Objective: OBJECTIVE: To describe a novel response of Atypical Holmes tremor to intrathecal baclofen (ITB).

Background: Holmes tremor is a rare low-frequency high-amplitude tremor predominantly affecting the proximal upper extremity. It commonly results from vascular or inflammatory lesions in the contralateral thalamus, cerebellum, or brainstem structures. [1]. Medical treatment and Deep brain stimulation (DBS) have shown mixed results [2-5].

Method: Case Report

Results: A 52-year-old Right-handed female with history of cryptogenic stroke presented to the movement disorder clinic for advanced management of post-stroke lower extremity tremor. The tremor developed in her right leg and to a lesser extent in the right arm a few weeks following the stroke. Exam showed a high-frequency constant tremor at rest, posture and with action predominantly involving the proximal right lower extremity with mild ipsilateral spasticity and generalized hyperreflexia. The tremor did not respond to sequential trials of pramipexole, levodopa, propranolol, topiramate, risperidone, and clonazepam. Brain MRI showed infarct in the left thalamus, upper midbrain, and cerebellum [fig1A,B,C]. Given her predominant lower extremity tremor with coexisting spasticity, ITB was favored over Deep brain stimulation (DBS) especially since DBS targets in the brain had suffered considerable tissue damage from the stroke. Patient underwent ITB testing with a 50-microgram-bolus which resulted in near resolution of spasticity and marked objective improvement in tremor and ambulation.Subjectively, the patient reported 80% tremor reduction after the test.

Conclusion: Through its action on spinal GABA-B receptors, ITB may suppress lower extremity cerebellar tremor in a manner similar to its effect on post-stroke spasticity. Weiss N et al described incidental improvement of upper extremity cerebellar intentional tremor after baclofen pump was implanted for spasticity [6].Our case is the first to demonstrate responsiveness of atypical Holmes tremor to ITB. ITB pump may be a promising neuromodulation option for medically-refractory atypical Holmes tremor of the lower extremity.

References: 1. Wei TS1, Hsu CS, Lee YC, Chang S Degeneration of paramedian nuclei in the thalamus induces Holmes tremor in a case of artery of Perc heron infarction. Medicine (Baltimore). 2017 Nov;96(46): e8633. doi: 10.1097/MD.0000000000008633. 2. Ramirez-Zamora A1, Okun MS2. Deep brain stimulation for the treatment of uncommon tremor syndromes Expert Rev Neurother. 2016 Aug;16(8):983-97. doi: 0.1080/14737175.2016.1194756. Epub 2016 Jun 9. 3. Kobayashi K, Katayama Y, Oshima H. Multitarget, dual-electrode deep brain stimulation of the thalamus and subthalamic area for treatment of Holmes’ tremor. J Neurosurg. 2014;120(5):1025–1032. 4. Romanelli P, Brontë-Stewart H, Courtney T. Possible necessity for deep brain stimulation of both the ventralis intermedius and subthalamic nuclei to resolve Holmes tremor. Case report. J Neurosurg. 2003;99(3):566–571. 5. Kilbane C, Ramirez-Zamora A, Ryapolova-Webb E. Pallidal stimulation for Holmes tremor: clinical outcomes and single-unit recordings in 4 cases. J Neurosurg. 2015;122(6):1306–1314. 6. Weiss N1, North RB, Ohara S, Lenz FA. Attenuation of cerebellar tremor with implantation of an intrathecal baclofen pump: the role of gamma-aminobutyric acidergic pathways. Case report J Neurosurg. 2003 Oct;99(4):768-71

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MDS Abstracts - https://www.mdsabstracts.org/abstract/improvement-of-lower-extremity-holmes-tremor-with-intrathecal-baclofen-first-report/