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颈部肌张力障碍中的震颤

Tremor in cervical dystonia.

作者信息

Beylergil Sinem Balta, Mukunda Krishna Nikhil, Elkasaby Mohamed, Perlmutter Joel S, Factor Stewart, Bäumer Tobias, Feurestein Jeanne, Shelton Erika, Bellows Steven, Jankovic Joseph, Mahajan Abhimanyu, Wamer-Rosen Tila, Reich Stephen G, Shukla Aparna Wagle, Malaty Irene, Espay Alberto, Duque Kevin, LeDoux Mark S, Saunders-Pullman Rachel, Leaver Katherine, Frank Samuel, Pantelyat Alexander, Fung Victor, Richardson Sarah Pirio, Berman Brian, Stover Natividad, Deik Andres, Ondo William, Groth Christopher, Jinnah Hyder A, Shaikh Aasef G

机构信息

Department of Neurology, Case Western Reserve University, Cleveland, OH, United States.

Department of Neurology and Neurologic Surgery, Washington University School of Medicine, St. Louis, MO, United States.

出版信息

Dystonia. 2024;3. doi: 10.3389/dyst.2024.11309. Epub 2024 Mar 21.

Abstract

BACKGROUND

Cervical dystonia (CD) is the most common form of focal dystonia encountered in the clinic. Approximately one-third of CD patients have co-existing tremor in the head and hands. Assessment of tremor as regular or irregular in context of its oscillation trajectory, frequency, and amplitude is a major clinical challenge and can confound the diagnosis of CD. The misdiagnosis may lead to therapeutic failures, poor quality of life, and poor utilization of medical and financial resources.

METHODS

We analyzed the largest cohort of CD patients ( = 3117) available to date, collected from 37 movement disorder centers in North America, Europe, and Asia. We used machine learning to determine what clinical features from clinician reports predicted the presence of tremor as well as its regular or irregular appearance.

RESULTS

Out of 3,117 CD patients, 1,367 had neck tremor. The neck tremor was interpreted as irregular in 1,022, regular in 345, and mixed (both irregular and regular) in 442. A feature importance analysis determined that greater severity of CD, longer disease duration, and older age, in descending order, predicted the presence of neck tremor. The probability of neck tremor was reduced if the dystonia affected other body parts in addition to the neck. We also found a significantly heightened risk for developing neck tremor in women. An additional feature importance analysis indicated that increased severity of dystonia affecting other body parts, severity of CD, and prolonged disease duration was associated with a lower likelihood of regular neck tremor while increased age predicted a higher likelihood.

CONCLUSION

Machine learning recognized the most relevant clinical features that can predict concurrent neck tremor and its irregularity in a large multi-center dystonia cohort. These results may facilitate a more accurate description of neck tremor and improved care path in CD.

摘要

背景

颈部肌张力障碍(CD)是临床上最常见的局灶性肌张力障碍形式。大约三分之一的CD患者头部和手部同时存在震颤。根据震颤的振荡轨迹、频率和幅度评估震颤是规则还是不规则是一项重大临床挑战,可能会混淆CD的诊断。误诊可能导致治疗失败、生活质量低下以及医疗和财政资源利用不佳。

方法

我们分析了迄今为止最大的CD患者队列(n = 3117),这些患者来自北美、欧洲和亚洲的37个运动障碍中心。我们使用机器学习来确定临床医生报告中的哪些临床特征可以预测震颤的存在及其规则或不规则表现。

结果

在3117例CD患者中,1367例有颈部震颤。其中1022例颈部震颤被判定为不规则,345例为规则,442例为混合(不规则和规则兼有)。特征重要性分析确定,CD病情严重程度越高、病程越长和年龄越大(按降序排列),预测颈部震颤存在的可能性越大。如果肌张力障碍除颈部外还累及其他身体部位,则颈部震颤的可能性降低。我们还发现女性发生颈部震颤的风险显著增加。另一项特征重要性分析表明,影响其他身体部位的肌张力障碍严重程度增加、CD严重程度和病程延长与规则颈部震颤的可能性降低相关,而年龄增加则预测可能性更高。

结论

机器学习识别出了在一个大型多中心肌张力障碍队列中可预测并发颈部震颤及其不规则性的最相关临床特征。这些结果可能有助于更准确地描述颈部震颤并改善CD的护理路径。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b567/11542928/6327be17e831/nihms-2022735-f0001.jpg

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