Suppr超能文献

多血管炎重叠综合征:一种罕见的临床实体。

Polyangiitis overlap syndrome: a rare clinical entity.

机构信息

Rheumatology, Department of Medicine and Surgery, University of Perugia, Piazzale Giorgio Menghini, 1, 06129, Perugia, PG, Italy.

Section of Anatomic Pathology and Histology, Department of Medicine and Surgery, University of Perugia, Perugia, Italy.

出版信息

Rheumatol Int. 2023 Mar;43(3):537-543. doi: 10.1007/s00296-023-05281-x. Epub 2023 Jan 31.

Abstract

Polyangiitis overlap syndrome is a rare clinical entity comprising patients with overlapping features of more than one vasculitis, usually eosinophilic granulomatosis with polyangiitis (EGPA) and granulomatosis with polyangiitis (GPA). Few cases of polyangiitis overlap syndrome have been described in the literature, mostly associated with c-ANCA, anti-proteinase (PR)-3 positivity, a protean clinical picture characterized by vasculitis, eosinophilia and eosinophilic infiltrates in tissues and a favorable response to steroids and immunosuppressant treatments. Herein, we present a case of a 66-year-old woman with nasal obstruction, external nose deformity, sensorineural hearing loss, peripheral blood eosinophilia, high titer anti-PR3 antibodies and lung involvement. Nasal septum biopsies showed inflammatory infiltrate with eosinophilic component; histopathology of the lung demonstrated necrotizing granulomas associated with inflammatory infiltrate composed of numerous neutrophils and some eosinophils. The patient was diagnosed with polyangiitis overlap syndrome and successfully treated with cyclophosphamide. Recognizing this entity is fundamental given the distinct clinical phenotype and outcomes to therapy in the complex scenario of ANCA-associated vasculitides.

摘要

巨细胞动脉炎重叠综合征是一种罕见的临床实体,其患者具有多种血管炎的重叠特征,通常为嗜酸性肉芽肿性多血管炎(EGPA)和肉芽肿性多血管炎(GPA)。文献中描述了少数巨细胞动脉炎重叠综合征病例,这些病例大多与 c-ANCA、抗蛋白酶(PR)-3 阳性、表现多样的临床特征有关,包括血管炎、嗜酸性粒细胞增多和组织中嗜酸性粒细胞浸润,对类固醇和免疫抑制剂治疗有良好的反应。在此,我们报告一例 66 岁女性,表现为鼻塞、外鼻畸形、感音神经性听力损失、外周血嗜酸性粒细胞增多、高滴度抗 PR3 抗体和肺部受累。鼻中隔活检显示炎症浸润伴嗜酸性粒细胞成分;肺组织病理学显示与炎症浸润相关的坏死性肉芽肿,炎症浸润由大量中性粒细胞和一些嗜酸性粒细胞组成。该患者被诊断为巨细胞动脉炎重叠综合征,并成功接受环磷酰胺治疗。鉴于在 ANCA 相关性血管炎的复杂情况下,该疾病具有独特的临床表型和治疗结果,因此识别该疾病至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/38fa/9968253/92ccf0da81c3/296_2023_5281_Fig1_HTML.jpg

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验