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@ARTICLE{Brggemann:137808,
      author       = {Brüggemann, Norbert and Kühn, Andrea and Schneider,
                      Susanne A and Kamm, Christoph and Wolters, Alexander and
                      Krause, Patricia and Moro, Elena and Steigerwald, Frank and
                      Wittstock, Matthias and Tronnier, Volker and Lozano, Andres
                      M and Hamani, Clement and Poon, Yu-Yan and Zittel, Simone
                      and Wächter, Tobias and Deuschl, Günther and Krüger,
                      Rejko and Kupsch, Andreas and Münchau, Alexander and
                      Lohmann, Katja and Volkmann, Jens and Klein, Christine},
      title        = {{S}hort- and long-term outcome of chronic pallidal
                      neurostimulation in monogenic isolated dystonia.},
      journal      = {Neurology},
      volume       = {84},
      number       = {9},
      issn         = {0028-3878},
      address      = {[S.l.]},
      publisher    = {Ovid},
      reportid     = {DZNE-2020-04130},
      pages        = {895-903},
      year         = {2015},
      abstract     = {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.},
      keywords     = {Adolescent / Adult / Child / Child, Preschool / Deep Brain
                      Stimulation: methods / Deep Brain Stimulation: trends /
                      Dystonia: diagnosis / Dystonia: therapy / Electrodes,
                      Implanted / Female / Follow-Up Studies / Globus Pallidus:
                      physiopathology / Humans / Male / Middle Aged /
                      Retrospective Studies / Treatment Outcome / Young Adult},
      cin          = {Ext UKT / AG Gasser 1},
      ddc          = {610},
      cid          = {I:(DE-2719)5000058 / I:(DE-2719)1210000},
      pnm          = {345 - Population Studies and Genetics (POF3-345)},
      pid          = {G:(DE-HGF)POF3-345},
      typ          = {PUB:(DE-HGF)16},
      pubmed       = {pmid:25653290},
      pmc          = {pmc:PMC6170184},
      doi          = {10.1212/WNL.0000000000001312},
      url          = {https://pub.dzne.de/record/137808},
}