Deep brain stimulation of the posterior subthalamic area as an alternative strategy for management of Holmes tremor: A case report and review of the literature
- Research Center for Neuromodulation and Pain, Shiraz University of Medical Sciences, Shiraz, Iran,
- Division of Neurosurgery, City of Hope Beckman Research Institute and Medical Center, Duarte, California, United States.
Ali Razmkon, Research Center for Neuromodulation and Pain, Shiraz University of Medical Sciences, Shiraz, Iran.
DOI:10.25259/SNI_435_2022Copyright: © 2022 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Omid Yousefi1, Mojtaba Dayyani2, Razieh Rezaei1, Hooman Kamran1, Ali Razmkon1. Deep brain stimulation of the posterior subthalamic area as an alternative strategy for management of Holmes tremor: A case report and review of the literature. 28-Oct-2022;13:489
How to cite this URL: Omid Yousefi1, Mojtaba Dayyani2, Razieh Rezaei1, Hooman Kamran1, Ali Razmkon1. Deep brain stimulation of the posterior subthalamic area as an alternative strategy for management of Holmes tremor: A case report and review of the literature. 28-Oct-2022;13:489. Available from: https://surgicalneurologyint.com/surgicalint-articles/deep-brain-stimulation-of-the-posterior-subthalamic-area-as-an-alternative-strategy-for-management-of-holmes-tremor-a-case-report-and-review-of-the-literature/
Background: Holmes tremor is often refractory to medical treatment and deep brain stimulation of the ventralis intermedius nucleus of the thalamus (VIM-DBS) is the intervention of choice in controlling the tremor. Herein, we present a beneficial alternative strategy for the management of such situations, considering the posterior subthalamic area (PSA) as the target of stimulation.
Case Description: We report a 57-year-old male with the right-sided tremor following a traumatic brain injury 20 years ago. He had been diagnosed with Holmes tremor that was not responsive to nonsurgical therapeutic options. When refractoriness confirmed, he became a candidate for VIM-DBS. During the operation, by performing macrostimulation with a maximum of 2 mA of amplitude, the tremor had no response to the stimulation of different tracts, and severe right hemi-body paresthesia occurred; therefore, we modified our approach and targeted the PSA, which resulted in satisfactory control of the tremor. The permanent lead was implanted into the left side PSA. At 1-year follow-up, the right side tremor was under complete control.
Conclusion: Our case and other similar pieces of evidence are consistently indicating the potential regulatory effects of PSA-DBS in controlling the Holmes tremor as a feasible alternative strategy when VIM-DBS does not provide a satisfactory response. However, further studies with larger sample size are required to evaluate the long-term response and its possible long-term stimulation-related effects.
Keywords: Deep brain stimulation, Holmes tremor, Nucleus ventralis intermedius, Posterior subthalamic area, Thalamus, Tremor
High-frequency stimulation and lesioning of the nucleus ventralis intermedius (VIM) are the conventional surgical procedures used for alleviating tremors of different pathologies. VIM-deep brain stimulation (DBS) was first introduced as the surgical treatment for Parkinson’s disease, and its safety and efficacy for the treatment of tremor dominant movement disorders such as essential tremor (ET) and Holmes tremor (HT) have been reported in several studies.[
HT, caused by a lesion in the basal ganglia and specific tracts, is believed to have a low response to medical treatments.[
The posterior subthalamic area (PSA) is among the targets and efficient in controlling the tremor by receiving high-frequency stimulation.[
We present a 57-year-old male with HT who had no response to medical treatment for 20 years and eventually became a candidate for VIM-DBS. Due to the weak response to VIM-DBS during surgery, the PSA-DBS was chosen as the alternative strategy. Herein, we discuss the rationale, technical challenges, and outcome of the PSA-DBS strategy, along with a comprehensive review of the pertinent literature.
A 57-year-old otherwise healthy male had fallen off a horse 20 years ago. He had had a traumatic brain injury and had been in coma for a month. Immediately after gaining consciousness, he developed dysarthria, and 6 months after the accident, he gradually developed right-sided hemiparesis and hemi-body tremor. Resting, action, and positional tremor have been significantly debilitating the patient and hindering him to perform his daily activities. He had also been unable to walk even with assistance and had been wheelchair bounded for the past 20 years.
The patient had received various medical treatments over the years, and none of them was effective in the control of the tremor. Eventually, he was referred to our movement disorder clinic in February 2020. As the possible therapeutic surgical intervention to tackle his tremor, unilateral VIMDBS was suggested to him and his family.
Nonstereotactic magnetic resonance imaging (MRI) (1.5 T Integra, Philips, Netherlands) was obtained a few days before surgery under light sedation (to prevent motion artifacts secondary to tremor). On the day of surgery, after installing the Cosman–Roberts–Wells (CRW) stereotactic frame (Integra Life, USA), a stereotactic computed tomography (CT) scan was done.
The planning process was carried out using StealthStation S8 (Medtronic, Minneapolis, USA). Initially, gadolinium-enhanced T1-weighted MRI was selected as a reference, and other MRI sequences and CT scan were superimposed on it and checked if the different sets of landmarks were matched accurately. After defining the anterior and posterior commissures on the axial T1 sequences, indirect targeting of VIM was accomplished through axial and coronal T2-weighted sequences. The stereotactic coordinates of the VIM target were X: −14.6, Y: −5, and Z: −2.
