Jonathan K Lai , José Luis López-Guillén , Rae SM Yeung , Mamoru Ayusawa , Brian W McCrindle , Anita Nagy
{"title":"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","authors":"Jonathan K Lai , José Luis López-Guillén , Rae SM Yeung , Mamoru Ayusawa , Brian W McCrindle , Anita Nagy","doi":"10.1016/j.carpath.2025.107766","DOIUrl":null,"url":null,"abstract":"<div><div><strong>Kawasaki disease (KD)</strong> 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.</div></div>","PeriodicalId":9451,"journal":{"name":"Cardiovascular Pathology","volume":"79 ","pages":"Article 107766"},"PeriodicalIF":1.9000,"publicationDate":"2025-08-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Fatal rupture of a giant left coronary artery aneurysm in an infant with Kawasaki disease. A case report with systematic literature review\",\"authors\":\"Jonathan K Lai , José Luis López-Guillén , Rae SM Yeung , Mamoru Ayusawa , Brian W McCrindle , Anita Nagy\",\"doi\":\"10.1016/j.carpath.2025.107766\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><div><strong>Kawasaki disease (KD)</strong> 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.</div></div>\",\"PeriodicalId\":9451,\"journal\":{\"name\":\"Cardiovascular Pathology\",\"volume\":\"79 \",\"pages\":\"Article 107766\"},\"PeriodicalIF\":1.9000,\"publicationDate\":\"2025-08-18\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Cardiovascular Pathology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S1054880725000511\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"CARDIAC & CARDIOVASCULAR SYSTEMS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Cardiovascular Pathology","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S1054880725000511","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"CARDIAC & CARDIOVASCULAR SYSTEMS","Score":null,"Total":0}
Fatal rupture of a giant left coronary artery aneurysm in an infant with Kawasaki disease. A case report with systematic literature review
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.
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.