open access

Vol 50, No 5 (2016)
Case reports
Submitted: 2016-01-21
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Deep brain stimulation for levodopa-refractory benign tremulous parkinsonism

Takuya Konno, Owen A. Ross, Robert E. Wharen, Ryan J. Uitti, Zbigniew K. Wszolek
DOI: 10.1016/j.pjnns.2016.05.008
·
Neurol Neurochir Pol 2016;50(5):383-386.

open access

Vol 50, No 5 (2016)
Case reports
Submitted: 2016-01-21

Abstract

Benign tremulous parkinsonism (BTP) is characterized by prominent resting tremor combined with action and postural components, and with only subtle rigidity and bradykinesia. This tremor is frequently disabling and poorly responsive to therapy with levodopa. Thus, BTP could be considered either as a distinct clinical disorder or a variant of PD. We present a case of a 57-year-old man who had a 3-year history of severe and functionally disabling resting tremor with action and postural features bilaterally but with left dominant hand predominance. There was only very mild rigidity and bradykinesia and no postural instability. His tremor was refractory to dopaminergic therapy, including carbidopa/levodopa. The dopamine transporter (DAT) imaging showed reduced tracer uptake in the putamen bilaterally, more so on the right side. He was treated with deep brain stimulation (DBS) targeting the right ventral intermediate nucleus of the thalamus. His tremor resolved immediately after procedure. The DAT imaging abnormalities indicate the presynaptic dopamine deficiency. In some autopsied BTP cases classic alpha-synuclein pathology of PD was observed. Thus, despite the lack of levodopa responsiveness BTP likely represents a variant of PD and not a distinct neurodegenerative disorder. DBS should be considered for patients with BTP PD variant despite their poor responsiveness to levodopa treatment.

Abstract

Benign tremulous parkinsonism (BTP) is characterized by prominent resting tremor combined with action and postural components, and with only subtle rigidity and bradykinesia. This tremor is frequently disabling and poorly responsive to therapy with levodopa. Thus, BTP could be considered either as a distinct clinical disorder or a variant of PD. We present a case of a 57-year-old man who had a 3-year history of severe and functionally disabling resting tremor with action and postural features bilaterally but with left dominant hand predominance. There was only very mild rigidity and bradykinesia and no postural instability. His tremor was refractory to dopaminergic therapy, including carbidopa/levodopa. The dopamine transporter (DAT) imaging showed reduced tracer uptake in the putamen bilaterally, more so on the right side. He was treated with deep brain stimulation (DBS) targeting the right ventral intermediate nucleus of the thalamus. His tremor resolved immediately after procedure. The DAT imaging abnormalities indicate the presynaptic dopamine deficiency. In some autopsied BTP cases classic alpha-synuclein pathology of PD was observed. Thus, despite the lack of levodopa responsiveness BTP likely represents a variant of PD and not a distinct neurodegenerative disorder. DBS should be considered for patients with BTP PD variant despite their poor responsiveness to levodopa treatment.

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Keywords

Benign tremulous parkinsonism, Parkinson' s disease, Deep brain stimulation, Dopamine transporter imaging, Tremor

About this article
Title

Deep brain stimulation for levodopa-refractory benign tremulous parkinsonism

Journal

Neurologia i Neurochirurgia Polska

Issue

Vol 50, No 5 (2016)

Pages

383-386

DOI

10.1016/j.pjnns.2016.05.008

Bibliographic record

Neurol Neurochir Pol 2016;50(5):383-386.

Keywords

Benign tremulous parkinsonism
Parkinson's disease
Deep brain stimulation
Dopamine transporter imaging
Tremor

Authors

Takuya Konno
Owen A. Ross
Robert E. Wharen
Ryan J. Uitti
Zbigniew K. Wszolek

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