RT Journal Article
SR Electronic
T1 Disruption of nigrostriatal and cerebellothalamic pathways in dopamine responsive Holmes’ tremor
JF Journal of Neurology, Neurosurgery & Psychiatry
JO J Neurol Neurosurg Psychiatry
FD BMJ Publishing Group Ltd
SP 921
OP 923
DO 10.1136/jnnp.2008.146324
VO 80
IS 8
A1 Seidel, S
A1 Kasprian, G
A1 Leutmezer, F
A1 Prayer, D
A1 Auff, E
YR 2009
UL http://jnnp.bmj.com/content/80/8/921.abstract
AB 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.