Lee Chang Woo, Krüger Marie T, Akram Harith, Zrinzo Ludvic, Yang Wanding, Hamilton Nick, Rubin John S, Birchall Martin A, Fishman Jonathan M
Department of Laryngology, National Voice Centre, Royal National ENT Hospital, University College London Hospitals NHS Foundation Trust, London, United Kingdom; UCL Division of Surgery and Interventional Science, London, United Kingdom.
Functional Neurosurgery Unit, National Hospital for Neurology and Neurosurgery, University College London Hospitals NHS Foundation Trust, London, United Kingdom; UCL Queen Square Institute of Neurology, London, United Kingdom.
J Voice. 2025 Mar 30. doi: 10.1016/j.jvoice.2025.03.017.
The aim of this study was to systematically review and evaluate neuromodulation therapies that are currently being explored/used in the treatment of laryngeal dystonia (LD).
A systematic review of the literature was performed following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines searching Embase, Medline, and Cochrane with the cover period January 1960-August 2024. The search strategy used was (("Laryngeal Dystonia" OR "Spasmodic Dysphonia") AND ("Neuromodulation" OR "Deep Brain Stimulation")). Articles were reviewed and screened based on the set inclusion and exclusion criteria. The Joanna Briggs Institute Critical Appraisal Checklists were used to assess the risk of bias in the included studies. Patient demographics and diagnosis, neuromodulation therapy used and its details, voice outcomes, and complications were extracted from each of the studies. A narrative review was subsequently synthesized.
Twelve studies met the selection criteria and were included. In total, 130 LD patients were identified with the following neuromodulation therapies being evaluated for their use in LD: vibro-tactile stimulation, laryngeal electrical stimulation, pharmacologic (sodium oxybate), repetitive transcranial magnetic stimulation, and deep brain stimulation (DBS). All included studies reported various degrees of improvement in patients' voice symptoms with the neuromodulation therapy used. No definitive conclusion could be drawn for their efficacy within the included studies consisting of six case reports, three proof-of-concept/pilot studies, and three phase I clinical trials that used a number of different voice outcome measures used to report treatment effects, and a general lack of long-term follow-up. No significant complications were reported for any of the neuromodulation therapies.
Most neuromodulation therapies being explored for LD are at a very early exploratory stage. Although more clinical trials are required, from the available evidence, sodium oxybate and DBS currently appear to have the most potential for translation into clinical practice.
本研究旨在系统回顾和评估目前正在探索/用于治疗喉肌张力障碍(LD)的神经调节疗法。
按照系统评价和Meta分析的首选报告项目(PRISMA)指南,对1960年1月至2024年8月期间的文献进行系统回顾,检索Embase、Medline和Cochrane数据库。使用的检索策略为((“喉肌张力障碍”或“痉挛性发声障碍”)且(“神经调节”或“深部脑刺激”))。根据设定的纳入和排除标准对文章进行审查和筛选。使用乔安娜·布里格斯研究所的批判性评价清单来评估纳入研究中的偏倚风险。从每项研究中提取患者人口统计学和诊断信息、所使用的神经调节疗法及其详细信息、语音结果和并发症。随后进行叙述性综述。
12项研究符合入选标准并被纳入。总共确定了130例LD患者,对以下用于LD治疗的神经调节疗法进行了评估:振动触觉刺激、喉部电刺激、药物治疗(羟丁酸钠)、重复经颅磁刺激和深部脑刺激(DBS)。所有纳入研究均报告,所使用的神经调节疗法使患者的语音症状有不同程度的改善。在所纳入的研究中,无法得出关于其疗效的确切结论,这些研究包括6例病例报告、3项概念验证/试点研究和3项I期临床试验,这些研究使用了多种不同的语音结果测量方法来报告治疗效果,并且普遍缺乏长期随访。未报告任何神经调节疗法有显著并发症。
大多数正在探索用于LD的神经调节疗法处于非常早期的探索阶段。尽管需要更多的临床试验,但从现有证据来看,羟丁酸钠和DBS目前似乎最有转化为临床实践的潜力。