Williams Maegan, Nagaraju Lakshmi, Gorelik Mark
Division of General Pediatrics, Children's Hospital of San Antonio, Baylor College of Medicine, 333 N Santa Rosa St, San Antonio, TX, USA.
Division of Cardiology, Children's Hospital of San Antonio, Baylor College of Medicine, 333 N Santa Rosa St, San Antonio, TX, USA.
Case Rep Rheumatol. 2020 Mar 28;2020:6249013. doi: 10.1155/2020/6249013. eCollection 2020.
Kawasaki disease (KD) is an inflammatory vasculitis and is the most common cause of acquired childhood heart disease in developed countries. Current treatment with intravenous immunoglobulin (IVIG) is often ineffective in patients with delayed or refractory disease. We present a case of combination anticytokine therapy in an infant with delayed and refractory KD. . A 3-month-old infant presented with refractory KD with giant aneurysms after a delayed diagnosis of one month. Use of combination anticytokine therapy led to resolution of giant aneurysms over approximately 6 months.
Our case is unique in effective use of anticytokine therapy in very delayed disease with giant aneurysms. Additionally, we review other cases for a broader perspective. Prospective study of anticytokine therapy for patients with giant aneurysms may be warranted.
川崎病（KD）是一种炎症性血管炎，是发达国家儿童后天性心脏病最常见的病因。目前静脉注射免疫球蛋白（IVIG）治疗对疾病延迟或难治的患者往往无效。我们报告一例对延迟和难治性KD婴儿采用联合抗细胞因子治疗的病例。一名3个月大的婴儿在延迟诊断1个月后出现伴有巨大动脉瘤的难治性KD。联合抗细胞因子治疗使巨大动脉瘤在大约6个月内消退。
我们的病例在对伴有巨大动脉瘤的极延迟疾病有效使用抗细胞因子治疗方面具有独特性。此外，我们从更广泛的角度回顾了其他病例。对伴有巨大动脉瘤的患者进行抗细胞因子治疗的前瞻性研究可能是必要的。
