Reliability of the CMT neuropathy score (second version) in Charcot-Marie-Tooth disease.
Reliability of the CMT neuropathy score (second version) in Charcot-Marie-Tooth disease.
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DOI：
10.1111/j.1529-8027.2011.00350.x
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发表时间：
2011-09
期刊：
影响因子：
--
通讯作者：
Pareyson D
中科院分区：
文献类型：
--
作者：
Murphy SM;Herrmann DN;McDermott MP;Scherer SS;Shy ME;Reilly MM;Pareyson D
Charcot-Marie-Tooth神经病变评分(CMTNS)是一种可靠有效的综合评分，包括症状、体征和神经生理学测试，已被用于CMT1A和CMT1X的自然历史研究，并作为CMT1A治疗试验的结果衡量标准。在一次关于夏科-玛丽-托斯病(CMT)预后衡量的国际研讨会之后，对CMTNS进行了修改，试图减少地板和天花板的影响，并使患者评估标准化，旨在提高其检测随时间变化和干预效果的敏感性。在对CMTNS(CMTNS2)的修改达成一致后，三名审查员评估了16名患者，以确定评价员间的可靠性；一名审查员在8周内对18名患者进行了两次评估，以确定评价员内的可靠性。三名检查员使用CMTNS和CMTNS2评估了63名患者，以确定修改如何改变评分。对于评分者间和评分者内的可靠性，CMT症状评分和CMT检查分数的组内相关系数(ICCs)为≥0.96。与CMTNS2相比，CMTNS的一些单独组成部分存在微小但显著的差异，主要是修改最多的组成部分。一项纵向研究正在进行中，以确定在检测随时间变化方面，CMTNS2是否比CMTNS更敏感。
The Charcot-Marie-Tooth neuropathy score (CMTNS) is a reliable and valid composite score comprising symptoms, signs, and neurophysiological tests, which has been used in natural history studies of CMT1A and CMT1X and as an outcome measure in treatment trials of CMT1A. Following an international workshop on outcome measures in Charcot-Marie-Tooth disease (CMT), the CMTNS was modified to attempt to reduce floor and ceiling effects and to standardize patient assessment, aiming to improve its sensitivity for detecting change over time and the effect of an intervention. After agreeing on the modifications made to the CMTNS (CMTNS2), three examiners evaluated 16 patients to determine inter-rater reliability; one examiner evaluated 18 patients twice within 8 weeks to determine intra-rater reliability. Three examiners evaluated 63 patients using the CMTNS and the CMTNS2 to determine how the modifications altered scoring. For inter- and intra-rater reliability, intra-class correlation coefficients (ICCs) were ≥0.96 for the CMT symptom score and the CMT examination score. There were small but significant differences in some of the individual components of the CMTNS compared with the CMTNS2, mainly in the components that had been modified the most. A longitudinal study is in progress to determine whether the CMTNS2 is more sensitive than the CMTNS for detecting change over time.
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