The Canadian multicenter trial of pallidal deep brain stimulation for cervical dystonia: preliminary results in three patients

Object Deep brain stimulation (DBS) of the globus pallidus internus (GPi) is beneficial for generalized dystonia and has been proposed as a treatment for cervical dystonia. The Canadian Stereotactic/Functional and Movement Disorders Groups designed a pilot project to investigate the following hypothesis: that bilateral DBS of the GPi will reduce the severity of cervical dystonia at 1 year of follow up, as scored in a blinded fashion by two neurologists using the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS). Secondary outcome measures included pain and disability subscores of the TWSTRS, Short Form–36 quality of life index, and the Beck Depression Inventory. Methods Three patients have undergone surgery in Calgary with a followup duration of 7.4 ± 5.9 months (mean ± standard deviation). One patient underwent inadvertent ineffective stimulation for the first 3 months and did not experience a benefit until DBS programming was corrected. All three patients had rapid response to stimulation, with the muscles relaxing immediately and abnormal movements improving within days. Total TWSTRS scores improved by 79%, and severity subscores improved significantly, from 15.7 ± 2.1 to 7.7 ± 2.9 (paired ttest, p = 0.02). Pain and disability subscores improved from 25.5 ± 4.1 to 3.3 ± 3.1 (paired ttest, p = 0.002) and from 13.3 ± 4.9 to 3.3 ± 4.2 (paired ttest, p = 0.06), respectively. Conclusions Although it is too early to reach broad conclusions, this report of preliminary results confirms the efficacy of DBS of the GPi for cervical dystonia.

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Bilateral Deep Brain Stimulation for Cervical Dystonia: Long-term Outcome in a Series of 10 Patients

Neurosurgery ◽

10.1227/neu.0b013e3181ec49c7 ◽

2010 ◽

Vol 67 (4) ◽

pp. 957-963 ◽

Cited By ~ 39

Author(s):

Francesco Cacciola ◽

Jibril Osman Farah ◽

Paul R Eldridge ◽

Patricia Byrne ◽

Telekath K Varma

Keyword(s):

Deep Brain Stimulation ◽

Brain Stimulation ◽

Cervical Dystonia ◽

Rating Scale ◽

Spasmodic Torticollis ◽

Long Term Outcome ◽

Stimulation Parameters ◽

Deep Brain

Abstract BACKGROUND: Bilateral globus pallidus internus (GPi) deep brain stimulation (DBS) was shown to be effective in cervical dystonia refractory to medical treatment in several small short-term and 1 long-term follow-up series. Optimal stimulation parameters and their repercussions on the cost/benefit ratio still need to be established. OBJECTIVE: To report our long-term outcome with bilateral GPi deep brain stimulation in cervical dystonia. METHODS: The Toronto Western Spasmodic Torticollis Rating Scale was evaluated in 10 consecutive patients preoperatively and at last follow-up. The relationship of improvement in postural severity and pain was analyzed and stimulation parameters noted and compared with those in a similar series in the literature. RESULTS: The mean (standard deviation) follow-up was 37.6 (16.9) months. Improvement in the total Toronto Western Spasmodic Torticollis Rating Scale score as evaluated at latest follow-up was 68.1% (95% confidence interval: 51.5-84.6). In 4 patients, there was dissociation between posture severity and pain improvement. Prevalently bipolar stimulation settings and high pulse widths and amplitudes led to excellent results at the expense of battery life. CONCLUSION: Improvement in all 3 subscale scores of the Toronto Western Spasmodic Torticollis Rating Scale with bilateral GPi deep brain stimulation seems to be the rule. Refinement of stimulation parameters might have a significant impact on the cost/benefit ratio of the treatment. The dissociation of improvement in posture severity and pain provides tangible evidence of the complex nature of cervical dystonia and offers interesting insight into the complex functional organization of the GPi.

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Subthalamic nucleus deep brain stimulation for severe idiopathic dystonia: impact on severity, neuropsychological status, and quality of life

Journal of Neurosurgery ◽

10.3171/jns-07/07/0029 ◽

2007 ◽

Vol 107 (1) ◽

pp. 29-36 ◽

Cited By ~ 100

Author(s):

Galit Kleiner-Fisman ◽

Grace S. Lin Liang ◽

Paul J. Moberg ◽

Anthony C. Ruocco ◽

Howard I. Hurtig ◽

...

