Caglayan Sengul, Yener Gulcin Otar, Ulu Kadir, Coskuner Taner, Guzel Meryem, Kalin Sevinc, Yazan Hakan, Erdogan Seher, Cakan Mustafa, Sozeri Betul
Department of Pediatric Rheumatology, University of Health Sciences, Umraniye Training and Research Hospital, Istanbul, Turkiye.
Department of Pediatric Rheumatology, Sanliurfa Training and Research Hospital, Sanliurfa, Turkiye.
North Clin Istanb. 2023 Jun 6;10(3):393-397. doi: 10.14744/nci.2022.01878. eCollection 2023.
Takayasu's arteritis (TA) is a rare chronic granulomatous vasculitis characterized by large-vessel involvement. The aorta and its main branches are most commonly involved. Although pulmonary artery involvement is common, hemoptysis or respiratory findings are rarely seen. Herein, we present a case of TA who developed anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis with diffuse alveolar hemorrhage after coronavirus disease 2019 (COVID-19) infection. A 17-year-old female patient with the diagnosis of TA presented with cough, bloody vomiting, and diarrhea. In follow-up, she developed tachypnea and dyspnea and was transferred to the pediatric intensive care unit. The findings on the chest computed tomography were compatible with acute COVID-19 infection, but the SARS-CoV2 reverse transcription-polymerase chain reaction test was negative, but SARS-CoV2 immunoglobulin (Ig) G and IgM antibody tests were positive. The patient was not vaccinated against COVID-19. The bronchoscopy showed bronchial mucosal fragility, bleeding foci, and mucosal bleeding. The broncoalveolar lavage hemosiderin-laden macrophages were seen in the histopathologic examination. The indirect immunofluorescence assay-ANCA test became 3 (+) with myeloperoxidase (MPO)-ANCA of 125 RU/ml (normal: <20). Cyclophosphamide and pulse steroid treatment were started. After immunosuppressive therapy, the patient condition improved and did not have hemoptysis again. The successful response was obtained by applying balloon angioplasty to the patient with bilateral renal artery stenosis. Types of post-COVID vasculitis include thromboembolic events, cutaneous vasculitis, Kawasaki-like vasculitis, myopericarditis, and ANCA-associated vasculitis. It is thought that COVID-19 may impair immune tolerance and trigger autoimmunity with cross-reaction. To the best of our knowledge, the third pediatric case was reported with MPO-ANCA-positive COVID-associated ANCA vasculitis.
高安动脉炎（TA）是一种罕见的慢性肉芽肿性血管炎，其特征为大血管受累。主动脉及其主要分支最常受累。虽然肺动脉受累常见，但咯血或呼吸系统表现罕见。在此，我们报告一例TA患者，在2019冠状病毒病（COVID-19）感染后发生抗中性粒细胞胞浆抗体（ANCA）相关血管炎并伴有弥漫性肺泡出血。一名诊断为TA的17岁女性患者出现咳嗽、血性呕吐和腹泻。在随访过程中，她出现呼吸急促和呼吸困难，并被转入儿科重症监护病房。胸部计算机断层扫描结果与急性COVID-19感染相符，但严重急性呼吸综合征冠状病毒2（SARS-CoV2）逆转录聚合酶链反应检测为阴性，而SARS-CoV2免疫球蛋白（Ig）G和IgM抗体检测为阳性。该患者未接种COVID-19疫苗。支气管镜检查显示支气管黏膜脆弱、出血灶和黏膜出血。组织病理学检查可见支气管肺泡灌洗中有含铁血黄素巨噬细胞。间接免疫荧光法ANCA检测髓过氧化物酶（MPO）-ANCA为125 RU/ml（正常：<20），呈3（+）。开始使用环磷酰胺和冲击性类固醇治疗。免疫抑制治疗后，患者病情改善，未再出现咯血。对双侧肾动脉狭窄患者进行球囊血管成形术取得了成功。COVID-19后血管炎的类型包括血栓栓塞事件、皮肤血管炎、川崎样血管炎、心肌心包炎和ANCA相关血管炎。据认为，COVID-19可能会损害免疫耐受并通过交叉反应触发自身免疫。据我们所知，这是第三例报告的MPO-ANCA阳性的COVID相关ANCA血管炎儿科病例。