Following the localization of the entrance point and the trajectory, microelectrode recording (MER) was performed using the Leadpoint system (Medtronic, Skovlundae, Denmark, and Shoreview, Minnesota, USA). The action potentials were recorded in the medial, central, and posterior tracts. The recording suggestive of the thalamic tremor was captured in all tracts from 4 mm above to 2 mm below the level of the target. Based on these findings, we decided to perform macrostimulation along the central tract. The high-frequency constant current stimulation was initiated from 4 mm above the target, under the supervision of a movement disorder neurologist. While stimulation was started from 0 mA and was increased stepwise by 1 mA at each level, the patients’ tremor was getting assessed continuously along with the ascending stimulation. The gradual increment of stimulation was cautiously continued till any stimulation-related adverse event was observed. Unfortunately, the stimulation was not effective in the central, posterior, medial, and anterior tracts. Resting, action, and intentional tremors did not respond to the stimulation with different amplitudes, and when the amplitude reached 2 mA, severe acute right hemi-body paresthesia was evident as an adverse event of stimulation. Using intraoperative lateral fluoroscopy and trunion reticles, we confirmed that the stimulation electrode was precisely placed in the correct location.
Nonetheless, due to the insufficient response to VIM stimulation, we decided to perform macrostimulation of PSA as a possible alternative strategy [
Conventional VIM-DBS versus the alternative approach, PSA-DBS.
A: Deep brain stimulation of VIM as the conventional intervention of choice was not helpful in reducing tremor. B: Deep brain stimulation of PSA as the alternative strategy resulted in satisfactory control of the tremor. #Trajectories of the leads demonstrated in this figure are putative and do not represent the actual tracts in the surgery. (The figure is created with BioRender.com).
Interestingly, PSA stimulation (2 mA) resulted in a considerable alleviation of all types of tremors. The patient and his family were reinformed about the favorable response of PSA-DBS, and after obtaining the consent, the permanent lead (DB-2201, Boston Scientific, USA) was implanted in the left PSA in a way that contact 1 was placed 2 mm below the defined target.
An immediate postoperative CT scan was done and the images were superimposed on the preoperative imaging to check the final location of the lead and any possible surgical inaccuracy or complications. The CT scan showed no misplacement or deviation of the lead from the defined target. As the last step, the neurostimulator (Vercise PC, Boston Scientific, USA) was implanted in the right upper part of the chest under general anesthesia. We avoided the left side insertion because implanted pulse generator (IPG) would interfere with the function of implanted cardiac devices (ICD) if the patient ever needed an ICD. After recovery, the patient was transferred to the intensive care unit and then transferred to the ward after 1 day.
The initial programming was performed 2 weeks after the surgery. Contact No. 2 of the implanted lead was selected as the active contact, and the unipolar stimulation parameters were adjusted as follows: Amplitude: 1.5 mA, pulse width: 60 µs, and frequency: 130 Hz.
Afterward, the tremor was significantly controlled and the patient gradually gained the ability to have a walker-assisted gait within 10 months and could accomplish his daily activities. At the time of the 1-year follow-up, he had an infrequent mild tremor and needed modest assistance with gait. Furthermore, no stimulation-related adverse event was observed, and the patient and his family were completely satisfied with the outcome of the surgery. Due to the unprogressive and completely alleviated tremor at the 1-year follow-up, the stimulation parameters were not readjusted at that point.
Among the movement disorders, HT usually does not show a satisfactory response to medical treatment. There are pieces of evidence indicating the efficacy of the surgical intervention, including DBS or lesioning, for the management of this disorder.[
VIM has been the conventional target used for the surgical treatment of tremor; however, new targets have been applied for cases in whom stimulation of the VIM seemed to be ineffective.[
Before the routine application of DBS, lesioning of PSA was a common procedure for the surgical management of tremor and Parkinson’s disease.[
In another study by Blomstedt et al.,[
There are a few reports in the literature representing the application of PSA-DBS on HT.
Furthermore, some studies suggest dual-target stimulation [
Some studies have done head to head comparison between the effectiveness of PSA-DBS and VIM-DBS. In a randomized and clinical trial by Barbe et al.,[
As the cerebello-thalamo-cortical circuit is related to ET, interfering with this circuit may alleviate the tremor.[
The exact underlying mechanism of tremor in HT is not known, which explains why the effectiveness of PSA-DBS has not been sharply demonstrated as well.[
Despite the advantages, PSA-DBS can be accompanied by stimulation-related adverse events. A study by Kim et al.[
Furthermore, a mini-review assessing the efficacy of PSADBS in patients with various types of tremors, including multiple sclerosis and posttraumatic-induced tremor, cerebellar tremor, HT, and spinocerebellar ataxia, reported that complications including dysphasia, dysarthria, or disequilibrium were mostly mild, transient, and not accompanied by tolerance, while the latter can be observed among the subjects who undergo VIM-DBS.[
In HT cases that are unresponsive to conventional VIMDBS, PSA-DBS can be a potential substitution approach to control the tremor effectively. However, almost all supporting evidence are limited to a few case reports. Therefore, further studies, especially the ones with larger sample size and possibly quasi-controlled designs, are needed to justify its potential efficacy along with the limitations and challenges compared to other targets, especially VIM.
The Ethics Committee of the Research Center for Neuromodulation and Pain (Shiraz, Iran) approved this study, and written informed consent from the presented case was obtained.
All relevant data have been reported in the manuscript.
The authors certify that they have obtained all appropriate patient consent.
Publication of this article was made possible by the James I. and Carolyn R. Ausman Educational Foundation.
There are no conflicts of interest.
The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.
We are grateful to our cooperative operating room staff and our patient and his family for trusting us to accomplish this uncommon surgical strategy.
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