Keyword(s):

Quality Of Life ◽

Deep Brain Stimulation ◽

Subthalamic Nucleus ◽

Brain Stimulation ◽

Rating Scale ◽

Short Form ◽

Spasmodic Torticollis ◽

Sf 36 ◽

Deep Brain

Object Medically refractory dystonia has recently been treated using deep brain stimulation (DBS) targeting the globus pallidus internus (GPI). Outcomes have varied depending on the features of the dystonia. There has been limited literature regarding outcomes for refractory dystonia following DBS of the subthalamic nucleus (STN). Methods Four patients with medically refractory, predominantly cervical dystonia underwent STN DBS. Intraoperative assessments with the patients in a state of general anesthesia were performed to determine the extent of fixed deformities that might predict outcome. Patients were rated using the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) and the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) preoperatively and 3 and 12 months following surgery by a rater blinded to the study. Mean changes and standard errors of the mean in scores were calculated for each subscore of the two scales. Scores were also analyzed using analysis of variance and probability values were generated. Neuropsychological assessments and quality of life ratings using the 36-Item Short Form Health Survey (SF-36) were evaluated longitudinally. Results Significant improvements were seen in motor (p = 0.04), disability (p = 0.02), and total TWSTRS scores (p = 0.03). Better outcomes were seen in those patients who did not have fixed deformities. There was marked improvement in the mental component score of the SF-36. Neuropsychological function was not definitively impacted as a result of the surgery. Conclusions Deep brain stimulation of the STN is a novel target for dystonia and may be an alternative to GPI DBS. Further studies need to be performed to confirm these conclusions and to determine optimal candidates and stimulation parameters.

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Pallidal Deep Brain Stimulation in Cervical Dystonia: Clinical Outcome in Four Cases

Canadian Journal of Neurological Sciences / Journal Canadien des Sciences Neurologiques ◽

10.1017/s0317167100003401 ◽

2004 ◽

Vol 31 (3) ◽

pp. 328-332 ◽

Cited By ~ 57

Author(s):

H.A. Eltahawy ◽

J. Saint-Cyr ◽

Y.Y. Poon ◽

E. Moro ◽

A.E. Lang ◽

...

Keyword(s):

Deep Brain Stimulation ◽

Brain Stimulation ◽

Cervical Dystonia ◽

Rating Scale ◽

Clinical Results ◽

Spasmodic Torticollis ◽

Pallidal Stimulation ◽

The Mean ◽

Deep Brain

Objective:Report on the clinical results following bilateral globus pallidus interna deep brain stimulation in four patients (one female and three males) with severe cervical dystonia, mean age 48 years (range 37-67).Methods:All four patients had failed extensive medical and botulinum toxin treatment. The mean duration of the disease was nine years (range 4-15 years). Patients were assessed pre and postoperatively using the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS). Preoperatively, the mean TWSTRS total score was 43.2 (range 28-60.5). Posteroventral pallidal deep brain stimulators were inserted using MRI and microelectrode recording guidance. Last follow-up was 15 months for the four patients.Results:Mean reduction in the TWSTRS total scores at last follow- up was 73% (range 61- 85%). Improvement in pain occurred soon after deep brain stimulation surgery. Motor improvement was delayed and prolonged over several months. Frequent adjustment in the stimulation parameters was necessary in the first three months.Conclusion:Bilateral pallidal stimulation is effective in management of selected cases of intractable cervical dystonia.

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Pallidal deep brain stimulation in primary Meige syndrome: clinical outcomes and psychiatric features

Journal of Neurology Neurosurgery & Psychiatry ◽

10.1136/jnnp-2020-323701 ◽

2020 ◽

Vol 91 (12) ◽

pp. 1343-1348

Author(s):

Qingpei Hao ◽

Dongliang Wang ◽

Jia OuYang ◽

Hu Ding ◽

Gaungyong Wu ◽

...

