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用阿那白滞素治疗小儿重症急性心肌心包炎:一例报告及文献综述

Treatment of pediatric severe acute myopericarditis with anakinra: a case report and literature review.

作者信息

Licciardi Francesco, Fornari Federico, Ferroni Francesca, Covizzi Carlotta, Riggi Chiara, Montin Davide

机构信息

Department of Pediatrics and Public Health, Università Degli Studi di Torino, Turin, Italy.

Immunorheumatology Unit, Ospedale Infantile Regina Margherita, Città Della Salute e Della Scienza, Turin, Italy.

出版信息

Front Pediatr. 2025 Apr 23;13:1544126. doi: 10.3389/fped.2025.1544126. eCollection 2025.

Abstract

Acute myocarditis (AM) is an inflammation of the myocardium with a rapid onset of typically <1 month. The use of anakinra (ANK) for treating inflammatory AM in adults has been recently described; however, while some reports are promising, its efficacy remains debated. Here, we present a case of severe AM with concomitant systemic symptoms [fever, elevated C-reactive protein (CRP)] in a pediatric patient who was successfully treated with high-dose ANK. A literature review of similar published cases is also presented. A 14-year-old boy was admitted for AM with concomitant pericarditis. At disease onset, the patient presented with high fever and elevated CRP (163 mg/L) and troponin I (14,816 ng/L). Treatment with ibuprofen (30 mg/kg/day), intravenous immunoglobulin (80 g in 24 h), and colchicine (0.5 mg per day) were initiated without benefit and with further worsening of contractile function [Ejection Fraction (EF) 26%]. Consequently, inotropic support and intravenous methylprednisolone were started, leading to a partial improvement of EF (45%). Due to the inability to reduce inotropic support, a rescue treatment with ANK (7 mg/kg/day) in continuous intravenous infusion was started, resulting in progressive improvement and normalization of left ventricular systolic function. Our literature review identified five case reports of pediatric AM successfully treated with ANK. Most cases presented elevated inflammatory markers (ferritin and CRP) and/or concomitant pericarditis. We conclude that ANK, especially at high doses, may be useful for treating severe pediatric AM, particularly when associated with severe inflammation and/or pericarditis.

摘要

急性心肌炎(AM)是一种心肌炎症,起病迅速,通常病程小于1个月。最近有关于使用阿那白滞素(ANK)治疗成人炎症性AM的报道;然而,尽管一些报道显示出前景,但其疗效仍存在争议。在此,我们报告一例患有严重AM并伴有全身症状(发热、C反应蛋白(CRP)升高)的儿科患者,该患者通过高剂量ANK成功治愈。同时还对已发表的类似病例进行了文献综述。一名14岁男孩因AM合并心包炎入院。疾病发作时,患者出现高热,CRP(163mg/L)和肌钙蛋白I(14816ng/L)升高。开始使用布洛芬(30mg/kg/天)、静脉注射免疫球蛋白(24小时内80g)和秋水仙碱(每天0.5mg)治疗,但无效且收缩功能进一步恶化[射血分数(EF)26%]。因此,开始使用强心支持治疗和静脉注射甲泼尼龙,使EF部分改善(45%)。由于无法减少强心支持治疗,开始持续静脉输注ANK(7mg/kg/天)进行抢救治疗,结果左心室收缩功能逐渐改善并恢复正常。我们的文献综述确定了五例用ANK成功治疗的儿科AM病例报告。大多数病例表现为炎症标志物(铁蛋白和CRP)升高和/或合并心包炎。我们得出结论,ANK,尤其是高剂量时,可能对治疗严重儿科AM有用,特别是当伴有严重炎症和/或心包炎时。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a43d/12055804/f345a917bb3b/fped-13-1544126-g001.jpg

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