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Published on: October 14, 2021
Benign tremulous parkinsonism
Keith A Josephs1, Joseph Y Matsumoto, J Eric Ahlskog
1Division of Movement Disorders, Department of Neurology, Mayo Clinic, Rochester, Minn, USA. josephs.keith@mayo.edu
Background:
Benign tremulous parkinsonism has never been precisely defined nor has the long-term course been studied.
Objective:
To report the clinical features and longitudinal course of patients with benign tremulous parkinsonism encountered in our movement disorders practice.
Design:
Computer search of medical records database.
Setting:
Mayo Clinic, Rochester, Minn.
Patients:
Of 116 patients identified, 16 (10 male and 6 female) had at least an 8-year history of this disease, had been examined by a senior movement disorders specialist, and had ultimately been diagnosed as having benign tremulous parkinsonism after an initial diagnosis of Parkinson disease (PD).
Interventions:
None.
Main Outcome Measures:
Age at onset of disease, response to levodopa therapy, tremor characteristics, and family history.
Results:
Mean disease duration was 11 years (range, 8-25 years) at last follow-up. Mean age at onset, 58.5 years, was younger than in most PD series, and most patients had a poor levodopa response (although levodopa trials were inadequate in some). A moderate to marked postural tremor was noted in 13 of the 16 patients, including 6 with a kinetic tremor. A family history of PD and/or tremor was reported in 10 (63%) of our patients. Three patients required thalamic deep brain surgery to treat their tremor.
Conclusions:
Benign tremulous parkinsonism may be a distinct clinical entity characterized by tremor predominance plus minimal progression of other aspects of parkinsonism. The tremor is often not very responsive to levodopa therapy. In this series, most patients had immediate family members with a diagnosis of tremor or PD.
Insights
Benign tremulous parkinsonism is a distinct neurological disorder characterized by tremor. This condition shows minimal progression in other parkinsonian symptoms and often has a poor response to levodopa therapy.
Area of Science:
- Neurology
- Movement Disorders
- Neurodegenerative Diseases
Background:
- Benign tremulous parkinsonism lacks a precise definition and long-term studies.
- Characterized by tremor, its clinical features and progression remain under-investigated.
Purpose of the Study:
- To define the clinical features and longitudinal course of benign tremulous parkinsonism.
- To differentiate this condition from Parkinson disease (PD).
Main Methods:
- Retrospective analysis of 116 patients with initial PD diagnosis.
- 16 patients with benign tremulous parkinsonism and at least an 8-year history were selected.
- Key data included age at onset, levodopa response, tremor type, and family history.
Main Results:
- Mean disease duration was 11 years; mean age at onset was 58.5 years.
- Most patients exhibited poor levodopa response and moderate to marked postural/kinetic tremor.
- A family history of tremor or PD was reported in 63% of patients; 3 required deep brain stimulation.
Conclusions:
- Benign tremulous parkinsonism may be a distinct entity with tremor predominance and slow progression.
- Tremor in this condition is often unresponsive to levodopa.
- A significant family history of tremor or PD is common in affected individuals.
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