Disruption of nigrostriatal and cerebellothalamic pathways in dopamine responsive Holmes’ tremor
- 1Department of Neurology, Medical University Vienna, Vienna, Austria
- 2Department of Radiology, Medical University Vienna, Vienna, Austria
- Dr S Seidel, Department of Neurology, Medical University Vienna, Währinger Gürtel 18-20, 1090 Vienna, Austria; stefan.seidel{at}meduniwien.ac.at
- Received 4 February 2008
- Revised 9 April 2008
- Accepted 16 April 2008
- Published Online First 1 May 2008
Abstract
Holmes’ tremor is an unusual combination of rest, postural and kinetic tremor of the extremities. Medical treatment of this condition still remains unsatisfactory. The case of a 20-year-old female patient is reported who developed right-sided Holmes’ tremor 9 months after a left-sided, cavernoma induced midbrain/pontine haemorrhage at the age of 16 years. Beta-CIT single photon emission computed tomography revealed abolished dopamine transporter activity in the left basal ganglia and striatum, in accordance with missing ipsilateral tegmento-frontal connectivity (medial forebrain bundle), demonstrated by diffusion tensor MRI. Tractography showed reduced fibre connectivity of the superior and middle cerebellar peduncles on the lesioned side. Administration of pramipexole and L-DOPA led to a clinically significant reduction in tremor severity. In conclusion, our results support the notion that Holmes’ tremor was a result of diminished striatal dopaminergic input in our patient.
Footnotes
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Competing interests: None.
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Patient consent: Obtained.
