137808 20240321220323.0 10.1212/WNL.0000000000001312 doi pmid:25653290 pmid pmc:PMC6170184 pmc 0028-3878 ISSN 1526-632X ISSN altmetric:3401942 altmetric DZNE-2020-04130 English 610 Brüggemann, Norbert P:(DE-HGF)0 0 Corresponding author Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia. [S.l.] 2015 Ovid online Crossref Ovid Technologies (Wolters Kluwer Health) 2015-02-04 print Crossref Ovid Technologies (Wolters Kluwer Health) 2015-03-03 article DRIVER Output Types/Journal article DataCite Journal Article journal journal PUB:(DE-HGF)16 1588685551_1067 PUB:(DE-HGF) ARTICLE BibTeX JOURNAL_ARTICLE ORCID Journal Article 0 EndNote 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. 345 - Population Studies and Genetics (POF3-345) G:(DE-HGF)POF3-345 POF3-345 POF III 0 Dataset connected to CrossRef, PubMed, Adolescent MeSH Adult MeSH Child MeSH Child, Preschool MeSH Deep Brain Stimulation: methods MeSH Deep Brain Stimulation: trends MeSH Dystonia: diagnosis MeSH Dystonia: therapy MeSH Electrodes, Implanted MeSH Female MeSH Follow-Up Studies MeSH Globus Pallidus: physiopathology MeSH Humans MeSH Male MeSH Middle Aged MeSH Retrospective Studies MeSH Treatment Outcome MeSH Young Adult MeSH Kühn, Andrea P:(DE-HGF)0 1 Schneider, Susanne A 2 Kamm, Christoph P:(DE-HGF)0 3 Wolters, Alexander 4 Krause, Patricia 5 Moro, Elena 6 Steigerwald, Frank 7 Wittstock, Matthias 8 Tronnier, Volker 9 Lozano, Andres M 10 Hamani, Clement 11 Poon, Yu-Yan 12 Zittel, Simone 13 Wächter, Tobias P:(DE-2719)9000333 14 dzne Deuschl, Günther 15 Krüger, Rejko P:(DE-2719)2811170 16 dzne Kupsch, Andreas 17 Münchau, Alexander 18 Lohmann, Katja P:(DE-HGF)0 19 Volkmann, Jens 20 Klein, Christine 21 10.1212/wnl.0000000000001312 : Ovid Technologies (Wolters Kluwer Health), 2015-02-04 9 895-903 journal-article Crossref Neurology 84 2015 0028-3878 10.1212/WNL.0000000000001312 Vol. 84, no. 9, p. 895 - 903 PERI:(DE-600)1491874-2 9 84:9<895 - 903 895-903 Neurology 84 2015 0028-3878 Pubmed Central http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6170184 oai:pub.dzne.de:137808 VDB Deutsches Zentrum für Neurodegenerative Erkrankungen I:(DE-588)1065079516 DZNE 14 P:(DE-2719)9000333 Deutsches Zentrum für Neurodegenerative Erkrankungen I:(DE-588)1065079516 DZNE 16 P:(DE-2719)2811170 DE-HGF Forschungsbereich Gesundheit Erkrankungen des Nervensystems G:(DE-HGF)POF3-340 G:(DE-HGF)POF3-345 G:(DE-HGF)POF3-300 Population Studies and Genetics 0 2015 Allianz-Lizenz StatID:(DE-HGF)0410 StatID JCR StatID:(DE-HGF)0100 StatID NEUROLOGY : 2017 DBCoverage StatID:(DE-HGF)0200 StatID SCOPUS DBCoverage StatID:(DE-HGF)0300 StatID Medline DBCoverage StatID:(DE-HGF)0310 StatID NCBI Molecular Biology Database DBCoverage StatID:(DE-HGF)0320 StatID PubMed Central DBCoverage StatID:(DE-HGF)0199 StatID Clarivate Analytics Master Journal List WoS StatID:(DE-HGF)0110 StatID Science Citation Index DBCoverage StatID:(DE-HGF)0150 StatID Web of Science Core Collection WoS StatID:(DE-HGF)0111 StatID Science Citation Index Expanded DBCoverage StatID:(DE-HGF)1110 StatID Current Contents - Clinical Medicine DBCoverage StatID:(DE-HGF)1030 StatID Current Contents - Life Sciences DBCoverage StatID:(DE-HGF)1050 StatID BIOSIS Previews IF >= 5 StatID:(DE-HGF)9905 StatID NEUROLOGY : 2017 I:(DE-2719)5000058 Ext UKT Ext Universitätsklinikum Tübingen 0 I:(DE-2719)1210000 AG Gasser 1 Parkinson Genetics 1 journal VDB I:(DE-2719)5000058 I:(DE-2719)1210000 UNRESTRICTED