Objective: To report a unique case of a patient with acquired Holmes tremor (HT) secondary to Human Immunodeficiency Virus (HIV) and progressive multifocal leukoencephalopathy (PML) effectively treated with bilateral deep brain stimulation (DBS).

Background: The treatment of HT, characterized by lower frequency and large amplitude proximal tremor, can be challenging, and there is currently no consensus for its management (1). Although DBS can be a partially effective treatment for HT, only a few reports and case series are available in the literature (1–4). We report a case in which contralateral DBS implantation resulted in significant improvement of symptoms in a patient with acquired HT.

Method: Case report

Results: A 58-year-old right-handed man presented with a 20-year history of bilateral arm tremor. Prior to onset, he had been diagnosed with HIV/AIDS and PML. Propranolol, topiramate, and carbidopa-levodopa were tried without success. Physical exam revealed a 4-5 Hz moderate amplitude resting tremor of both arms that increased with posture and intention. Brain MRI showed patchy areas of encephalomalacia throughout the brain in a watershed distribution. Due to the medically refractory HT resulting in significant functional impairment, he underwent DBS implantation of the left ventral intermediate nucleus and posterior subthalamic area (VIM/PSA) . Although his right arm tremor was markedly improved, his contralateral tremor was still resulting in significant disability. One year after, he underwent DBS implantation of his right VIM/PSA. At six months follow up, there were no significant side-effects. With his DBS stimulation turned off and on, respectively, he scored 82 and 58 on the Fahn-Tolosa-Marin rating scale of tremor. While the stimulation was off, he was unable to hold the pen to perform any of its drawings.

Conclusion: Medical therapy for patients with HT can be challenging and is seldom fully effective (1,2). Limited avaiable data suggests that unilateral DBS treatment of HT can result in significant clinical improvement in tremors refractory to medical therapy (1,3). To our knowledge, this is the first report of bilateral DBS VIM/PSA implantation successfully treating HT with no significant side effects.

References: 1. Wang KL, Wong JK, Eisinger RS, Carbunaru S, Smith C, Hu W, et al. Therapeutic Advances in the Treatment of Holmes Tremor: Systematic Review. Neuromodulation. Blackwell Publishing Inc.; 2020.
2. Ramirez-Zamora A, Okun MS. Deep brain stimulation for the treatment of uncommon tremor syndromes. Vol. 16, Expert Review of Neurotherapeutics. Taylor and Francis Ltd; 2016. p. 983–97.
3. Espinoza Martinez JA, Arango GJ, Fonoff ET, Reithmeier T, Escobar OA, Furlanetti L, et al. Deep brain stimulation of the globus pallidus internus or ventralis intermedius nucleus of thalamus for Holmes tremor. Neurosurgical Review. 2015 Oct 13;38(4):753–63.
4. Ghanchi H, Siddiqi I, Patchana T, Ananda A. Acquired Holmes Tremor in a Human Immunodeficiency Virus Immune Reconstitution Inflammatory Syndrome Patient Treated with Deep Brain Stimulation. World Neurosurgery. 2020 Sep 1;141:253–9.

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