Lau Richard A, Bains Ramandeep, Suraweera Duminda, Ma Jane, Heinze Emil R, Wong Andrew L, Clements Philip J
UCLA-Olive View Rheumatology Program, Division of Rheumatology, Olive View-UCLA Medical Center, 14445 Olive View Drive, 2B182, Sylmar, CA 91342, USA.
UCLA-Olive View Internal Medicine Program, Department of Medicine, Olive View-UCLA Medical Center, 14445 Olive View Drive, 2B182, Sylmar, CA 91342, USA.
Case Rep Rheumatol. 2017;2017:2421760. doi: 10.1155/2017/2421760. Epub 2017 Feb 28.
This paper describes one patient with Antineutrophil Cytoplasmic Antibody- (ANCA-) associated vasculitis who initially presented with multiple ischemic fingers and toes. On further evaluation, the patient was also found to have pulmonary-renal involvement and episcleritis. The diagnosis was supported with a positive cANCA (anti-proteinase 3) and a bronchoscopy consistent with diffuse alveolar hemorrhage. Although the patient refused a tissue biopsy, clinical presentation including nasal ulceration, sinus congestion, and epistaxis and anti-proteinase 3 antibody were more consistent with Granulomatosis with Polyangiitis (GPA) rather than Microscopic Polyangiitis (MPA) or Eosinophilic Granulomatosis with Polyangiitis (EGPA) based on the recently presented ACR/EULAR Provisional 2017 Classification Criteria for GPA (Luqmani et al., 2016). The patient responded well to therapy including high dose steroids and cyclophosphamide, with improvement of all organs involved and had no further digital ischemia or gangrene on follow-up. We include a review of the English literature summarizing presentation, management, and outcome of 16 similar cases.
本文描述了一名抗中性粒细胞胞浆抗体(ANCA)相关性血管炎患者,该患者最初表现为多个手指和脚趾缺血。进一步评估发现,该患者还存在肺肾受累及巩膜外层炎。cANCA(抗蛋白酶3)阳性及支气管镜检查结果符合弥漫性肺泡出血,支持了诊断。尽管患者拒绝组织活检,但基于最近发布的2017年美国风湿病学会/欧洲抗风湿病联盟GPA临时分类标准(Luqmani等人,2016年),包括鼻溃疡、鼻窦充血和鼻出血在内的临床表现以及抗蛋白酶3抗体,更符合肉芽肿性多血管炎(GPA)而非显微镜下多血管炎(MPA)或嗜酸性肉芽肿性多血管炎(EGPA)。该患者对包括大剂量类固醇和环磷酰胺在内的治疗反应良好,所有受累器官均有改善,随访期间未出现进一步的手指缺血或坏疽。我们对英文文献进行了综述,总结了16例类似病例的临床表现、治疗及结局。