001     137808
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024 7 _ |a 10.1212/WNL.0000000000001312
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024 7 _ |a pmc:PMC6170184
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024 7 _ |a 0028-3878
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024 7 _ |a 1526-632X
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024 7 _ |a altmetric:3401942
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037 _ _ |a DZNE-2020-04130
041 _ _ |a English
082 _ _ |a 610
100 1 _ |a Brüggemann, Norbert
|0 P:(DE-HGF)0
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|e Corresponding author
245 _ _ |a Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia.
260 _ _ |a [S.l.]
|c 2015
|b Ovid
264 _ 1 |3 online
|2 Crossref
|b Ovid Technologies (Wolters Kluwer Health)
|c 2015-02-04
264 _ 1 |3 print
|2 Crossref
|b Ovid Technologies (Wolters Kluwer Health)
|c 2015-03-03
336 7 _ |a article
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336 7 _ |a ARTICLE
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336 7 _ |a Journal Article
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520 _ _ |a Deep brain stimulation of the internal pallidum (GPi-DBS) is an established therapeutic option in treatment-refractory dystonia, and the identification of factors predicting surgical outcome is needed to optimize patient selection.In this retrospective multicenter study, GPi-DBS outcome of 8 patients with DYT6, 9 with DYT1, and 38 with isolated dystonia without known monogenic cause (non-DYT) was assessed at early (1-16 months) and late (22-92 months) follow-up using Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) scores.At early follow-up, mean reduction of dystonia severity was greater in patients with DYT1 (BFMDRS score: -60%) and non-DYT dystonia (-52%) than in patients with DYT6 dystonia (-32%; p = 0.046). Accordingly, the rate of responders was considerably lower in the latter group (57% vs >90%; p = 0.017). At late follow-up, however, GPi-DBS resulted in comparable improvement in all 3 groups (DYT6, -42%; DYT1, -44; non-DYT, -61%). Additional DBS of the same or another brain target was performed in 3 of 8 patients with DYT6 dystonia with varying results. Regardless of the genotype, patients with a shorter duration from onset of dystonia to surgery had better control of dystonia postoperatively.Long-term GPi-DBS is effective in patients with DYT6, DYT1, and non-DYT dystonia. However, the effect of DBS appears to be less predictable in patients with DYT6, suggesting that pre-DBS genetic testing and counseling for known dystonia gene mutations may be indicated. GPi-DBS should probably be considered earlier in the disease course.This study provides Class IV evidence that long-term GPi-DBS improves dystonia in patients with DYT1, DYT6, and non-DYT dystonia.
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650 _ 2 |a Adolescent
|2 MeSH
650 _ 2 |a Adult
|2 MeSH
650 _ 2 |a Child
|2 MeSH
650 _ 2 |a Child, Preschool
|2 MeSH
650 _ 2 |a Deep Brain Stimulation: methods
|2 MeSH
650 _ 2 |a Deep Brain Stimulation: trends
|2 MeSH
650 _ 2 |a Dystonia: diagnosis
|2 MeSH
650 _ 2 |a Dystonia: therapy
|2 MeSH
650 _ 2 |a Electrodes, Implanted
|2 MeSH
650 _ 2 |a Female
|2 MeSH
650 _ 2 |a Follow-Up Studies
|2 MeSH
650 _ 2 |a Globus Pallidus: physiopathology
|2 MeSH
650 _ 2 |a Humans
|2 MeSH
650 _ 2 |a Male
|2 MeSH
650 _ 2 |a Middle Aged
|2 MeSH
650 _ 2 |a Retrospective Studies
|2 MeSH
650 _ 2 |a Treatment Outcome
|2 MeSH
650 _ 2 |a Young Adult
|2 MeSH
700 1 _ |a Kühn, Andrea
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700 1 _ |a Schneider, Susanne A
|b 2
700 1 _ |a Kamm, Christoph
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700 1 _ |a Wolters, Alexander
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700 1 _ |a Krause, Patricia
|b 5
700 1 _ |a Moro, Elena
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700 1 _ |a Steigerwald, Frank
|b 7
700 1 _ |a Wittstock, Matthias
|b 8
700 1 _ |a Tronnier, Volker
|b 9
700 1 _ |a Lozano, Andres M
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700 1 _ |a Hamani, Clement
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700 1 _ |a Poon, Yu-Yan
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700 1 _ |a Zittel, Simone
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700 1 _ |a Wächter, Tobias
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700 1 _ |a Deuschl, Günther
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700 1 _ |a Krüger, Rejko
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700 1 _ |a Kupsch, Andreas
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700 1 _ |a Münchau, Alexander
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700 1 _ |a Lohmann, Katja
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700 1 _ |a Volkmann, Jens
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700 1 _ |a Klein, Christine
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773 1 8 |a 10.1212/wnl.0000000000001312
|b : Ovid Technologies (Wolters Kluwer Health), 2015-02-04
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773 _ _ |a 10.1212/WNL.0000000000001312
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856 7 _ |2 Pubmed Central
|u http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6170184
909 C O |o oai:pub.dzne.de:137808
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