The minimal clinically important change in the motor section of the Burke-Fahn-Marsden Dystonia Rating Scale for generalized dystonia: Results from deep brain stimulation

2021 ◽  
Author(s):  
Esther Cubo Delgado ◽  
Joan Miravite ◽  
Sara Calvo ◽  
Kathryn Cooper ◽  
Deborah Raymond ◽  
...  
Keyword(s):  
Deep Brain Stimulation ◽  
Brain Stimulation ◽  
Rating Scale ◽  
Important Change ◽  
Generalized Dystonia ◽  
Clinically Important Change ◽  
Deep Brain

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

2008 ◽  
Vol 62 (suppl_1) ◽  
pp. ONS217-ONS225 ◽  
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.

Pallidal deep brain stimulation for longstanding severe generalized dystonia in Hallervorden—Spatz syndrome

2004 ◽  
Vol 100 (4) ◽  
pp. 706-709 ◽  
Author(s):  
Atsushi Umemura ◽  
Jurg L. Jaggi ◽  
Carol A. Dolinskas ◽  
Matthew B. Stern ◽  
Gordon H. Baltuch
Keyword(s):  
Deep Brain Stimulation ◽  
Brain Stimulation ◽  
Rating Scale ◽  
Motor Functioning ◽  
Content Type ◽  
Pallidal Stimulation ◽  
Upper And Lower Extremities ◽  
Generalized Dystonia ◽  
Fine Print ◽  
Deep Brain

✓ Generalized dystonia is one of the most disabling movement disorders. Ablative stereotactic surgery such as pallidotomy has been performed for medically refractory dystonia. Recently, deep brain stimulation (DBS) has appeared as an alternative to ablative procedures. Nevertheless, there have been few published reports detailing improvement in dystonia with DBS. This 36-year-old man with Hallervorden—Spatz syndrome suffered from intractable primary generalized dystonia for 28 years. He was completely dependent for activities of daily living and wheelchair bound because of continuous severe dystonic movements in the face, tongue, neck, trunk, and upper and lower extremities while at rest. The Burke-Fahn-Marsden (BFM) Dystonia Rating Scale score was 112 (maximum 120 points). Bilateral DBS of the globus pallidus internus was performed and resulted in marked improvement in motor functioning and dystonic symptoms with a significant reduction in disability. The BFM score improved to 22.5 points (80% improvement) at 3 months postsurgery and the patient's dystonia was still well suppressed 1 year after surgery. Bilateral pallidal stimulation is an effective and safe treatment for intractable generalized dystonia in Hallervorden—Spatz syndrome, even if the disability is severe and longstanding.

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

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

<b><i>Introduction:</i></b> 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. <b><i>Objective:</i></b> 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. <b><i>Methods:</i></b> 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. <b><i>Results:</i></b> 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%). <b><i>Conclusions:</i></b> 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.

Mapping efficacious deep brain stimulation for pediatric dystonia

2021 ◽  
pp. 1-11
Author(s):  
Ailish Coblentz ◽  
Gavin J. B. Elias ◽  
Alexandre Boutet ◽  
Jurgen Germann ◽  
Musleh Algarni ◽  
...  
Keyword(s):  
Deep Brain Stimulation ◽  
Clinical Outcome ◽  
Brain Stimulation ◽  
Pediatric Patients ◽  
Rating Scale ◽  
Structural Connectivity ◽  
Response To Treatment ◽  
Internal Globus Pallidus ◽  
Probabilistic Map ◽  
Deep Brain

