Division of Nephrology, Department of Medicine, Kyoto Prefectural University of Medicine, 465 Kajii-cho, Kamigyo-ku, Kyoto, 602-8566, Japan.
Division of Surgical Pathology, Department of Pathology, Kyoto Prefectural University of Medicine, Kyoto, Japan.
CEN Case Rep. 2022 Feb;11(1):90-96. doi: 10.1007/s13730-021-00635-5. Epub 2021 Aug 13.
A 75-year-old man with fever was diagnosed with alveolar hemorrhage. Antineutrophil cytoplasmic antibodies for myeloperoxidase and proteinase 3 were absent. He received corticosteroid therapy, which immediately improved his symptoms and chest radiological findings. After the discontinuation of corticosteroids, fever and general fatigue relapsed, and renal function deteriorated with hematuria and proteinuria. A nerve conduction study revealed mononeuritis multiplex. Renal biopsy demonstrated focal necrotizing crescentic glomerulonephritis with endocapillary proliferative lesions, immunofluorescence C3 deposits, and electron-microscopic subepithelial hump-like deposits. Nephritis-associated plasmin receptor (NAPlr) and plasmin activity, biomarkers of infection-related glomerulonephritis, were positive in glomeruli. Although pathological findings suggested infection-related glomerulonephritis (IRGN), clinical manifestations, such as alveolar hemorrhage and mononeuritis multiplex, suggested systemic small vessel vasculitis. After corticosteroid therapy, systemic symptoms disappeared, and the gradual amelioration of hematuria and proteinuria was observed. Based on the clinical symptoms for which steroid therapy was effective, the patient was considered to have systemic small vessel vasculitis, the etiology of which may have been associated with infection.
一位 75 岁发热男性患者被诊断为肺泡出血。髓过氧化物酶和蛋白酶 3 的抗中性粒细胞胞浆抗体阴性。他接受了皮质类固醇治疗,症状和胸部影像学表现立即得到改善。皮质类固醇停药后,发热和全身乏力再次出现,肾功能恶化,出现血尿和蛋白尿。神经传导研究显示多发性单神经炎。肾活检显示局灶坏死性新月体性肾小球肾炎,伴有毛细血管内增生病变,免疫荧光 C3 沉积和电子显微镜下上皮下驼峰样沉积物。肾炎相关纤溶酶受体 (NAPlr) 和纤溶酶活性,感染相关肾小球肾炎的生物标志物,在肾小球中呈阳性。尽管病理发现提示感染相关肾小球肾炎 (IRGN),但肺泡出血和多发性单神经炎等临床表现提示系统性小血管血管炎。皮质类固醇治疗后,全身症状消失,血尿和蛋白尿逐渐改善。基于对类固醇治疗有效的临床症状,考虑该患者患有系统性小血管血管炎,其病因可能与感染有关。