Keyword(s):

Deep Brain Stimulation ◽

Sleep Quality ◽

Brain Stimulation ◽

Rating Scale ◽

Short Form ◽

Depression Scale ◽

Motor Symptoms ◽

Meige Syndrome ◽

Sf 36 ◽

Deep Brain

ObjectivesTo study the efficacy and safety of bilateral globus pallidus internus deep brain stimulation (GPi-DBS) in refractory Meige syndrome (MS) and evaluate the psychiatric disorders before and after surgery.MethodsTwenty-two patients with MS treated with bilateral GPi-DBS were retrospectively analysed before surgery and after continuous neurostimulation. Before surgery, patients were assessed by the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS), Self-Rating Depression Scale, Medical Outcomes Study 36-Item Short-Form General Health Survey (SF-36) and Pittsburgh Sleep Quality Index (PQSI), which corresponded to motor symptoms, depressive state, quality of life and sleep quality, respectively. The implantable pulse generator of each patient was activated at 1 month after surgery. At 1 month, 3 months, 6 months and 12 months after continuous neurostimulation, all patients were evaluated by the same scales above.ResultsThe BFMDRS movement scores decreased from 15.0±5.3 before surgery to 3.5±4.5 at 12 months after neurostimulation, with a mean improvement of 78% (p<0.001). The BFMDRS disability scores improved from 7.4±4.9 before surgery to 4.0±4.6 at 12 months after neurostimulation, with a mean improvement of 56% (p<0.001). The postoperative SF-36 scores had the remarkable improvement compared with baseline scores. Impaired sleep quality was found in 82% of patients and depression in 64% before surgery, which didn’t neither obtained amelioration after continuous neurostimulation.ConclusionsBilateral pallidal neurostimulation is a beneficial therapeutic option for refractory MS, which could improve the motor symptoms except for depression and sleep quality.

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Subthalamic nucleus deep brain stimulation in isolated dystonia

Neurology ◽

10.1212/wnl.0000000000003451 ◽

2016 ◽

Vol 88 (1) ◽

pp. 25-35 ◽

Cited By ~ 51

Author(s):

Jill L. Ostrem ◽

Marta San Luciano ◽

Kristen A. Dodenhoff ◽

Nathan Ziman ◽

Leslie C. Markun ◽

...

Keyword(s):

Deep Brain Stimulation ◽

Subthalamic Nucleus ◽

Brain Stimulation ◽

Rating Scale ◽

Spasmodic Torticollis ◽

Link Type ◽

Term Tolerability ◽

Stn Dbs ◽

Deep Brain

Objective:To report long-term safety and efficacy outcomes of a large cohort of patients with medically refractory isolated dystonia treated with subthalamic nucleus (STN) deep brain stimulation (DBS).Methods:Twenty patients (12 male, 8 female; mean age 49 ± 16.3 years) with medically refractory isolated dystonia were studied (14 were followed for 36 months). The primary endpoints were change in Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) motor score and Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) total score at 36 months compared to preoperative baseline. Multiple secondary outcomes were also assessed (ClinicalTrials.govNCT00773604).Results:Eighteen of 20 patients showed improvement 12 months after STN DBS with sustained benefit persisting for 3 years (n = 14). At 36 months, BFMDRS motor scores improved 70.4% from a mean 17.9 ± 8.5 to 5.3 ± 5.6 (p = 0.0002) and total TWSTRS scores improved 66.6% from a mean 41.0 ± 18.9 to 13.7 ± 17.9 (p = 0.0002). Improvement at 36 months was equivalent to that seen at 6 months. Disability and quality of life measures were also improved. Three hardware-related and 24 stimulation-related nonserious adverse events occurred between years 1 and 3 (including 4 patients with dyskinesia).Conclusions:This study offers support for long-term tolerability and sustained effectiveness of STN DBS in the treatment of severe forms of isolated dystonia.Classification of evidence:This study provides Class IV evidence that STN DBS decreases long-term dystonia severity in patients with medically refractory isolated dystonia.

