Palha Vanessa, Sa Ana, Pimentel Teresa, Oliveira Narciso, Rocha Sofia, Silva Ana Isabel, Capela Carlos
Department of Internal Medicine, Hospital de Braga, Braga, Portugal.
Department of Nephrology, Hospital de Braga, Braga, Portugal.
J Med Cases. 2021 Oct;12(10):405-410. doi: 10.14740/jmc3742. Epub 2021 Sep 29.
Anti-glomerular basement membrane (anti-GBM) disease and anti-neutrophil cytoplasm antibody (ANCA)-associated vasculitis are the main causes of pulmonary-renal syndrome (PRS). The concurrence of both ANCA - myeloperoxidase (MPO) and proteinase 3 (PR3) - and anti-GBM antibodies has been described, although positivity for all three antibodies has rarely been reported. The natural history of triple-positive patients as well as the best therapeutic approach remains unknown. We describe a case of an 80-year-old woman that presented to the emergency department with a 3-month history of progressive fatigue, malaise and anorexia, and 5 weeks of cough with blood-streaked sputum and progressive peripheral edema. Through the complementary study, a rare diagnosis of PRS with triple-seropositive for both ANCA (MPO and PR3) and anti-GBM antibodies was made in a patient with untreated chronic hepatitis B virus infection. She was treated with glucocorticoid, cyclophosphamide, plasma exchange and entecavir, with pulmonary recovery. Renal function did not improve. After 2 years, the patient is still in dialysis, but did not have relapse of alveolar hemorrhage and ANCA and anti-GBM antibody titers remain negative. The authors intend to warn to PRS, in particular this rare cause, since delaying diagnosis can lead to significant morbidity and mortality for patients.
抗肾小球基底膜(anti-GBM)病和抗中性粒细胞胞浆抗体(ANCA)相关性血管炎是肺肾综合征(PRS)的主要病因。虽然三种抗体均呈阳性的情况鲜有报道,但ANCA(髓过氧化物酶(MPO)和蛋白酶3(PR3))与抗GBM抗体同时存在的情况已有描述。三阳性患者的自然病史以及最佳治疗方法仍不清楚。我们描述了一例80岁女性患者,她因进行性疲劳、不适和厌食3个月,咳嗽伴痰中带血及进行性外周水肿5周就诊于急诊科。通过辅助检查,在一名未经治疗的慢性乙型肝炎病毒感染患者中诊断出罕见的PRS,其ANCA(MPO和PR3)及抗GBM抗体均呈三阳性。她接受了糖皮质激素、环磷酰胺、血浆置换和恩替卡韦治疗,肺部得以恢复。肾功能未改善。两年后,患者仍在进行透析,但未出现肺泡出血复发,ANCA和抗GBM抗体滴度仍为阴性。作者旨在提醒注意PRS,尤其是这种罕见病因,因为延迟诊断可能导致患者出现严重的发病率和死亡率。