Disruption of nigrostriatal and cerebellothalamic pathways in dopamine responsive Holmes' tremor. Issue 8 (1st May 2008)

• Record Type: Journal Article
• Authors: Seidel, S; Kasprian, G; Leutmezer, F; Prayer, D; Auff, E
• Is Part Of: Journal of neurology, neurosurgery and psychiatry. Volume 80:Issue 8(2009)
• Page Start: 921
• Page End: 923
• Publication Date: 2008-05-01
• Subjects: Neurology -- Periodicals; Nervous system -- Surgery -- Periodicals; Psychiatry -- Periodicals; 616.8
• DOI: 10.1136/jnnp.2008.146324
• Languages: English
• ISSNs: 0022-3050

Abstract

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.