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通过分析日常生活中使用计算机时的键盘打字情况来监测亨廷顿病的运动表型。

Monitoring the Motor Phenotype in Huntington's Disease by Analysis of Keyboard Typing During Real Life Computer Use.

机构信息

Department of Neurology, University Hospital Ulm, Ulm, Germany.

NeuraMetrix, Inc., San Francisco, CA, USA.

出版信息

J Huntingtons Dis. 2021;10(2):259-268. doi: 10.3233/JHD-200451.

Abstract

BACKGROUND

Besides cognitive and psychiatric abnormalities, motor symptoms are the most prominent in Huntington's disease. The manifest disease is preceded by a prodromal phase with subtle changes such as fine motor disturbances or concentration problems.

OBJECTIVE

Movement disorders show a high variation in their clinical manifestation depending on condition and external influences. Therefore, devices for continuous measurements, which patients use in their daily life and which can monitor motor abnormalities, in addition to the medical examination, might be useful. The aim of current scientific efforts is to find markers that reflect the prodromal phase in gene carriers. This is important for future interventional studies, as future therapies should be applied at the stage of neuronal dysfunction, i.e., before the clinical manifestation.

METHODS

We performed a software-supported, continuous monitoring of keyboard typing on the participants' own computer to evaluate this method as a tool to assess the motor phenotype in HD. We included 40 participants and obtained sufficient data from 25 participants, 12 of whom were manifest HD patients, 7 HD gene expansion carriers (HDGEC) and 6 healthy controls.

RESULTS

In a cross-sectional analysis we found statistically significant higher typing inconsistency in HD patients compared to controls. Typing inconsistency compared between HDGEC and healthy controls showed a trend to higher inconsistency levels in HDGEC. We found correlations between typing cadence and clinical scores: the UHDRS finger tapping item, the composite UHDRS and the CAP score.

CONCLUSION

The typing cadence inconsistency is an appropriate marker to evaluate fine motor skills of HD patients and HDGEC and is correlated to established clinical measurements.

摘要

背景

除了认知和精神异常,运动症状是亨廷顿病中最突出的。在明显的疾病之前,有一个前驱期,有细微的变化,如精细运动障碍或注意力问题。

目的

运动障碍的临床表现有很大的差异,取决于病情和外部影响。因此,除了医学检查之外,使用患者在日常生活中使用的连续测量设备来监测运动异常可能是有用的。目前科学努力的目标是找到反映基因携带者前驱期的标志物。这对于未来的干预性研究很重要,因为未来的治疗应该在神经元功能障碍阶段应用,即在临床表现之前。

方法

我们使用软件支持的方法对参与者自己的计算机进行连续的键盘打字监测,以评估这种方法作为评估 HD 运动表型的工具。我们纳入了 40 名参与者,其中 25 名参与者获得了足够的数据,其中 12 名是明显的 HD 患者,7 名是 HD 基因扩展携带者(HDGEC),6 名是健康对照组。

结果

在横断面分析中,我们发现 HD 患者的打字不一致性明显高于对照组。与健康对照组相比,HDGEC 之间的打字不一致性显示出 HDGEC 中不一致性水平较高的趋势。我们发现打字节奏与临床评分之间存在相关性:UHDRS 手指敲击项目、UHDRS 综合评分和 CAP 评分。

结论

打字节奏的不一致性是评估 HD 患者和 HDGEC 精细运动技能的合适标志物,与已建立的临床测量相关。

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