神经源性反常声带运动：中枢和周围神经系统疾病的系统综述

文献信息

中文摘要

目的： 系统综合已发表的关于与确诊的中枢或周围神经系统疾病相关的反常声带运动的证据，重点关注神经解剖学相关性、临床表现、管理策略和治疗结果。
方法： 按照PRISMA 2020指南进行系统综述，并在PROSPERO注册。检索了六个数据库（PubMed/MEDLINE、Embase、Scopus、Cochrane Library、ScienceDirect和Google Scholar），从建库至2025年12月。纳入报告任何年龄患者中与确诊神经系统疾病相关的PVFM的研究。符合条件的设计包括病例报告、病例系列和观察性研究。使用预定义标准进行数据提取，并使用Joanna Briggs Institute（JBI）批判性评估工具和ROBINS-I框架评估偏倚风险。
结果： 纳入11项研究，共102名患者，其中42名（19名儿童，23名成人）患有与确诊神经系统疾病相关的反常声带运动。神经系统病因包括脑干畸形、癫痫、脑瘫、神经肌肉接头疾病、基底节疾病、神经退行性疾病和离子通道病。本综述设计无法估计所有PVFM病例中神经系统原因的患病率；在个别队列中，54%的婴儿PVFM患者被识别出神经系统疾病，33%的多系统萎缩患者记录到反常声带运动。在9项可分类病因的研究中，6项涉及可逆或可治疗的情况，3项涉及进行性神经退行性疾病，尽管大多数个体患者属于后一组，因为神经退行性队列更大。针对潜在神经系统疾病的治疗在大多数可逆病例中导致显著改善或缓解，而在进行性疾病中症状通常持续。
结论： PVFM常与器质性神经系统疾病相关，应被视为神经控制 disrupted 的表现，而不仅仅是功能性疾病。早期神经系统评估和疾病特异性管理可能改善结果，特别是在非典型或难治性表现中。

英文摘要

OBJECTIVE(S): To systematically synthesize published evidence on paradoxical vocal fold motion associated with confirmed central or peripheral neurological disorders, with emphasis on neuroanatomic correlations, clinical presentation, management strategies, and treatment outcomes.
METHODS: A systematic review was conducted following PRISMA 2020 guidelines and registered in PROSPERO. Six databases (PubMed/MEDLINE, Embase, Scopus, Cochrane Library, ScienceDirect, and Google Scholar) were searched from inception to December 2025. Studies reporting PVFM associated with a confirmed neurological disorder in patients of any age were included. Eligible designs comprised case reports, case series, and observational studies. Data extraction was performed using predefined criteria, and risk of bias was assessed using Joanna Briggs Institute (JBI) critical appraisal tools and the ROBINS-I framework.
RESULTS: Eleven studies were included, enrolling 102 patients, of whom 42 (19 pediatric, 23 adult) had paradoxical vocal fold motion in association with a confirmed neurological disorder. Neurological etiologies encompassed brainstem malformations, epilepsy, cerebral palsy, neuromuscular junction disorders, basal ganglia diseases, neurodegenerative disorders, and ion-channelopathies. The review design precluded estimation of the prevalence of neurological causes among all PVFM cases; within individual cohorts, neurological disease was identified in 54% of infants with PVFM, and paradoxical vocal fold motion was documented in 33% of patients with multiple system atrophy. Six of the nine studies with classifiable etiologies involved reversible or treatable conditions and three involved progressive neurodegenerative disease, although most individual patients fell into the latter group because the neurodegenerative cohorts were larger. Treatment directed at the underlying neurological disorder resulted in substantial improvement or resolution in most reversible cases, whereas symptoms often persisted in progressive diseases.
CONCLUSION: PVFM is frequently associated with organic neurological disorders and should be recognized as a manifestation of disrupted neural control rather than solely a functional condition. Early neurological evaluation and disease-specific management may improve outcomes, particularly in atypical or refractory presentations.