Subthalamic Nucleus Stimulation in Pediatric Isolated Dystonia: A 10-Year Follow-up

Xu, Wenying; Li, Hongxia; Zhang, Chencheng; Sun, Bomin; Wu, Yiwen; Li, Dianyou

doi:10.1017/cjn.2020.32

Cited by 14 publications

(14 citation statements)

References 29 publications

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“…Shorter disease duration correlated positively with larger DBS treatment efficacy, with a more persistent improvement and larger treatment potential with STN stimulation. Similar experience has been described by others [39][40][41][42][43][44][45][46][47][48][49][50][51][52][53]. STN DBS has also been successfully used in the management of Meige syndrome with motor score improvements in the range of 42.1-74% [54][55][56][57] and in tardive dystonia with improvements of 88-100% at up to 12-year follow-up periods [41,58,59].…”

Section: Discussionsupporting

confidence: 64%

Subthalamic Nucleus Deep Brain Stimulation in Post-Infarct Dystonia

Tambirajoo

Furlanetti

Samuel

et al. 2020

Stereotact Funct Neurosurg

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Dystonia secondary to cerebral infarcts presents months to years after the initial insult, is usually unilateral and causes significant morbidity. Deep brain stimulation (DBS) of the globus pallidus internus (GPi) is established as the most frequent target in the management of the dystonic symptoms. We report our experience with subthalamic nucleus (STN) DBS in 3 patients with post-infarct dystonia, in whom GPi DBS was not confidently possible due to the presence of striatal infarcts. Two patients had unilateral STN DBS implantation, whereas the third patient had bilateral STN DBS implantation for bilateral dystonic symptoms. Prospectively collected preoperative and postoperative functional assessment data including imaging, medication and neuropsychology evaluations were analyzed with regard to symptom improvement. Median follow-up period was 38.3 months (range 26-43 months). All patients had clinically valuable improvements in dystonic symptoms and pain control despite variable improvements in the Burke-Fahn-Marsden dystonia rating scores. In our series, we have demonstrated that STN DBS could be an alternative in the management of post-infarct dystonia in patients with abnormal striatal anatomy which precludes GPi DBS. A multidisciplinary teambased approach is essential for patient selection and management.

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Section: Discussionsupporting

confidence: 64%

Subthalamic Nucleus Deep Brain Stimulation in Post-Infarct Dystonia

Tambirajoo

Furlanetti

Samuel

et al. 2020

Stereotact Funct Neurosurg

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“…There is no data directly comparing GPi versus subthalamic nucleus (STN) or thalamic stimulation in the pediatric population. STN stimulation demonstrated good outcomes in one pediatric study of 9 patients with isolated dystonia [35]. A phase 1 trial exploring bilateral thalamic DBS targeting the nucleus ventralis intermedius/ ventral oral posterior (Vim/Vop) in pediatric patients with acquired dystonia showed outcomes comparable to pallidal stimulation [64].…”

Section: Surgical Considerations In Pediatric Dbsmentioning

confidence: 99%

“…DBS is associated with a significant decrease in medication requirements and sometimes in complete elimination of dystonia medications [21]. Limited series have reported long-term benefits in pediatric cohorts, at 10 years and beyond [35, 73].…”

Section: Long-term Outcomesmentioning

confidence: 99%

“…Blinded studies comparing pallidal DBS to sham stimulation in isolated generalized and segmental dystonia showed Burke-Fahn-Marsden dystonia rating score (BFMDRS) improvements ranging from 39.3% at 3 months to 54.6% at 12 months [14,15]. Additional studies have confirmed remarkable shortterm benefits in children, albeit with some variability depending on the underlying etiology, in a wide variety of dystonia subtypes (Table 1) [8,[16][17][18][19][20][21][22][23][24][25][26][27][28][29][30][31][32][33][34][35].…”

Section: Introductionmentioning

confidence: 99%

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Long-Term Outcomes of Deep Brain Stimulation for Pediatric Dystonia

