由单一炎性脱髓鞘病变引起的对左旋多巴有反应的霍姆斯震颤。
Levodopa-responsive Holmes' Tremor Caused by a Single Inflammatory Demyelinating Lesion.
作者信息
Katschnig-Winter Petra, Koegl-Wallner Mariella, Pendl Tamara, Fazekas Franz, Schwingenschuh Petra
机构信息
Department of Neurology, Medical University of Graz, Graz, Austria.
Department of Neurology, Medical University of Graz, Graz, Austria ; Division of Neuroradiology, Department of Radiology, Medical University of Graz, Graz, Austria.
出版信息
Tremor Other Hyperkinet Mov (N Y). 2015 Sep 25;5:339. doi: 10.7916/D8WQ033X. eCollection 2015.
BACKGROUND
Holmes' tremor is characterized by a combination of rest, postural, and kinetic tremor that is presumably caused by interruption of cerebello-thalamo-cortical and nigrostriatal pathways. Medical treatment remains unsatisfactory.
CASE REPORT
A 16-year-old girl presented with Holmes' tremor caused by a transient midbrain abnormality on magnetic resonance imaging (MRI). To explore the discrepancy between persistent tremor and resolved MRI changes, we performed dopamine transporter single-photon emission computed tomography (DaT-SPECT) with a 123I-ioflupane that revealed nearly absent DaT binding in the right striatum. Levodopa dramatically improved the tremor.
DISCUSSION
This is only the second report of a transient midbrain MRI abnormality disrupting nigrostriatal pathways. The case highlights the sometimes limited sensitivity of morphologic imaging for identifying the functional consequences of tissue damage and confirms that DaT imaging may serve as a predictor for levodopa responsiveness in Holmes' tremor.
背景
霍姆斯震颤的特征是静止性、姿势性和运动性震颤并存，推测是由小脑 - 丘脑 - 皮质和黑质纹状体通路中断所致。药物治疗效果仍不尽人意。
病例报告
一名16岁女孩因磁共振成像（MRI）显示中脑短暂异常而出现霍姆斯震颤。为探究持续性震颤与MRI改变消退之间的差异，我们用123I - 碘氟潘进行了多巴胺转运体单光子发射计算机断层扫描（DaT - SPECT），结果显示右侧纹状体几乎没有DaT结合。左旋多巴显著改善了震颤。
讨论
这是第二例关于中脑MRI短暂异常破坏黑质纹状体通路的报告。该病例凸显了形态学成像在识别组织损伤功能后果方面有时存在的有限敏感性，并证实DaT成像可作为霍姆斯震颤中左旋多巴反应性的预测指标。
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