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000137808 0247_ $$2ISSN$$a1526-632X
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000137808 041__ $$aEnglish
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000137808 1001_ $$0P:(DE-HGF)0$$aBrüggemann, Norbert$$b0$$eCorresponding author
000137808 245__ $$aShort- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia.
000137808 260__ $$a[S.l.]$$bOvid$$c2015
000137808 264_1 $$2Crossref$$3online$$bOvid Technologies (Wolters Kluwer Health)$$c2015-02-04
000137808 264_1 $$2Crossref$$3print$$bOvid Technologies (Wolters Kluwer Health)$$c2015-03-03
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000137808 520__ $$aDeep 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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000137808 650_2 $$2MeSH$$aAdolescent
000137808 650_2 $$2MeSH$$aAdult
000137808 650_2 $$2MeSH$$aChild
000137808 650_2 $$2MeSH$$aChild, Preschool
000137808 650_2 $$2MeSH$$aDeep Brain Stimulation: methods
000137808 650_2 $$2MeSH$$aDeep Brain Stimulation: trends
000137808 650_2 $$2MeSH$$aDystonia: diagnosis
000137808 650_2 $$2MeSH$$aDystonia: therapy
000137808 650_2 $$2MeSH$$aElectrodes, Implanted
000137808 650_2 $$2MeSH$$aFemale
000137808 650_2 $$2MeSH$$aFollow-Up Studies
000137808 650_2 $$2MeSH$$aGlobus Pallidus: physiopathology
000137808 650_2 $$2MeSH$$aHumans
000137808 650_2 $$2MeSH$$aMale
000137808 650_2 $$2MeSH$$aMiddle Aged
000137808 650_2 $$2MeSH$$aRetrospective Studies
000137808 650_2 $$2MeSH$$aTreatment Outcome
000137808 650_2 $$2MeSH$$aYoung Adult
000137808 7001_ $$0P:(DE-HGF)0$$aKühn, Andrea$$b1
000137808 7001_ $$aSchneider, Susanne A$$b2
000137808 7001_ $$0P:(DE-HGF)0$$aKamm, Christoph$$b3
000137808 7001_ $$aWolters, Alexander$$b4
000137808 7001_ $$aKrause, Patricia$$b5
000137808 7001_ $$aMoro, Elena$$b6
000137808 7001_ $$aSteigerwald, Frank$$b7
000137808 7001_ $$aWittstock, Matthias$$b8
000137808 7001_ $$aTronnier, Volker$$b9
000137808 7001_ $$aLozano, Andres M$$b10
000137808 7001_ $$aHamani, Clement$$b11
000137808 7001_ $$aPoon, Yu-Yan$$b12
000137808 7001_ $$aZittel, Simone$$b13
000137808 7001_ $$0P:(DE-2719)9000333$$aWächter, Tobias$$b14$$udzne
000137808 7001_ $$aDeuschl, Günther$$b15
000137808 7001_ $$0P:(DE-2719)2811170$$aKrüger, Rejko$$b16$$udzne
000137808 7001_ $$aKupsch, Andreas$$b17
000137808 7001_ $$aMünchau, Alexander$$b18
000137808 7001_ $$0P:(DE-HGF)0$$aLohmann, Katja$$b19
000137808 7001_ $$aVolkmann, Jens$$b20
000137808 7001_ $$aKlein, Christine$$b21
000137808 77318 $$2Crossref$$3journal-article$$a10.1212/wnl.0000000000001312$$b : Ovid Technologies (Wolters Kluwer Health), 2015-02-04$$n9$$p895-903$$tNeurology$$v84$$x0028-3878$$y2015
000137808 773__ $$0PERI:(DE-600)1491874-2$$a10.1212/WNL.0000000000001312$$gVol. 84, no. 9, p. 895 - 903$$n9$$p895-903$$q84:9<895 - 903$$tNeurology$$v84$$x0028-3878$$y2015
000137808 8567_ $$2Pubmed Central$$uhttp://www.ncbi.nlm.nih.gov/pmc/articles/PMC6170184
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