Bilateral subthalamic nucleus stimulation in refractory status dystonicus

Barbosa, Breno José Alencar Pires; Carra, Rafael Bernhardt; Duarte, Kléber Paiva; Godinho, Fábio; Andrade, Daniel Ciampi de; Teixeira, Manoel Jacobsen; Barbosa, Egberto Reis; Cury, Rubens Gisbert

doi:10.1016/j.jns.2018.03.007

Cited by 7 publications

(15 citation statements)

References 20 publications

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“…Although most previous DBS treatment studies for pediatric dystonia have targeted the GPi, 9,11,20 some other studies have indicated that the STN could be a promising alternative DBS target for select pediatric patients with dystonia. 21,22 The present study substantiates and extends the latter view and studies by indicating that STN-DBS could serve as a safe and effective alternative treatment option for at least some cases of medically intractable pediatric isolated dystonia. In this context, it is noteworthy that we also found that the clinical effectiveness of STN-DBS was even maintained in one patient when the battery became depleted and not replaced and, in another patient, when the stimulation was turned off.…”

Section: Discussionsupporting

confidence: 85%

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

Zhang

et al. 2020

Can. J. Neurol. Sci.

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ABSTRACT:Objective:To evaluate the short-term and long-term clinical effectiveness and safety of subthalamic nucleus deep brain stimulation (STN-DBS) for medically intractable pediatric isolated dystonia.Methods:Using a longitudinal retrospective design, we assessed the clinical outcomes of nine patients who underwent STN-DBS for treatment-refractory pediatric isolated dystonia one decade ago (mean age at surgery: 15.9 ± 4.5 years). The primary clinical outcome used was assessed by retrospective video analyses of patients’ dystonia symptoms using the Burke–Fahn–Marsden Dystonia Rating Scale (BFMDRS). Clinical assessments were performed at baseline, 1-year follow-up (1-yr FU), and 10-year follow-up (10-yr FU). Adverse side effects, including surgery-related, device-related, and stimulation-related effects, were also documented.Results:After STN-DBS surgery, the mean improvement in the BFMDRS motor score was 77.1 ± 26.6% at 1-yr FU and 90.4 ± 10.4% at 10-yr FU. Similarly, the mean BFMDRS disability score was improved by 69.5 ± 13.6% at 1-yr FU and by 86.5 ± 13.9% at 10-yr FU. The clinical improvements gained at 10-yr FU were significantly larger than those observed at 1-yr FU. Negative correlations were found between the duration of disease to age at surgery ratio (DD/AS) and the improvements in the BFMDRS motor score and total score at 1-yr FU and 10-yr FU.Conclusion:To our knowledge, this study provides the first clinical evidence for the short- and long-term effectiveness and safety of STN-DBS for pediatric isolated dystonia. Additionally, putative evidence is provided that earlier STN-DBS intervention in patients with refractory pediatric isolated dystonia may improve short- and long-term clinical outcomes.

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Section: Discussionsupporting

confidence: 85%

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

Zhang

et al. 2020

Can. J. Neurol. Sci.

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“…Of note, STN DBS has also been used as a salvage treatment in a range of dystonias previously treated with pallidotomy or GPi DBS with good results as well as in status dystonicus [42,[64][65][66][67][68][69][70]. These reports therefore supported our choice of STN as a target in this cohort of patients with post stroke dystonia and a structurally normal STN in whom the GPi could not be targeted confidently, either because of its direct damage as shown on the MRI or its close proximity to the infarct raising concern about potential functional involvement.…”

Section: Discussionmentioning

confidence: 99%

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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“…The remainder of the published cases received unilateral GPI-DBS with or without contralateral GPI lesioning (109). A few case reports described good dystonia outcomes in children post subthalamic nucleus (STN) DBS (110)(111)(112). STN has been chosen often in children with GPI lesions (metabolic strokes) or postfailed GPI-DBS.…”

Section: Deep Brain Stimulation Targetsmentioning

confidence: 99%

Approach to the Treatment of Pediatric Dystonia

Gorodetsky¹,

Fasano

2022

Dystonia

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Dystonia is the most common movement disorder in the pediatric population. It can affect normal motor development and cause significant motor disability. The treatment of pediatric dystonia can be very challenging as many children tend to be refractory to standard pharmacological interventions. Pharmacological treatment remains the first-line approach in pediatric dystonia. However, despite the widespread use of different ani-dystonia medications, the literature is limited to small clinical studies, case reports, and experts’ opinions. Botulinum neurotoxin (BoNT) is a well-established treatment in adults with focal and segmental dystonia. Despite the widespread use of BoNT in adult dystonia the data to support its use in children is limited with the majority extrapolated from the spasticity literature. For the last 2 decades, deep brain stimulation (DBS) has been used for a wide variety of dystonic conditions in adults and children. DBS gained increased popularity in the pediatric population because of the dramatic positive outcomes reported in some forms of genetic dystonia and the subsequent consensus that DBS is generally safe and effective. This review summarizes the available evidence supporting the efficacy and safety of pharmacological treatment, BoNT, and DBS in pediatric dystonia and provides practical frameworks for the adoption of these modalities.

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Bilateral subthalamic nucleus stimulation in refractory status dystonicus

Cited by 7 publications

References 20 publications

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

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

Subthalamic Nucleus Deep Brain Stimulation in Post-Infarct Dystonia

Approach to the Treatment of Pediatric Dystonia

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