OBJECTIVEThe objective of this study was to report the authors’ experience with deep brain stimulation (DBS) of the internal globus pallidus (GPi) as a treatment for pediatric dystonia, and to elucidate substrates underlying clinical outcome using state-of-the-art neuroimaging techniques.METHODSA retrospective analysis was conducted in 11 pediatric patients (6 girls and 5 boys, mean age 12 ± 4 years) with medically refractory dystonia who underwent GPi-DBS implantation between June 2009 and September 2017. Using pre- and postoperative MRI, volumes of tissue activated were modeled and weighted by clinical outcome to identify brain regions associated with clinical outcome. Functional and structural networks associated with clinical benefits were also determined using large-scale normative data sets.RESULTSA total of 21 implanted leads were analyzed in 11 patients. The average follow-up duration was 19 ± 20 months (median 5 months). Using a 7-point clinical rating scale, 10 patients showed response to treatment, as defined by scores < 3. The mean improvement in the Burke-Fahn-Marsden Dystonia Rating Scale motor score was 40% ± 23%. The probabilistic map of efficacy showed that the voxel cluster most associated with clinical improvement was located at the posterior aspect of the GPi, comparatively posterior and superior to the coordinates of the classic GPi target. Strong functional and structural connectivity was evident between the probabilistic map and areas such as the precentral and postcentral gyri, parietooccipital cortex, and brainstem.CONCLUSIONSThis study reported on a series of pediatric patients with dystonia in whom GPi-DBS resulted in variable clinical benefit and described a clinically favorable stimulation site for this cohort, as well as its structural and functional connectivity. This information could be valuable for improving surgical planning, simplifying programming, and further informing disease pathophysiology.

Pallidal Deep Brain Stimulation for Primary Dystonia in Children

Neurosurgery ◽  
2011 ◽  
Vol 68 (3) ◽  
pp. 738-743 ◽  
Author(s):  
Abilash Haridas ◽  
Michele Tagliati ◽  
Irene Osborn ◽  
Ioannis Isaias ◽  
Yakov Gologorsky ◽  
...  
Keyword(s):  
Deep Brain Stimulation ◽  
Clinical Improvement ◽  
Brain Stimulation ◽  
Functional Disability ◽  
Rating Scale ◽  
Retrospective Chart Review ◽  
Consecutive Series ◽  
Internal Globus Pallidus ◽  
Deep Brain

Abstract BACKGROUND: Deep brain stimulation (DBS) at the internal globus pallidus (GPi) has replaced ablative procedures for the treatment of primary generalized dystonia (PGD) because it is adjustable, reversible, and yields robust clinical improvement that appears to be long lasting. OBJECTIVE: To describe the long-term responses to pallidal DBS of a consecutive series of 22 pediatric patients with PGD. METHODS: Retrospective chart review of 22 consecutive PGD patients, ≤21 years of age treated by one DBS team over an 8-year period. The Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) was used to evaluate symptom severity and functional disability, pre- and post-operatively. Adverse events and medication changes were also noted. RESULTS: The median follow-up was 2 years (range, 1-8 years). All 22 patients reached 1-year follow-up; 14 reached 2 years, and 11 reached 3 years. The BFMDRS motor subscores were improved 84%, 93%, and 94% (median) at these time points. These motor responses were matched by equivalent improvements in function, and the response to DBS resulted in significant reductions in oral and intrathecal medication requirements after 12 and 24 months of stimulation. There were no hemorrhages or neurological complications related to surgery and no adverse effects from stimulation. Significant hardware-related complications were noted, in particular, infection (14%), which delayed clinical improvement. CONCLUSION: Pallidal DBS is a safe and effective treatment for PGD in patients &lt;21 years of age. The improvement appears durable. Improvement in device design should reduce hardware-related complications over time.

scholarly journals Pediatric Deep Brain Stimulation Using Awake Recording and Stimulation for Target Selection in an Inpatient Neuromodulation Monitoring Unit

2018 ◽  
Vol 8 (7) ◽  
pp. 135 ◽  
Author(s):  
Terence D. Sanger ◽  
Mark Liker ◽  
Enrique Arguelles ◽  
Ruta Deshpande ◽  
Arash Maskooki ◽  
...  
Keyword(s):  
Deep Brain Stimulation ◽  
Basal Ganglia ◽  
Brain Stimulation ◽  
Rating Scale ◽  
Target Selection ◽  
Test Stimulation ◽  
Depth Electrodes ◽  
A New Technique ◽  
High Degree ◽  
Deep Brain

