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复发性川崎病:间隔超过4年出现三次独立发作的病例报告

Recurrent Kawasaki Disease: A Case Report of Three Separate Episodes at >4-Year Intervals.

作者信息

Goswami Nikita, Marzan Katherine, De Oliveira Elizabeth, Wagner-Lees Sharon, Szmuszkovicz Jacqueline

机构信息

CHLA Pediatric Rheumatology, Children's Hospital Los Angeles, 4650 Sunset Blvd, Mailstop #60, Los Angeles, CA 90027, USA.

Pacific Pediatric Cardiology, Pacific Pediatric Cardiology Medical Group, Pasadena, CA 91105, USA.

出版信息

Children (Basel). 2018 Nov 21;5(11):155. doi: 10.3390/children5110155.

DOI:10.3390/children5110155
PMID:30469337
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6262567/
Abstract

Kawasaki disease (KD) is a self-limited systemic vasculitis, most often occurring in children 1⁻5 years old. It has a 2% recurrence rate and is associated with coronary aneurysms (CA), which can develop within two weeks of onset. A 25% increased risk is noted in patients who are recalcitrant to treatment. We describe a patient with recurrence of KD three times, approximately four years apart. A 10-year-old female with two previous episodes of KD, at 11 months and five years of age), in which she met five out of five criteria for KD and had no coronary involvement, presented with 15 days of fever, conjunctivitis and mucocutaneous changes. Infectious work-up was negative, and she was diagnosed with incomplete KD meeting three out of five criteria. An echocardiogram (ECHO) on day 12 revealed dilation of the right coronary artery (RCA) and left coronary artery (LCA). Treatment with intravenous immunoglobulin (IVIG) and high-dose aspirin was started at an outside hospital. After transfer, serial ECHOs showed evolving coronary aneurysms, left anterior descending (LAD) z-score + 8.2 and RCA z-score + 4.0. She received 10 mg/kg infliximab (day 18) and began clopidogrel. A cardiac MRI (day 20) demonstrated progression of the LAD aneurysm, with a z-score + 13, and warfarin was started. To our knowledge, this is the first report of recurrent KD occurring three times at ~5 year intervals.

摘要

川崎病(KD)是一种自限性全身性血管炎,最常发生于1至5岁的儿童。其复发率为2%,并与冠状动脉瘤(CA)相关,冠状动脉瘤可在发病后两周内形成。治疗抵抗的患者发生冠状动脉瘤的风险增加25%。我们描述了一名KD复发三次的患者,复发间隔约为四年。一名10岁女性,曾在11个月和5岁时两次患KD,当时她符合KD的五项标准中的五项且无冠状动脉受累,此次出现发热、结膜炎和皮肤黏膜改变15天。感染相关检查结果为阴性,她被诊断为不完全KD,符合五项标准中的三项。第12天的超声心动图(ECHO)显示右冠状动脉(RCA)和左冠状动脉(LCA)扩张。在外院开始静脉注射免疫球蛋白(IVIG)和高剂量阿司匹林治疗。转诊后,系列ECHO显示冠状动脉瘤进展,左前降支(LAD)z值为+8.2,RCA z值为+4.0。她接受了10mg/kg英夫利昔单抗治疗(第18天)并开始使用氯吡格雷。心脏磁共振成像(第20天)显示LAD动脉瘤进展,z值为+13,并开始使用华法林。据我们所知,这是首次关于KD以约5年的间隔复发三次的报告。

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本文引用的文献

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Recurrence of Fever After Initial Intravenous Immunoglobulin Treatment in Children With Kawasaki Disease.川崎病患儿初次静脉注射免疫球蛋白治疗后发热复发
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Infliximab for intensification of primary therapy for Kawasaki disease: a phase 3 randomised, double-blind, placebo-controlled trial.英夫利昔单抗强化川崎病一线治疗:一项 3 期随机、双盲、安慰剂对照试验。
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