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March 03, 2015; 84 (9) Article

Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia

Norbert Brüggemann, Andrea Kühn, Susanne A. Schneider, Christoph Kamm, Alexander Wolters, Patricia Krause, Elena Moro, Frank Steigerwald, Matthias Wittstock, Volker Tronnier, Andres M. Lozano, Clement Hamani, Yu-Yan Poon, Simone Zittel, Tobias Wächter, Günther Deuschl, Rejko Krüger, Andreas Kupsch, Alexander Münchau, Katja Lohmann, Jens Volkmann, Christine Klein
First published February 4, 2015, DOI: https://doi.org/10.1212/WNL.0000000000001312
Norbert Brüggemann
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Andrea Kühn
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Susanne A. Schneider
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Christoph Kamm
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Alexander Wolters
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Patricia Krause
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Elena Moro
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Frank Steigerwald
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Matthias Wittstock
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Volker Tronnier
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Andres M. Lozano
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Clement Hamani
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Yu-Yan Poon
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Simone Zittel
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Tobias Wächter
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Günther Deuschl
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Rejko Krüger
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Andreas Kupsch
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Alexander Münchau
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Katja Lohmann
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Jens Volkmann
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Christine Klein
From the Institute of Neurogenetics (N.B., S.A.S., S.Z., A.M., K.L., C. Klein), University of Lübeck; Department of Neurology (N.B.), University Hospital Schleswig-Holstein, Campus Lübeck; Department of Neurology (A. Kühn, P.K.), Virchow Clinics, University Berlin Charité; Department of Neurology (S.A.S., G.D.), University Hospital Schleswig-Holstein, Campus Kiel; Department of Neurology (C. Kamm, A.W., M.W.), University Hospital Rostock, Germany; Movement Disorders Center (E.M., Y.-Y.P.), Toronto Western Hospital, University of Toronto, UHN, Canada; Movement Disorders Unit (E.M.), Division of Psychiatry and Neurology, CHU Grenoble, Joseph Fourier University, Grenoble, France; Department of Neurology (F.S., J.V.), University Hospital Würzburg; Department of Neurosurgery (V.T.), University Hospital Lübeck, Germany; Division of Neurosurgery (A.M.L., C.H.), Department of Surgery, University of Toronto, Canada; Center for Neurology and Hertie-Institute for Clinical Brain Research (T.W., R.K.), University Hospital Tübingen, Center for Integrative Neurosciences, University of Tübingen and German Center for Neurodegenerative Diseases (DZNE), Tübingen; Clinical and Experimental Neuroscience (R.K.), Luxembourg Centre for Systems Biomedicine, University of Luxembourg; and Department of Neurology and Stereotactic Neurosurgery (A. Kupsch), Basal Ganglia Research Group, Otto von Guericke University Magdeburg, Germany.
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Citation
Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia
Norbert Brüggemann, Andrea Kühn, Susanne A. Schneider, Christoph Kamm, Alexander Wolters, Patricia Krause, Elena Moro, Frank Steigerwald, Matthias Wittstock, Volker Tronnier, Andres M. Lozano, Clement Hamani, Yu-Yan Poon, Simone Zittel, Tobias Wächter, Günther Deuschl, Rejko Krüger, Andreas Kupsch, Alexander Münchau, Katja Lohmann, Jens Volkmann, Christine Klein
Neurology Mar 2015, 84 (9) 895-903; DOI: 10.1212/WNL.0000000000001312

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Abstract

Objectives: 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.

Methods: 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.

Results: 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.

Conclusions: 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.

Classification of evidence: This study provides Class IV evidence that long-term GPi-DBS improves dystonia in patients with DYT1, DYT6, and non-DYT dystonia.

GLOSSARY

ANOVA=
analysis of variance;
BFMDRS=
Burke-Fahn-Marsden Dystonia Rating Scale;
DBS=
deep brain stimulation;
GPi=
globus pallidus interna;
THAP1=
THAP domain containing, apoptosis associated protein 1;
TOR1A=
torsin family 1, member A (torsin A);
Vim=
ventralis intermedius

Footnotes

  • Go to Neurology.org for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Supplemental data at Neurology.org

  • Received June 19, 2014.
  • Accepted in final form November 12, 2014.
  • © 2015 American Academy of Neurology
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