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以双侧输尿管狭窄伴肾积水为首发表现的肉芽肿性多血管炎(韦格纳肉芽肿):一例报告并文献复习

Bilateral Ureteral Stenosis with Hydronephrosis as First Manifestation of Granulomatosis with Polyangiitis (Wegener's Granulomatosis): A Case Report and Review of the Literature.

作者信息

Suillot Joelle, Bollmann Jürg, Rotman Samuel, Descombes Eric

机构信息

Service of Nephrology, HFR Cantonal Hospital Fribourg, Fribourg, Switzerland.

University of Fribourg, Fribourg, Switzerland.

出版信息

Case Rep Nephrol. 2020 Dec 21;2020:7189497. doi: 10.1155/2020/7189497. eCollection 2020.

Abstract

Ureteral stenosis is a rare manifestation of granulomatosis with polyangiitis (formerly known as Wegener's granulomatosis). We report the case of a 76-year-old woman with progressive renal failure in which bilateral hydronephrosis due to ureteral stenosis was the first manifestation of the disease. Our patient also had renal involvement with pauci-immune crescentic glomerulonephritis associated with high titers of anti-proteinase 3 c-ANCAs, but no involvement of the upper or lower respiratory tract. The hydronephrosis and renal function rapidly improved under immunosuppressive therapy with high-dose corticosteroids and intravenous pulse cyclophosphamide. We reviewed the literature and found only ten other reported cases of granulomatosis with polyangiitis/Wegener's granulomatosis and intrinsic ureteral stenosis: in two cases, the presenting clinical manifestation was unilateral hydronephrosis and in only two others was the hydronephrosis bilateral, but this complication developed during a relapse of the disease. This case emphasizes the importance of including ANCA-related vasculitis in the differential diagnosis of unusual cases of unilateral or bilateral ureteral stenosis.

摘要

输尿管狭窄是肉芽肿性多血管炎(以前称为韦格纳肉芽肿)的一种罕见表现。我们报告了一例76岁女性进行性肾衰竭病例,其中因输尿管狭窄导致的双侧肾积水是该疾病的首发表现。我们的患者还存在肾脏受累,表现为寡免疫性新月体性肾小球肾炎,伴有高滴度的抗蛋白酶3胞浆型抗中性粒细胞胞浆抗体(c-ANCAs),但上下呼吸道均未受累。在高剂量皮质类固醇和静脉脉冲环磷酰胺免疫抑制治疗下,肾积水和肾功能迅速改善。我们回顾了文献,仅发现其他十例肉芽肿性多血管炎/韦格纳肉芽肿合并输尿管固有狭窄的报道病例:其中两例的临床表现为单侧肾积水,另外只有两例为双侧肾积水,但这种并发症是在疾病复发期间出现的。该病例强调了在单侧或双侧输尿管狭窄异常病例的鉴别诊断中纳入抗中性粒细胞胞浆抗体相关性血管炎的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3fdb/7773467/52692e0364ea/CRIN2020-7189497.001.jpg

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