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川崎病并发双侧面神经麻痹:病例报告及文献复习。

Bilateral facial nerve palsy complicating Kawasaki disease: A case report and literature review.

机构信息

Department of Pediatrics, Chongqing University Three Gorges Hospital, Chongqing, China.

School of Medicine, Chongqing University, Chongqing, China.

出版信息

Medicine (Baltimore). 2024 Aug 16;103(33):e39389. doi: 10.1097/MD.0000000000039389.

DOI:10.1097/MD.0000000000039389
PMID:39151496
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11332778/
Abstract

RATIONALE

Kawasaki disease (KD) manifests as an acute, self-limited vasculitis disease that constitutes the primary cause of acquired heart disease in children under 5 years of age. Facial nerve palsy (FNP) is a rare complication associated with coronary artery lesions (CALs) in patients with KD. Patients with KD and FNP usually present atypically, leading to a delayed diagnosis and treatment of KD.

PATIENT CONCERNS

A 4-month-old boy with fever, left FNP and bilateral conjunctival injection with spontaneous resolution, was admitted to the hospital, received a short course of intravenous dexamethasone, and experienced rapid FNP recovery on the first admission. The patient experienced a resurgence of fever, bilateral conjunctival injection, and right FNP, which led to readmission. Physical examination revealed redness at the site of Bacillus Calmette-Guérin inoculation, reddening of lips, and desquamation of the distal extremities. Echocardiography revealed right-sided CALs.

DIAGNOSES

The patient initially missed KD on the first admission, and was later diagnosed with complete KD with FNP on the second admission.

INTERVENTIONS AND OUTCOMES

After a short course of intravenous dexamethasone, the left FNP resolved quickly. However, right FNP recurred after corticosteroids withdrawal. Meanwhile, more typical symptoms were observed, and KD was diagnosed. Treatment ensued with intravenous immunoglobulin (IVIG), aspirin, and dexamethasone. The patient achieved rapid remission, without recurrence. Echocardiography continued to show normal findings during 1-year follow-up after discharge.

LESSONS

The clinical symptoms of FNP complicating KD in children are atypical and can easily lead to delayed diagnosis and treatment. FNP in patients with KD may serve as a risk factor for CALs, which are more challenging to resolve than the FNP itself. Adding corticosteroids to IVIG may be recommended to reduce IVIG resistance, decrease the risk of developing CALs, and alleviate CALs.

摘要

背景

川崎病(KD)表现为一种急性、自限性血管炎疾病,是 5 岁以下儿童获得性心脏病的主要病因。面神经麻痹(FNP)是 KD 患者并发冠状动脉病变(CALs)的罕见并发症。KD 合并 FNP 的患者通常表现不典型,导致 KD 的诊断和治疗延迟。

病例介绍

一名 4 月龄男婴,发热,左侧 FNP,双侧结膜炎,自行缓解,入院后给予短程静脉注射地塞米松,首次入院 FNP 迅速恢复。患者再次发热,双侧结膜炎,右侧 FNP,再次入院。体格检查发现卡介苗接种部位发红,唇红,四肢远端脱皮。超声心动图显示右侧 CALs。

诊断

患儿首次入院时漏诊 KD,第二次入院时诊断为完全性 KD 合并 FNP。

干预和结果

静脉注射地塞米松后,左侧 FNP 迅速缓解。但糖皮质激素停药后右侧 FNP 复发。同时,出现更多典型症状,确诊为 KD。予静脉注射免疫球蛋白(IVIG)、阿司匹林和地塞米松治疗。患儿迅速缓解,无复发。出院后 1 年随访期间,超声心动图持续正常。

教训

儿童 KD 合并 FNP 的临床症状不典型,易导致诊断和治疗延迟。KD 合并 FNP 可能是 CALs 的危险因素,CALs 比 FNP 本身更难消退。KD 患儿加用地塞米松可能有助于减少 IVIG 抵抗,降低 CALs 风险,缓解 CALs。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ab1/11332778/dc5a3b8a6ee1/medi-103-e39389-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ab1/11332778/dc5a3b8a6ee1/medi-103-e39389-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4ab1/11332778/dc5a3b8a6ee1/medi-103-e39389-g001.jpg

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