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Location of Active Contacts in Patients with Primary Dystonia Treated with Globus Pallidus Deep Brain Stimulation

Operative Neurosurgery ◽

10.1227/01.neu.0000317396.16089.bc ◽

2008 ◽

Vol 62 (suppl_1) ◽

pp. ONS217-ONS225 ◽

Cited By ~ 20

Author(s):

Clement Hamani ◽

Elena Moro ◽

Cindy Zadikoff ◽

Yu-Yan Poon ◽

Andres M. Lozano

Keyword(s):

Parkinson’S Disease ◽

Parkinson's Disease ◽

Deep Brain Stimulation ◽

Globus Pallidus ◽

Brain Stimulation ◽

Cervical Dystonia ◽

Rating Scale ◽

Globus Pallidus Internus ◽

Generalized Dystonia ◽

Deep Brain

Abstract Objective: Deep brain stimulation of the globus pallidus internus has been used for the treatment of various forms of dystonia, but the factors influencing postoperative outcomes remain unknown. We compared the location of the contacts being used for stimulation (active contacts) in patients with cervical dystonia, generalized dystonia, and Parkinson's disease and correlated the results with clinical outcome. Methods: Postoperative magnetic resonance scans of 13 patients with cervical dystonia, six patients with generalized dystonia, and five patients with Parkinson's disease who underwent globus pallidus internus deep brain stimulation were analyzed. We assessed the location of the active contacts relative to the midcommisural point and in relation to the anteroposterior and mediolateral boundaries of the pallidum. Postoperative outcome was measured with the Toronto Western Spasmodic Torticollis Rating Scale (for cervical dystonia) and the Burke-Fahn-Marsden Dystonia Rating Scale (for generalized dystonia) during the last follow-up. Results: We found that the location of the active contacts relative to the midcom-misural point and the internal boundaries of the pallidum was similar across the groups. In our series, the contacts used for stimulation were clustered in the posterolateral region of the pallidum. Within that region, we found no correlation between the location of the contacts and postoperative outcome. Conclusion: The location of the active contacts used for globus pallidus internus deep brain stimulation was similar in patients with cervical dystonia, generalized dystonia, and Parkinson's disease.

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Relationship between higher rates of adverse events in deep brain stimulation using standardized prospective recording and patient outcomes

Neurosurgical FOCUS ◽

10.3171/2010.4.focus10100 ◽

2010 ◽

Vol 29 (2) ◽

pp. E4 ◽

Cited By ~ 20

Author(s):

Adam P. Burdick ◽

Hubert H. Fernandez ◽

Michael S. Okun ◽

Yueh-Yun Chi ◽

Charles Jacobson ◽

...

Keyword(s):

