛素化紊乱:脱链的炎症。
Nat Rev Rheumatol. 2022 Aug;18(8):435-447. doi: 10.1038/s41584-022-00778-4. Epub 2022 May 6.
9
Distinction between clonal and paraclonal cutaneous involvements in VEXAS syndrome.VEXAS综合征中克隆性和副克隆性皮肤受累的区分
Exp Hematol Oncol. 2022 Feb 16;11(1):6. doi: 10.1186/s40164-022-00262-5.
10
Thrombosis in VEXAS syndrome.VEXAS 综合征中的血栓形成。
J Thromb Thrombolysis. 2022 May;53(4):965-970. doi: 10.1007/s11239-021-02608-y. Epub 2021 Nov 24.