Department of Internal Medicine (IV), Osaka Medical College, 2-7 Daigaku-machi, Takatsuki, Osaka, 569-8686, Japan.
Rheumatol Int. 2018 Apr;38(4):689-696. doi: 10.1007/s00296-017-3875-2. Epub 2017 Nov 10.
Eosinophilic granulomatosis with polyangiitis (EGPA) is characterized by necrotizing vasculitis of small-sized vessels with extravascular granulomas and eosinophilic infiltration. The case of a 48-year-old Japanese woman with EGPA, who presented concurrently with subarachnoid hemorrhage (SAH) and coronary vasculitis, is reported. She initially presented with bronchial asthma, and then 8 months later she developed various symptoms caused by systemic eosinophilic vasculitis and was admitted to our hospital. Three days after admission, she started oral corticosteroid therapy, and her 2009 Five-Factor Score (FFS) was 0. However, she developed an SAH, followed by coronary vasculitis 1 day later. With extensive treatment with a combination of betamethasone, cyclophosphamide, intravenous immunoglobulin, and rituximab, her systemic vasculitis improved dramatically. This seems to be the first case of EGPA with SAH and coronary vasculitis. In previous reports of EGPA with SAH, 4 of 11 cases developed SAH as an exacerbation of systemic vasculitis during remission induction therapy. The present patient also had SAH during remission induction therapy. However, the period between bronchial asthma and SAH was only 8 months. This is the shortest among case reports of EGPA with SAH. In addition, the present patient rapidly developed coronary vasculitis. These findings suggest that EGPA causes SAH and coronary vasculitis as early complications of systemic vasculitis. In EGPA, it is necessary to pay careful attention to rapid changes of disease activity, even when the FFS indicates a good prognosis.
嗜酸性肉芽肿性多血管炎(EGPA)的特征为小血管坏死性血管炎、血管外肉芽肿和嗜酸性粒细胞浸润。本文报告了一例同时并发蛛网膜下腔出血(SAH)和冠状动脉血管炎的 48 岁日本女性 EGPA 病例。该患者最初表现为支气管哮喘,8 个月后出现各种系统性嗜酸性血管炎症状而被收入我院。入院 3 天后开始口服皮质类固醇治疗,2009 年 Five-Factor 评分(FFS)为 0。但随后她发生了 SAH,1 天后又出现了冠状动脉血管炎。经过广泛治疗,联合使用倍他米松、环磷酰胺、静脉注射免疫球蛋白和利妥昔单抗后,她的系统性血管炎得到了显著改善。这似乎是首例 EGPA 合并 SAH 和冠状动脉血管炎的病例。在之前报道的 EGPA 合并 SAH 中,11 例中有 4 例在缓解诱导治疗期间全身性血管炎恶化时发生了 SAH。本患者也在缓解诱导治疗期间发生了 SAH。但从支气管哮喘到 SAH 的时间仅为 8 个月。这是 EGPA 合并 SAH 病例报告中最短的时间间隔。此外,本患者迅速发生了冠状动脉血管炎。这些发现表明,EGPA 导致 SAH 和冠状动脉血管炎作为全身性血管炎的早期并发症。在 EGPA 中,即使 FFS 提示预后良好,也需要密切关注疾病活动的快速变化。