Yu Ruohan, Zhang Lina, Long Ting, Gao Hui, Xu Jing, Zhang Tong, Li Shengguang
Department of Rheumatology and Immunology, Peking University International Hospital, Beijing, China.
Department of Pathology, Peking University International Hospital, Beijing, China.
Front Immunol. 2025 Jan 29;16:1544263. doi: 10.3389/fimmu.2025.1544263. eCollection 2025.
Anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) is a systemic necrotizing vasculitis that predominantly affects small vessels. In this report, we present a typical case of granulomatosis with polyangiitis (GPA) complicated by spontaneous renal hemorrhage (SRH), a rare but potentially severe condition. The patient developed SRH during immunosuppressive therapy but recovered following conservative treatment. We then conducted a systematic literature review on SRH in the context of AAV, and analyzed clinical features, management strategies, and patient prognosis. A total of 15 patients were enrolled for statistical analysis, comprising the one case reported in the current study and 14 from the literature. Of these patients, nine presented with GPA and six showed microscopic polyangiitis (MPA), with a sex distribution of 3:2 males to females. The average patient age was 54.5 years, and ranged from 25 to 82 years. Acute flank pain was the most common clinical manifestation, and was occasionally accompanied by anemia and shock. Treatment varied for the different patients. Eight patients received glucocorticoid and immunosuppressive agents that included rituximab, cyclophosphamide, and azathioprine; five patients underwent transcatheter arterial embolization (TAE); and one patient underwent nephrectomy. Our findings indicate that SRH typically occurs early in the course of AAV and correlates with disease activity, with renal aneurysm rupture as the primary cause. More than half of the patients respond well to corticosteroids and immunosuppressants. Timely TAE is essential for patients showing persistent deterioration despite conservative management.
抗中性粒细胞胞浆抗体(ANCA)相关性血管炎(AAV)是一种主要累及小血管的系统性坏死性血管炎。在本报告中,我们呈现了一例典型的肉芽肿性多血管炎(GPA)并发自发性肾出血(SRH)的病例,这是一种罕见但可能严重的病症。该患者在免疫抑制治疗期间发生了SRH,但经保守治疗后康复。然后,我们针对AAV背景下的SRH进行了系统的文献综述,并分析了临床特征、管理策略和患者预后。共有15例患者纳入统计分析,包括本研究报告的1例以及文献中的14例。在这些患者中,9例为GPA,6例为显微镜下多血管炎(MPA),男女比例为3:2。患者的平均年龄为54.5岁,范围在25至82岁之间。急性胁腹痛是最常见的临床表现,偶尔伴有贫血和休克。不同患者的治疗方法各异。8例患者接受了糖皮质激素和免疫抑制剂治疗,包括利妥昔单抗、环磷酰胺和硫唑嘌呤;5例患者接受了经导管动脉栓塞术(TAE);1例患者接受了肾切除术。我们的研究结果表明,SRH通常发生在AAV病程早期,与疾病活动相关,主要原因是肾动脉瘤破裂。超过半数的患者对皮质类固醇和免疫抑制剂反应良好。对于经保守治疗仍持续恶化的患者,及时进行TAE至关重要。