Deep Brain Stimulation ◽

Adverse Events ◽

Motor Function ◽

Brain Stimulation ◽

Rating Scale ◽

Short Form ◽

Obsessive Compulsive ◽

Compulsive Disorder ◽

Significant Difference ◽

Deep Brain

Object Adverse event (AE) rates for deep brain stimulation (DBS) are variable, due to various methodologies used for identifying, collecting, and reporting AEs. This lack of a prospective, standardized AE collection method is a shortcoming in the advancement of DBS. In this paper the authors disclose the standardized and prospectively recorded AE data from their institution, correlated with clinical outcome and quality of life (QOL) measures. Methods All patients who underwent operations at the authors' institution for Parkinson disease (PD), essential tremor, dystonia, other tremor, and obsessive-compulsive disorder were included. Complications occurring intraoperatively or within the first 180 days following surgery were recorded, analyzed, and classified as mild, moderate, or severe, regardless of their perceived relationship to the procedure. The presence, frequency, and severity of AEs were compared with the following outcome measurements: postoperative change in the QOL scales (Medical Outcomes Study 36-Item Short-Form Survey, 39-Item PD Questionnaire); motor scales (Tremor Rating Scale, Unified Dystonia Rating Scale, Unified PD Rating Scale); and Patient Global Impression Scale (PGIS). Results Two hundred seventy DBS procedures were performed in 198 patients. Three hundred AEs were recorded in 146 (54.1%) of the 270 procedures, and the AEs were recorded in 119 (60.1%) of 198 patients. Of the 198 patients, the maximum severity of AEs was mild in 28 (14.1%), moderate in 35 (17.7%), and severe in 56 (28.3%). Of the 300 AEs, 102 (34.1%) of 299 were mild, 106 (35.5%) were moderate, and 91 (30.4%) were severe. The AEs were classified as probably not stimulation induced in 10 (3.4%) of 297, probably in 44 (14.9%), unclear for 89 (30%), and not applicable to stimulation in 154 (51.9%). Adverse events were also classified as probably related to surgery in 111 (37.2%) of 298, possibly related in 96 (32.2%), and probably not related to surgery in 91 (30.5%). There was no significant difference (p = 0.22) in QOL outcomes among patients who had no AEs compared with those who experienced mild, moderate, or severe AEs. There was no significant difference in QOL outcomes between patients who did not experience an AE compared with those who experienced any AE. There was no significant difference in the mean General PGIS score between patients without an AE versus those with any AE, as well as on the Symptom-Specific PGIS. Motor function outcomes did not vary between patients with or without AEs. For patients with PD with or without AEs, there was no significant difference in preoperative off-medicine Unified PD Rating Scale score and postoperative 6-month on-medication/on-stimulation change scores (p = 0.59). For patients with tremor there were no differences between those with or without AEs on the Tremor Rating Scale for motor function or activities of daily living. Patients with dystonia with and without AEs showed no differences in the Unified Dystonia Rating Scale. Conclusions Prospectively and systematically recording AEs may result in higher AE rates, but this does not correlate with poorer QOL, motor function, or patient-oriented outcome scores.

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Prospective randomized double-blind trial of bilateral thalamic deep brain stimulation in adults with Tourette syndrome

Journal of Neurosurgery ◽

10.3171/jns-07/11/1004 ◽

2007 ◽

Vol 107 (5) ◽

pp. 1004-1014 ◽

Cited By ~ 191

Author(s):

Robert J. Maciunas ◽

Brian N. Maddux ◽

David E. Riley ◽

Christina M. Whitney ◽

Mike R. Schoenberg ◽

...

Keyword(s):

Deep Brain Stimulation ◽

Tourette Syndrome ◽

Outcome Measures ◽

Brain Stimulation ◽

Rating Scale ◽

Primary Outcome Measure ◽

Secondary Outcome ◽

Double Blind ◽

Exact Test ◽

Deep Brain

Object The severity of Tourette syndrome (TS) typically peaks just before adolescence and diminishes afterward. In some patients, however, TS progresses into adulthood, and proves to be medically refractory. The authors conducted a prospective double-blind crossover trial of bilateral thalamic deep brain stimulation (DBS) in five adults with TS. Methods Bilateral thalamic electrodes were implanted. An independent programmer established optimal stimulator settings in a single session. Subjective and objective results were assessed in a double-blind randomized manner for 4 weeks, with each week spent in one of four states of unilateral or bilateral stimulation. Results were similarly assessed 3 months after unblinded bilateral stimulator activation while repeated open programming sessions were permitted. Results In the randomized phase of the trial, a statistically significant (p < 0.03, Friedman exact test) reduction in the modified Rush Video-Based Rating Scale score (primary outcome measure) was identified in the bilateral on state. Improvement was noted in motor and sonic tic counts as well as on the Yale Global Tic Severity Scale and TS Symptom List scores (secondary outcome measures). Benefit was persistent after 3 months of open stimulator programming. Quality of life indices were also improved. Three of five patients had marked improvement according to all primary and secondary outcome measures. Conclusions Bilateral thalamic DBS appears to reduce tic frequency and severity in some patients with TS who have exhausted other available means of treatment.

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Target-Specific Effects of Deep Brain Stimulation for Tourette Syndrome: A Systematic Review and Meta-Analysis

Frontiers in Neurology ◽

10.3389/fneur.2021.769275 ◽

2021 ◽

Vol 12 ◽

Author(s):

Laura Wehmeyer ◽

Thomas Schüller ◽

Jana Kiess ◽

Petra Heiden ◽

Veerle Visser-Vandewalle ◽

...

