Dopa‐responsive childhood dystonia: a forme fruste with writer's cramp; triggered by exercise

Dopa‐responsive childhood dystonia: a forme fruste with writer's cramp; triggered by exercise
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多巴反应性儿童肌张力障碍:运动引发的写作痉挛的形式障碍;

DOI:
10.1111/j.1469-8749.1997.tb08204.x
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发表时间:
1997
影响因子:
3.8
通讯作者:
P. Zesiger
P. Zesiger
中科院分区:
医学2区
文献类型:
--
作者:
T. Deonna;E. Roulet;J. Ghika;P. Zesiger

文献摘要

被引文献

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在过去2年中,对一名11岁女孩进行了行走困难和疲劳评估。书写也很困难,在短时间的书写后会出现“抽筋”。神经系统检查大部分时间正常,但在晚上和运动后,有时可以看到异常的步行姿势和罕见的足部张力障碍运动。母亲被发现患有轻度帕金森症,在左旋多巴治疗时无症状。在女儿身上,所有症状和体征在左旋多巴上消失,但在停药时又恢复了。通过录像和书写样本的计算机分析记录治疗期间和治疗结束时的变化。5年随访期间情况稳定。我们提请注意多巴敏感性儿童肌张力障碍的这种“形式障碍”,它随着运动而变得明显,并且很容易被识别。我们还讨论并举例说明了用于分析写作的方法。
An 11‐year‐old girl was evaluated for walking difficulties and fatigue at the end of the day in the last 2 years. Handwriting was also difficult with ‘cramps’ after a short time of writing. Neurological examination was normal most of the time but in the evening and after exercise, an abnormal walking posture and rare dystonic movements of the foot could sometimes be seen. The mother was found to have mild parkinsonism and is asymptomatic on L‐dopa. In the daughter, all symptoms and signs disappeared on L‐dopa, but returned when the drug was withdrawn. The changes on‐ and off‐treatment were documented with videofilms and computerized analysis of writing samples. The situation has been stable during a 5‐year follow‐up. We draw attention to this ‘forme fruste’ of dopa‐sensitive childhood dystonia which becomes manifest with exercise and which can easily go unrecognized. We also discuss and illustrate the methods used for the analysis of writing.