Levodopa-responsive Holmes’ Tremor Caused by a Single Inflammatory Demyelinating Lesion

Background: Holmes’ tremor is characterized by a combination of rest, postural, and kinetic tremor that is presumably caused by interruption of cerebellothalamo-cortical and nigrostriatal pathways. Medical treatment remains unsatisfactory.Case Report: A 16-year-old girl presented with Holmes’ tremor...

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Bibliographic Details
Published in:Tremor and other hyperkinetic movements (New York, N.Y.) N.Y.), 2015-09, Vol.5, p.339
Main Authors: Katschnig-Winter, Petra, Koegl-Wallner, Mariella, Pendl, Tamara, Fazekas, Franz, Schwingenschuh, Petra
Format: Article
Language:English
Subjects:
Case reports
Conflicts of interest
Dopamine
Funding
Hemorrhage
Magnetic resonance imaging
Medical imaging
Multiple sclerosis
Neuroimaging
Patients
Phenomenology
Remission (Medicine)
Stroke
Tremor (Muscular contraction)
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Summary:Background: Holmes’ tremor is characterized by a combination of rest, postural, and kinetic tremor that is presumably caused by interruption of cerebellothalamo-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 (DaTSPECT) 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.
ISSN:2160-8288
2160-8288
DOI:10.5334/tohm.257