Fatal rupture of a giant left coronary artery aneurysm in an infant with Kawasaki disease. A case report with systematic literature review

IF 1.9 4区 医学 Q2 CARDIAC & CARDIOVASCULAR SYSTEMS
Jonathan K Lai , José Luis López-Guillén , Rae SM Yeung , Mamoru Ayusawa , Brian W McCrindle , Anita Nagy
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引用次数: 0

Abstract

Kawasaki disease (KD) is a systemic vasculitis of childhood that may lead to coronary artery aneurysms. Rupture of a giant coronary aneurysm is an exceptionally rare but often fatal complication. We report the case of a 4-month-old infant with treatment-resistant Kawasaki disease who developed rapidly enlarging giant aneurysms in all coronary artery branches despite aggressive immunomodulatory therapy. The patient died from cardiac tamponade due to rupture of a giant left anterior descending coronary artery aneurysm. Full autopsy revealed widespread vasculitis affecting multiple visceral arteries in addition to coronary artery involvement. Histological analysis demonstrated panarteritis with medial destruction and no evidence of thrombosis or myocardial infarction. A systematic review of reported cases demonstrated comparable clinical patterns and outcomes. This report underscores the potential for systemic vascular involvement in severe Kawasaki disease and highlights the importance of early recognition and intensified treatment in refractory cases.
一名患有川崎病的婴儿巨大左冠状动脉瘤破裂致死性。一例报告并系统文献复习
川崎病是一种儿童期全身性血管炎,可导致冠状动脉瘤。巨大冠状动脉瘤破裂是一种非常罕见但往往致命的并发症。我们报告一例4个月大的婴儿患有治疗抵抗性川崎病,尽管积极的免疫调节治疗,但在所有冠状动脉分支中发展迅速扩大的巨大动脉瘤。病人死于巨大的左冠状动脉前降动脉瘤破裂引起的心包填塞。全面尸检显示广泛的血管炎,除了冠状动脉受累外,还影响到多个内脏动脉。组织学分析显示泛动脉炎伴内侧破坏,无血栓形成或心肌梗死的证据。对报告病例的系统回顾显示了可比较的临床模式和结果。本报告强调了严重川崎病累及全身血管的可能性,并强调了对难治性病例早期识别和强化治疗的重要性。
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来源期刊
Cardiovascular Pathology
Cardiovascular Pathology 医学-病理学
CiteScore
7.50
自引率
2.70%
发文量
71
审稿时长
18 days
期刊介绍: Cardiovascular Pathology is a bimonthly journal that presents articles on topics covering the entire spectrum of cardiovascular disease. The Journal''s primary objective is to publish papers on disease-oriented morphology and pathogenesis from clinicians and scientists in the cardiovascular field. Subjects covered include cardiovascular biology, prosthetic devices, molecular biology and experimental models of cardiovascular disease.
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