Keyword(s):

Systematic Review ◽

Deep Brain Stimulation ◽

Tourette Syndrome ◽

Brain Stimulation ◽

Rating Scale ◽

Meta Analysis ◽

Secondary Outcome ◽

Centromedian Nucleus ◽

Treatment Refractory ◽

Deep Brain

Background: Extended research has pointed to the efficacy of deep brain stimulation (DBS) in treatment of patients with treatment-refractory Tourette syndrome (TS). The four most commonly used DBS targets for TS include the centromedian nucleus–nucleus ventrooralis internus (CM-Voi) and the centromedian nucleus–parafascicular (CM-Pf) complexes of the thalamus, and the posteroventrolateral (pvIGPi) and the anteromedial portion of the globus pallidus internus (amGPi). Differences and commonalities between those targets need to be compared systematically.Objective: Therefore, we evaluated whether DBS is effective in reducing TS symptoms and target-specific differences.Methods: A PubMed literature search was conducted according to the PRISMA guidelines. Eligible literature was used to conduct a systematic review and meta-analysis.Results: In total, 65 studies with 376 patients were included. Overall, Yale Global Tic Severity Scale (YGTSS) scores were reduced by more than 50 in 69% of the patients. DBS also resulted in significant reductions of secondary outcome measures, including the total YGTSS, modified Rush Video-Based Tic Rating Scale (mRVRS), Yale-Brown Obsessive Compulsive Scale (YBOCS), and Becks Depression Inventory (BDI). All targets resulted in significant reductions of YGTSS scores and, with the exception of the CM-Pf, also in reduced YBOCS scores. Interestingly, DBS of pallidal targets showed increased YGTSS and YBOCS reductions compared to thalamic targets. Also, the meta-analysis including six randomized controlled and double-blinded trials demonstrated clinical efficacy of DBS for TS, that remained significant for GPi but not thalamic stimulation in two separate meta-analyses.Conclusion: We conclude that DBS is a clinically effective treatment option for patients with treatment-refractory TS, with all targets showing comparable improvement rates. Future research might focus on personalized and symptom-specific target selection.

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Pallidal Deep Brain Stimulation in Patients with Prior Bilateral Pallidotomy and Selective Peripheral Denervation for Treatment of Dystonia

Stereotactic and Functional Neurosurgery ◽

10.1159/000509822 ◽

2020 ◽

pp. 1-5

Author(s):

Assel Saryyeva ◽

Hans Holger Capelle ◽

Thomas Mehari Kinfe ◽

Christoph Schrader ◽

Joachim K. Krauss

Keyword(s):

Deep Brain Stimulation ◽

Brain Stimulation ◽

Rating Scale ◽

Spasmodic Torticollis ◽

Term Outcome ◽

Long Term Outcome ◽

Generalized Dystonia ◽

A Prospective Study ◽

Deep Brain

Introduction: Deep brain stimulation (DBS) of the globus pallidus internus has become an accepted treatment for severe isolated idiopathic and inherited dystonia. Patients who had other forms of surgery earlier, such as radiofrequency lesioning or selective peripheral denervation, however, usually are not considered candidates for DBS. Objective: The aim of this study was to evaluate the long-term outcome of pallidal DBS in a rare subgroup of patients who had undergone both pallidotomy and selective peripheral denervation previously with a waning effect over the years. Methods: Pallidal DBS was performed according to a prospective study protocol in 2 patients with isolated idiopathic dystonia, and patients were followed for a period of at least 6 years. Results: Both patients benefitted from long-lasting amelioration of dystonia after pallidal DBS, which was comparable to that of patients who did not have previous surgeries. In a 62-year-old female with cervical dystonia both the Burke-Fahn-Marsden (BFM) and the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) motor scores were improved at follow-up 8 years after surgery (50 and 39%). In a 32-year-old male with generalized dystonia, the BFM motor and disability scores showed marked improvement at 6.5 years of follow-up (82 and 66%). Conclusions: Pallidal DBS can yield marked and long-lasting improvement in patients who underwent both pallidotomy and selective peripheral denervation earlier. Therefore, such patients, in general, should not be excluded from DBS.

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