Malatt

Tagliati²

2022

Pediatr Neurosurg

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Background Deep brain stimulation (DBS) has been utilized for over two decades to treat medication refractory dystonia in children. Short-term benefit has been demonstrated for inherited, isolated and idiopathic cases, with less efficacy in heredodegenerative and acquired dystonias. The ongoing publication of long-term outcomes warrants a critical assessment of available information as pediatric patients are expected to live most of their lives with these implants. Summary We performed a review of the literature for data describing motor and neuropsychiatric outcomes, in addition to complications, five or more years after DBS placement in patients undergoing DBS surgery for dystonia at an age younger than 21. We identified 20 articles including individual data on long-term motor outcomes after DBS for a total of 78 patients. In addition, we found five articles reporting long-term outcomes after DBS in nine patients with status dystonicus. The majority of patients were implanted within the globus pallidus internus, with only a few cases targeting the subthalamic nucleus and ventrolateral posterior nucleus of the thalamus. The average follow-up was 8.5 years, with a range up to 22 years. Long-term outcomes showed a sustained motor benefit, with median Burke-Fahn-Marsden dystonia rating score improvement ranging from 2.5% to 93.2% in different dystonia subtypes. Patients with inherited, isolated and idiopathic dystonias had greater improvement than those with heredodegenerative and acquired dystonias. Sustained improvements in quality of life were also reported, without the development of significant cognitive or psychiatric co-morbidities. Late adverse events tended to be hardware-related, with minimal stimulation-induced effects. Key Messages While data regarding long-term outcomes is somewhat limited, particularly with regards to neuropsychiatric outcomes and adverse events, improvement in motor outcomes appear to be preserved more than 5 years after DBS placement.

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“…A dual-target, crossover sham-controlled study has directly compared the clinical effects of GPi and STN stimulations in patients with dystonia, which indicated that the STN was a more effective target for dystonia than the GPi (17). STN-DBS is believed to improve the repetitive motions, abnormal posture, and quality of life of refractory patients with isolated dystonia (12,13,15,(18)(19)(20)(21). The motor outcomes of STN-DBS in the treatment of dystonia have been extensively studied (12)(13)(14)(15)(16)(18)(19)(20)(21).…”

Section: Introductionmentioning

confidence: 99%

Weight Change After Subthalamic Nucleus Deep Brain Stimulation in Patients With Isolated Dystonia

Lai

et al. 2021

Front. Neurol.

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Purpose: Deep brain stimulation of the subthalamic nucleus (STN-DBS) is an effective treatment method for advanced Parkinson's disease (PD) and isolated dystonia and provides marked improvement of major motor symptoms. In addition, non-motor effects have been reported including weight gain (WG) in patients with PD after STN-DBS. However, it is still unclear whether patients with isolated dystonia also experience WG.Methods: Data from 47 patients with isolated dystonia who underwent bilateral STN-DBS surgery between October 2012 and June 2019 were retrospectively collected. The severity of dystonia was assessed via the Burke–Fahn–Marsden Dystonia Rating Scale (BFMDRS). Changes in the body mass index (BMI) and BFMDRS score were analyzed using paired Student's t-tests. Regression analysis was performed to identify factors that affected the BMI after surgery.Results: Postoperative WG was observed in 78.7% of patients. The percentage of overweight and obese patients increased from 25.5% (before STN-DBS) to 48.9% (at the last follow-up). The mean BMI and mean percentage change in BMI increased by 1.32 ± 1.83 kg/m2 (P < 0.001) and 6.28 ± 8.34%, respectively. BMI increased more in female than in male patients. At the last follow-up, BFMDRS movement and disability scores improved by 69.76 ± 33.23% and 65.66 ± 31.41%, respectively (both P < 0.001). The final regression model analysis revealed that sex and preoperative BMI alone were independently associated with BMI change (P < 0.05).Conclusions: STN-DBS is associated with postoperative WG with patients with isolated dystonia. WG is more prominent in female patients and is associated with preoperative weight but not with the efficacy of STN-DBS on motor symptoms.

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Subthalamic Nucleus Stimulation in Pediatric Isolated Dystonia: A 10-Year Follow-up

Cited by 14 publications

References 29 publications

Subthalamic Nucleus Deep Brain Stimulation in Post-Infarct Dystonia

Subthalamic Nucleus Deep Brain Stimulation in Post-Infarct Dystonia

Long-Term Outcomes of Deep Brain Stimulation for Pediatric Dystonia

Weight Change After Subthalamic Nucleus Deep Brain Stimulation in Patients With Isolated Dystonia

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