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一名2个月大男婴患非典型川崎病并伴有巨大冠状动脉瘤:病例报告

Atypical Kawasaki disease with giant coronary artery aneurysms in a 2-month-old boy: a case report.

作者信息

Navidi Najmeh, Najibi Babak, Dinarvand Negar, Zamani Amirmohammad, Fathi Mohammad Reza

机构信息

Department of Pediatrics, Faculty of Medicine, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran.

Hyperlipidemia Research Center, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran.

出版信息

J Med Case Rep. 2024 Dec 20;18(1):630. doi: 10.1186/s13256-024-04987-1.

DOI:10.1186/s13256-024-04987-1
PMID:39707546
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11660496/
Abstract

BACKGROUND

Kawasaki disease is an acute inflammatory disorder primarily affecting medium-sized blood vessels, especially in infants and young children under 5 years old, characterized by inflammation of the arterial walls, including coronary arteries. While predominantly affecting young children, rarely Kawasaki disease is seen in adults. Reporting this case is crucial due to the patient's very young age and the disease's unusual presentation following vaccination, contributing to the limited literature on Kawasaki disease in very young infants.

CASE PRESENTATION

A full-term, 2-month-and-10-day-old Iranian boy presented with persistent fever unresponsive to treatment, beginning 5 days after receiving the 5-in-1 vaccine. Although physical examination results were normal, his medical history included maternal hypothyroidism during pregnancy and neonatal jaundice that did not require hospitalization. Initial laboratory tests revealed bicytopenia, thrombocytosis on the complete blood count, and elevated erythrocyte sedimentation rate and C-reactive protein levels. Echocardiography identified giant coronary artery aneurysms. The patient was diagnosed with atypical Kawasaki disease and treated with intravenous immunoglobulin, methylprednisolone, infliximab, and aspirin, which resolved his fever.

CONCLUSIONS

This case emphasizes the importance of considering Kawasaki disease as a differential diagnosis in infants with persistent, treatment-resistant fever and abnormal complete blood count findings. Early diagnosis and timely intervention are essential to prevent severe complications, such as coronary artery aneurysms and possible mortality. It underscores the need for heightened awareness and vigilance among healthcare providers in similar clinical scenarios.

摘要

背景

川崎病是一种主要影响中等大小血管的急性炎症性疾病,尤其多见于5岁以下的婴幼儿,其特征为动脉壁炎症,包括冠状动脉。虽然川崎病主要影响幼儿,但在成人中罕见。报告该病例至关重要,因为患者年龄极小,且疾病在接种疫苗后有不寻常表现,这有助于补充关于极小婴儿川崎病的有限文献。

病例介绍

一名足月出生、2个月零10天大的伊朗男婴,在接种五联疫苗5天后开始出现持续发热,对治疗无反应。尽管体格检查结果正常,但其病史包括母亲孕期甲状腺功能减退和新生儿黄疸,但无需住院治疗。初步实验室检查显示全血细胞计数有双细胞减少、血小板增多,红细胞沉降率和C反应蛋白水平升高。超声心动图检查发现巨大冠状动脉瘤。该患者被诊断为非典型川崎病,并接受静脉注射免疫球蛋白、甲泼尼龙、英夫利昔单抗和阿司匹林治疗,发热症状得以缓解。

结论

该病例强调了在持续发热且治疗无效、全血细胞计数异常的婴儿中,将川崎病作为鉴别诊断的重要性。早期诊断和及时干预对于预防严重并发症(如冠状动脉瘤和可能的死亡)至关重要。它强调了医疗保健提供者在类似临床情况下提高认识和警惕的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5a01/11660496/1aa01a72a443/13256_2024_4987_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5a01/11660496/1aa01a72a443/13256_2024_4987_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5a01/11660496/1aa01a72a443/13256_2024_4987_Fig1_HTML.jpg

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本文引用的文献

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Atypical Kawasaki Disease Complicated With Coronary Artery Aneurysm: A Case Report and Review of Literature.非典型川崎病合并冠状动脉瘤:一例报告并文献复习
Cureus. 2021 Feb 27;13(2):e13589. doi: 10.7759/cureus.13589.
2
Kawasaki Disease Complicated With Macrophage Activation Syndrome: Case Reports and Literature Review.川崎病合并巨噬细胞活化综合征:病例报告及文献综述
Front Pediatr. 2019 Nov 1;7:423. doi: 10.3389/fped.2019.00423. eCollection 2019.
3
Kawasaki disease and 13-valent pneumococcal conjugate vaccination among young children: A self-controlled risk interval and cohort study with null results.
川崎病与小儿 13 价肺炎球菌结合疫苗接种:一项无显著结果的自身对照风险间隔和队列研究。
PLoS Med. 2019 Jul 2;16(7):e1002844. doi: 10.1371/journal.pmed.1002844. eCollection 2019 Jul.
4
Kawasaki disease shock syndrome complicated with macrophage activation syndrome in a 5-month old boy: A case report.一名5个月大男童患川崎病休克综合征并发巨噬细胞活化综合征:病例报告
Medicine (Baltimore). 2019 Jan;98(4):e14203. doi: 10.1097/MD.0000000000014203.
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A 3-month-old infant with atypical Kawasaki disease.一名患有非典型川崎病的3个月大婴儿。
BMJ Case Rep. 2018 May 30;2018:bcr-2017-221456. doi: 10.1136/bcr-2017-221456.
6
Kawasaki Disease Complicated With Macrophage Activation Syndrome: A Systematic Review.川崎病合并巨噬细胞活化综合征:一项系统评价
J Pediatr Hematol Oncol. 2017 Aug;39(6):445-451. doi: 10.1097/MPH.0000000000000872.
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Diagnosis, Treatment, and Long-Term Management of Kawasaki Disease: A Scientific Statement for Health Professionals From the American Heart Association.川崎病的诊断、治疗和长期管理:美国心脏协会发布的一份面向医疗保健专业人员的科学声明。
Circulation. 2017 Apr 25;135(17):e927-e999. doi: 10.1161/CIR.0000000000000484. Epub 2017 Mar 29.
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The incidence of Kawasaki disease after vaccination within the UK pre-school National Immunisation Programme: an observational THIN database study.英国学龄前儿童国家免疫规划疫苗接种后川崎病的发病率:一项基于THIN数据库的观察性研究
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