Gewily Mohamed, Plan Elodie L, Yousefi Elham, König Franz, Posch Martin, Hopfner Franziska, Höglinger Günter, Karlsson Mats O
Department of Pharmacy, Uppsala University, Uppsala, Sweden.
Center for Medical Data Science, Medical University of Vienna, Vienna, Austria.
Mov Disord. 2024 Dec;39(12):2181-2189. doi: 10.1002/mds.30001. Epub 2024 Aug 29.
Progressive supranuclear palsy (PSP) is a neurodegenerative, late-onset disease that is challenging in terms of assessment. The Progressive Supranuclear Palsy Rating Scale (PSPRS), a 28-item clinician-reported scale, is the most established clinical outcome assessment method. Recently, the U.S. Food and Drug Administration (FDA) has proposed a subscale of 10 items as an alternative to full PSPRS.
To quantitatively evaluate and compare the properties of full PSPRS and the FDA subscale using item response theory. To develop a progression model of the disease and assess relative merits of study designs and analysis options.
Data of 979 patients from four interventional trials and two registries were available for analysis. Our investigation was divided into: (1) estimating informativeness of the 28 items; (2) estimating disease progression; and (3) comparing the scales, trial designs, and analysis options with respect to power to detect a clinically relevant treatment effect.
PSPRS item scores had a low pairwise correlation (r = 0.17 ± 0.14) and the items irritability, sleep difficulty, and postural tremor were uncorrelated with the other items. The FDA-selected items displayed higher correlation (r = 0.35 ± 0.14) and were the basis for a longitudinal item response model including disease progression. Trial simulations indicated that identification of a disease-modifying treatment effect required less than half the study size if the analysis was based on longitudinal item information compared with total scores at end-of-treatment.
A longitudinal item response model based on the FDA-selected PSPRS items is a promising tool in evaluating treatments for PSP. © 2024 The Author(s). Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.
进行性核上性麻痹(PSP)是一种神经退行性迟发性疾病,评估具有挑战性。进行性核上性麻痹评定量表(PSPRS)是一个由临床医生报告的包含28个条目的量表,是最成熟的临床结局评估方法。最近,美国食品药品监督管理局(FDA)提出了一个包含10个条目的子量表作为完整PSPRS的替代方案。
使用项目反应理论定量评估和比较完整PSPRS与FDA子量表的特性。建立该疾病的进展模型,并评估研究设计和分析选项的相对优点。
来自四项干预试验和两个登记处的979名患者的数据可供分析。我们的研究分为:(1)估计28个条目的信息量;(2)估计疾病进展;(3)比较量表、试验设计和分析选项在检测临床相关治疗效果方面的效能。
PSPRS条目得分的两两相关性较低(r = 0.17 ± 0.14),易怒、睡眠困难和姿势性震颤条目与其他条目不相关。FDA选择的条目显示出更高的相关性(r = 0.35 ± 0.14),并且是包括疾病进展的纵向项目反应模型的基础。试验模拟表明,如果分析基于纵向项目信息,与治疗结束时的总分相比,识别疾病修饰治疗效果所需的研究规模不到一半。
基于FDA选择的PSPRS条目的纵向项目反应模型是评估PSP治疗方法的一个有前景的工具。© 2024作者。《运动障碍》由Wiley Periodicals LLC代表国际帕金森和运动障碍协会出版。