Objective: To report the long-term outcome of a case of rubral tremor responding well to levodopa, however complicated by fluctuations and dyskinesia.

Background: Rubral tremor (RT) presents with action, posture, and rest tremor. Typically lesions to the brainstem, thalamus, or cerebellum are implicated. Pharmacologic treatments for RT include benzodiazepines, anticholinergics, and levodopa, with variable responses. Treatment with levodopa has been reported to be effective in ~54% of patients, suggesting a role of dopaminergic denervation in disease pathophysiology.

Method: Patient was seen at UCSF Movement Disorders clinic over 20 years. Medical records were reviewed, and patient was interviewed and consented.

Results: A 20 year old right handed woman developed visual changes, left leg weakness, and left arm tremor following right midbrain cavernoma hemorrhage. These symptoms resolved after a few months. However, she had re-hemorrhage four years later, causing visual changes, facial droop, and left arm tremor. Despite resolution of her other symptoms, tremor persisted, and trials of benztropine, gabapentin, clonazepam, and topiramate were ineffective. At 31 years old, she was referred to our clinic for refractory tremor. On our evaluation, tremor was present at rest, posture, and action, consistent with rubral tremor. She also had left sided ataxia, however no parkinsonism.  She was started on carbidopa levodopa IR 25/100 mg, escalated to two tablets three times a day, which provided 50-75% improvement in tremor. Four years later, she developed wearing off 3-4 hours after each dose. Entacapone provided some improvement in fluctuations.  Later she was switched to carbidopa levodopa ER. She subsequently developed OFF dystonia and biphasic dyskinesia. Addition of opicapone reduced wearing off, and dyskinesia improved with reduction in carbidopa levodopa ER. She continues to require careful titration and adjustments to medications.

Conclusion: In this patient with RT, levodopa was effective in reducing tremor. Interestingly, despite a presumably static injury, our patient developed wearing off and biphasic dyskinesia on prolonged levodopa therapy. The latter has improved somewhat with typical PD treatment approaches to motor complications.

References: Raina GB, Cersosimo MG, Folgar SS, Giugni JC, Calandra C, Paviolo JP, et al. Holmes tremor: clinical description, lesion localization, and treatment in a series of 29 cases. Neurology. (2016) 86:931–8. 10.1212/WNL.0000000000002440

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MDS Abstracts - https://www.mdsabstracts.org/abstract/development-of-motor-complications-in-rubral-tremor-treated-with-levodopa/