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菊池-藤本病后出现的脑干脑炎作为一种非典型神经系统并发症

Brainstem Encephalitis as an Atypical Neurologic Complication Following Kikuchi-Fujimoto Disease.

作者信息

Tan Youjiang, Chuah Tyngyu

机构信息

Department of Neurology, Singapore General Hospital, Singapore.

Department of Rheumatology, Sengkang General Hospital, Singapore.

出版信息

Case Rep Neurol Med. 2025 Jul 14;2025:3086387. doi: 10.1155/crnm/3086387. eCollection 2025.

DOI:10.1155/crnm/3086387
PMID:40693116
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12279417/
Abstract

We report an uncommon and peculiar case of a patient who developed brainstem encephalitis between three and four months after recovering from an episode of Kikuchi-Fujimoto disease (KFD). She presented acutely with oscillopsia and persistent irrepressible hiccups, for which brainstem stroke was initially suspected. Brain magnetic resonance imaging was negative for ischemic strokes but demonstrated an enhancing T2-hyperintense lesion within the area postrema of the medulla oblongata extending into the upper cervical cord. Workup for infections etiologies and demyelinating disorders of the central nervous system including neuromyelitis optica, multiple sclerosis, and antimyelin oligodendrocyte glycoprotein antibody disease, were unremarkable. Prior to the administration of immunosuppressive treatment, she spontaneously and rapidly recovered, remaining well over a 3-year period of follow-up. We reviewed prevailing scientific literature and identified similar, albeit rare, cases of encephalitis which were attributed to KFD, which we added to our discussion.

摘要

我们报告了一例罕见且特殊的病例,一名患者在菊池-藤本病(KFD)发作康复后三到四个月间发生了脑干脑炎。她急性起病,出现视振荡和持续性无法抑制的呃逆,最初怀疑为脑干卒中。脑部磁共振成像显示无缺血性卒中,但在延髓最后区发现一个T2高信号强化病灶,延伸至上颈髓。对包括视神经脊髓炎、多发性硬化和抗髓鞘少突胶质细胞糖蛋白抗体病在内的中枢神经系统感染病因和脱髓鞘疾病的检查均无异常。在给予免疫抑制治疗之前,她自发且迅速康复,在3年的随访期间一直状况良好。我们查阅了现有科学文献,发现了类似的、虽罕见但归因于KFD的脑炎病例,并将其纳入我们的讨论。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fe81/12279417/0a483934062e/CRINM2025-3086387.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fe81/12279417/0a483934062e/CRINM2025-3086387.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fe81/12279417/0a483934062e/CRINM2025-3086387.001.jpg

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本文引用的文献

1
Kikuchi-Fujimoto Disease Presenting as Autoimmune Limbic Encephalitis: A Case Report with Review of Literature.以自身免疫性边缘叶脑炎形式表现的菊池-藤本病:一例病例报告并文献复习
Ann Indian Acad Neurol. 2025 Jan 1;28(1):116-119. doi: 10.4103/aian.aian_498_24. Epub 2024 Oct 8.
2
An atypical neurologic complication of Kikuchi-Fujimoto Disease.菊池-藤本病的一种非典型神经并发症。
Neurol Neuroimmunol Neuroinflamm. 2020 Mar 16;7(3). doi: 10.1212/NXI.0000000000000707. Print 2020 May.
3
Three children of meningoencephalitis with Kikuchi necrotizing lymphadenitis.
三名患有脑膜脑炎合并菊池坏死性淋巴结炎的儿童。
Brain Dev. 2018 Mar;40(3):251-255. doi: 10.1016/j.braindev.2017.09.009. Epub 2017 Oct 16.
4
Kikuchi-Fujimoto disease presenting as brainstem encephalitis with secondary blepharospasm.以脑干脑炎伴继发性眼睑痉挛为表现的菊池-藤本病。
J Neurosci Rural Pract. 2016 Jan-Mar;7(1):157-60. doi: 10.4103/0976-3147.165395.
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Clinical outcome and predictive factors of recurrence among patients with Kikuchi's disease.菊池病患者的临床结局及复发的预测因素
Int J Infect Dis. 2009 May;13(3):322-6. doi: 10.1016/j.ijid.2008.06.022. Epub 2009 Feb 8.
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Brainstem encephalitis with Kikuchi-Fujimoto disease.伴菊池-藤本病的脑干脑炎
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