Deep brain stimulation (DBS) for secondary (acquired, combined) dystonia does not reach the high degree of efficacy achieved in primary (genetic, isolated) dystonia. We hypothesize that this may be due to variability in the underlying injury, so that different children may require placement of electrodes in different regions of basal ganglia and thalamus. We describe a new targeting procedure in which temporary depth electrodes are placed at multiple possible targets in basal ganglia and thalamus, and probing for efficacy is performed using test stimulation and recording while children remain for one week in an inpatient Neuromodulation Monitoring Unit (NMU). Nine Children with severe secondary dystonia underwent the NMU targeting procedure. In all cases, 4 electrodes were implanted. We compared the results to 6 children who had previously had 4 electrodes implanted using standard intraoperative microelectrode targeting techniques. Results showed a significant benefit, with 80% of children with NMU targeting achieving greater than 5-point improvement on the Burke–Fahn–Marsden Dystonia Rating Scale (BFMDRS), compared with 50% of children using intraoperative targeting. NMU targeting improved BFMDRS by an average of 17.1 whereas intraoperative targeting improved by an average of 10.3. These preliminary results support the use of test stimulation and recording in a Neuromodulation Monitoring Unit (NMU) as a new technique with the potential to improve outcomes following DBS in children with secondary (acquired) dystonia. A larger sample size will be needed to confirm these results.

Subthalamic nucleus deep brain stimulation after bilateral pallidotomy in the treatment of generalized dystonia

2014 ◽  
Vol 20 (1) ◽  
pp. 131-133 ◽  
Author(s):  
Malgorzata Dec ◽  
Marcin Tutaj ◽  
Monika Rudzińska ◽  
Andrzej Szczudlik ◽  
Henryk Koziara ◽  
...  
Keyword(s):  
Deep Brain Stimulation ◽  
Subthalamic Nucleus ◽  
Brain Stimulation ◽  
Generalized Dystonia ◽  
Deep Brain

scholarly journals Deep brain stimulation treatment in dystonia: a bibliometric analysis

2020 ◽  
Vol 78 (9) ◽  
pp. 586-592
Author(s):  
Clarice LISTIK ◽  
Eduardo LISTIK ◽  
Rubens Gisbert CURY ◽  
Egberto Reis BARBOSA ◽  
Manoel Jacobsen TEIXEIRA ◽  
...  
Keyword(s):  
Deep Brain Stimulation ◽  
Bibliometric Analysis ◽  
Brain Stimulation ◽  
The United States ◽  
Scientific Journals ◽  
Long Term Outcomes ◽  
Generalized Dystonia ◽  
Research Domain ◽  
Deep Brain

ABSTRACT Background: Dystonia is a heterogeneous disorder that, when refractory to medical treatment, may have a favorable response to deep brain stimulation (DBS). A practical way to have an overview of a research domain is through a bibliometric analysis, as it makes it more accessible for researchers and others outside the field to have an idea of its directions and needs. Objective: To analyze the 100 most cited articles in the use of DBS for dystonia treatment in the last 30 years. Methods: The research protocol was performed in June 2019 in Elsevier’s Scopus database, by retrieving the most cited articles regarding DBS in dystonia. We analyzed authors, year of publication, country, affiliation, and targets of DBS. Results: Articles are mainly published in Movement Disorders (19%), Journal of Neurosurgery (9%), and Neurology (9%). European countries offer significant contributions (57% of our sample). France (192.5 citations/paper) and Germany (144.1 citations/paper) have the highest citation rates of all countries. The United States contributes with 31% of the articles, with 129.8 citations/paper. The publications are focused on General outcomes (46%), followed by Long-term outcomes (12.5%), and Complications (11%), and the leading type of dystonia researched is idiopathic or inherited, isolated, segmental or generalized dystonia, with 27% of articles and 204.3 citations/paper. Conclusions: DBS in dystonia research is mainly published in a handful of scientific journals and focused on the outcomes of the surgery in idiopathic or inherited, isolated, segmental or generalized dystonia, and with globus pallidus internus as the main DBS target.

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