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以声带麻痹为首发症状的重症肌无力,发生在带状疱疹疫苗接种后一周:一例病例报告及文献复习

Myasthenia Gravis Presenting With Vocal Cord Paralysis As the Initial Symptom Occurring One Week Following Herpes Zoster Vaccination: A Case Report and Review of the Literature.

作者信息

Asada Shimpei, Odera Hikaru, Morishita Koji, Mori Shusuke

机构信息

Department of Critical Care and Emergency Medicine, Tokyo Women's Medical University, Tokyo, JPN.

Department of Acute Critical Care and Disaster Medicine, Institute of Science Tokyo, Tokyo, JPN.

出版信息

Cureus. 2025 May 4;17(5):e83454. doi: 10.7759/cureus.83454. eCollection 2025 May.

DOI:10.7759/cureus.83454
PMID:40462824
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12132085/
Abstract

Myasthenia gravis (MG) is an autoimmune neuromuscular junction disorder primarily characterized by fluctuating skeletal muscle weakness. While it typically presents with ocular or generalized symptoms, vocal cord paralysis (VCP) as an initial manifestation is exceedingly rare and often leads to diagnostic delays. We report the case of an 86-year-old Japanese woman who developed progressive hoarseness, dysphagia, and respiratory distress approximately one week after receiving the inactivated herpes zoster vaccine (Shingrix®). Initial evaluation revealed unilateral VCP, which progressed to bilateral paralysis, requiring emergency intubation and subsequent tracheostomy. Despite extensive imaging and laboratory investigations, the etiology remained unclear until the patient developed diplopia and ptosis around hospital day 25. Although both anti-acetylcholine receptor (AChR) and anti-muscle-specific kinase (MuSK) antibodies were negative, the diagnosis of MG was confirmed through positive edrophonium and ice pack tests. Treatment included pyridostigmine, plasma exchange, and immunosuppressive therapy with tacrolimus and prednisolone. While ptosis improved, bilateral VCP and diplopia persisted at one year, and the patient remained tracheostomy-dependent but functionally independent. This case adds to the limited literature on laryngeal-type MG, a focal subtype marked by bulbar symptoms and often negative for AChR antibodies. Our review of post-2000 literature identified 13 cases of MG with VCP, 10 of which presented with VCP initially. Notably, nine required airway management, underscoring the potential for rapid respiratory compromise. This is the first reported case of MG development temporally associated with herpes zoster vaccination. Although a definitive causal link cannot be established, the close temporal sequence - fever and systemic immune activation shortly after vaccination, followed by MG onset - raises the possibility of an immune-mediated trigger. Previous reports have described MG onset after various other vaccines, including HPV, HBV, influenza, and COVID-19, suggesting that vaccination may rarely unmask or precipitate MG in susceptible individuals. Nonetheless, large-scale studies have not demonstrated a significant increase in autoimmune disease following vaccination, supporting the overall safety of immunization programs. This case highlights the importance of recognizing atypical MG presentations such as isolated VCP, particularly in the context of recent immune stimulation. Early diagnosis and appropriate treatment are critical for improving outcomes and preventing life-threatening complications such as airway obstruction. Heightened clinical suspicion is warranted in similar scenarios to facilitate timely intervention.

摘要

重症肌无力(MG)是一种自身免疫性神经肌肉接头疾病,主要特征为骨骼肌无力波动。虽然其通常表现为眼部或全身性症状,但以声带麻痹(VCP)作为初始表现极为罕见,且常导致诊断延迟。我们报告一例86岁日本女性病例,该患者在接种灭活带状疱疹疫苗(Shingrix®)后约一周出现进行性声音嘶哑、吞咽困难和呼吸窘迫。初始评估发现单侧VCP,随后进展为双侧麻痹,需要紧急插管及后续气管切开术。尽管进行了广泛的影像学和实验室检查,但在患者住院第25天左右出现复视和上睑下垂之前,病因仍不明确。虽然抗乙酰胆碱受体(AChR)抗体和抗肌肉特异性激酶(MuSK)抗体均为阴性,但依酚氯铵试验和冰敷试验阳性确诊为MG。治疗包括吡啶斯的明、血浆置换以及使用他克莫司和泼尼松龙进行免疫抑制治疗。虽然上睑下垂有所改善,但双侧VCP和复视在1年后仍持续存在,患者仍依赖气管切开术,但功能上可独立生活。该病例补充了关于喉型MG的有限文献,喉型MG是一种以延髓症状为特征且AChR抗体常为阴性的局灶性亚型。我们对2000年后文献的回顾发现13例伴有VCP的MG病例,其中10例最初表现为VCP。值得注意的是,9例需要气道管理,这突出了快速呼吸功能不全的可能性。这是首例报告的与带状疱疹疫苗接种在时间上相关的MG病例。虽然无法确定明确的因果关系,但紧密的时间顺序——接种疫苗后不久出现发热和全身免疫激活,随后发生MG——增加了免疫介导触发因素的可能性。既往报告描述了在接种各种其他疫苗后发生MG,包括人乳头瘤病毒(HPV)疫苗、乙肝病毒(HBV)疫苗、流感疫苗和2019冠状病毒病(COVID - 19)疫苗,提示接种疫苗可能在易感个体中罕见地引发或促使MG发病。尽管如此,大规模研究并未证明接种疫苗后自身免疫性疾病显著增加,这支持了免疫接种计划的总体安全性。该病例强调了识别非典型MG表现(如孤立性VCP)的重要性,特别是在近期有免疫刺激的情况下。早期诊断和适当治疗对于改善预后及预防诸如气道梗阻等危及生命的并发症至关重要。在类似情况下应提高临床怀疑度以便及时干预。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1a0f/12132085/d44360ef325a/cureus-0017-00000083454-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1a0f/12132085/a14d27d7d6ea/cureus-0017-00000083454-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1a0f/12132085/d44360ef325a/cureus-0017-00000083454-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1a0f/12132085/a14d27d7d6ea/cureus-0017-00000083454-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1a0f/12132085/d44360ef325a/cureus-0017-00000083454-i02.jpg

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本文引用的文献

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Laryngeal Myasthenia Gravis in Voice Patients: Clinical, Serologic, and Neuromuscular Characterization of Seronegative Patients for Antibodies Against the Acetylcholine Receptor and Muscle-Specific Kinase.嗓音疾病患者中的喉重症肌无力:抗乙酰胆碱受体和肌肉特异性激酶抗体血清阴性患者的临床、血清学及神经肌肉特征
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Myasthenia gravis after the third dose of human papillomavirus 9-valent vaccine: A case report.九价人乳头瘤病毒疫苗接种第三针后出现重症肌无力:病例报告。
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重症肌无力性眼病:当前概述
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COVID-19 Vaccination and Late-Onset Myasthenia Gravis: A New Case Report and Review of the Literature.COVID-19 疫苗接种与迟发性重症肌无力:一例新病例报告及文献复习。
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