Institute for Neural Computation, University of California, San Diego, 9500 Gilman Dr, La Jolla, CA 92093, United States of America.
Department of Neurological Sciences, Rush University Medical Center, 1620 W Harrison St, Chicago, IL 60612, United States of America.
J Neurol Sci. 2020 Dec 15;419:117205. doi: 10.1016/j.jns.2020.117205. Epub 2020 Nov 1.
To investigate hypothesized sources of error when quantifying the effect of the sensory trick in cervical dystonia (CD) with the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS-2), test strategies to mitigate them, and provide guidance for future research on the sensory trick.
Previous analyses suggested the sensory trick (or "alleviating maneuver", AM) item be removed from the TWSTRS-2 because of its poor clinimetric properties. We hypothesized three sources of clinimetric weakness for rating the AM: 1) whether patients were given sufficient time to demonstrate their AM; 2) whether patients' CD was sufficiently severe for detecting AM efficacy; and 3) whether raters were inadvertently rating the item in reverse of scale instructions. We tested these hypotheses with video recordings and TWSTRS-2 ratings by one "site rater" and a panel of five "video raters" for each of 185 Dystonia Coalition patients with isolated CD.
Of 185 patients, 23 (12%) were not permitted sufficient testing time to exhibit an AM, 23 (12%) had baseline CD too mild to allow confident rating of AM effect, and 1 site- and 1 video-rater each rated the AM item with a reverse scoring convention. When these confounds were eliminated in step-wise fashion, the item's clinimetric properties improved.
The AM's efficacy can contribute to measuring CD motor severity by addressing identified sources of error during its assessment and rating. Given the AM's sensitive diagnostic and potential pathophysiologic significance, we also provide guidance on modifications to how AMs can be assessed in future CD research.
研究量化感觉技巧对颈肌张力障碍(CD)的影响时,多伦多西部痉挛性斜颈评定量表(TWSTRS-2)假设的误差源,测试减轻这些误差的策略,并为未来感觉技巧的研究提供指导。
先前的分析表明,由于感觉技巧(或“缓解动作”,AM)项目的临床计量特性较差,应从 TWSTRS-2 中删除。我们假设了三种对 AM 进行评分的临床计量弱点来源:1)是否给予患者足够的时间来展示他们的 AM;2)患者的 CD 是否足够严重以检测 AM 的疗效;以及 3)评估者是否无意中以与量表说明相反的方式对项目进行评分。我们通过一位“站点评估者”和五位“视频评估者”对 185 名患有孤立性 CD 的痉挛性斜颈患者的视频记录和 TWSTRS-2 评分进行了测试。
在 185 名患者中,有 23 名(12%)没有足够的测试时间来展示 AM,有 23 名(12%)基线 CD 太轻,无法对 AM 效果进行有信心的评分,有 1 名站点评估者和 1 名视频评估者以相反的评分方式对 AM 项目进行了评分。当以逐步方式消除这些混杂因素时,该项目的临床计量特性得到了改善。
通过在评估和评分过程中解决 AM 评估的已确定误差源,AM 的疗效可以有助于测量 CD 运动严重程度。鉴于 AM 的敏感诊断和潜在病理生理意义,我们还提供了有关如何在未来 CD 研究中评估 AM 的修改建议。