Title |
Long-term results of deep brain stimulation in a cohort of eight children with isolated dystonia
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Published in |
Journal of Neurology, August 2016
|
DOI | 10.1007/s00415-016-8253-6 |
Pubmed ID | |
Authors |
P. Krause, K. Lauritsch, A. Lipp, A. Horn, B. Weschke, A. Kupsch, K. L. Kiening, G.-H. Schneider, A. A. Kühn |
Abstract |
Pallidal deep brain stimulation (DBS) is an established treatment for patients with severe isolated dystonia. However, clinical evidence for the long-term use of DBS in children is limited and controlled trials have not yet been conducted. Here, we provide the long-term results of up to 13 years of pallidal DBS in eight pediatric patients with generalized idiopathic or hereditary isolated dystonia (five males, mean age at surgery 12.5 ± 3.5 years), as assessed by retrospective video rating. Video rating was performed at three time points: pre-operative, 1-year short-term follow-up (1y-FU) and long-term last FU (LT-FU, up to 13 years). Symptom severity and disability were assessed using the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS). Disability scores were obtained from clinical charts and during the last FU. The mean improvement in BFMDRS motor score was 54.4 ± 8.9 % at 1y-FU and 42.9 ± 11.6 % at LT-FU; the disability scores improved by 59.8 ± 10.3 and 63.3 ± 7.8 %, respectively. Electrode dislocation was noted in one patient and implantable pulse generator dislocation in another, both requiring surgical intervention; no further serious adverse events occurred. Our study presents the first blinded video rating assessment of the short- and long-term effects of pallidal DBS in children with idiopathic or hereditary isolated dystonia. Results confirm that pallidal DBS is a safe and efficacious long-term treatment in children, with overall motor improvement similar to that described in controlled trials in adults. |
Mendeley readers
Geographical breakdown
Country | Count | As % |
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Unknown | 43 | 100% |
Demographic breakdown
Readers by professional status | Count | As % |
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Researcher | 9 | 21% |
Student > Master | 6 | 14% |
Student > Bachelor | 4 | 9% |
Student > Postgraduate | 4 | 9% |
Other | 3 | 7% |
Other | 7 | 16% |
Unknown | 10 | 23% |
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Pharmacology, Toxicology and Pharmaceutical Science | 1 | 2% |
Computer Science | 1 | 2% |
Social Sciences | 1 | 2% |
Other | 3 | 7% |
Unknown | 14 | 33% |