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

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Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia. / Brüggemann, Norbert; Kühn, Andrea; Schneider, Susanne A; Kamm, Christoph; Wolters, Alexander; Krause, Patricia; Moro, Elena; Steigerwald, Frank; Wittstock, Matthias; Tronnier, Volker; Lozano, Andres M; Hamani, Clement; Poon, Yu-Yan; Zittel-Dirks, Simone; Wächter, Tobias; Deuschl, Günther; Krüger, Rejko; Kupsch, Andreas; Münchau, Alexander; Lohmann, Katja; Volkmann, Jens; Klein, Christine.

In: NEUROLOGY, Vol. 84, No. 9, 03.03.2015, p. 895-903.

Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review

Harvard

Brüggemann, N, Kühn, A, Schneider, SA, Kamm, C, Wolters, A, Krause, P, Moro, E, Steigerwald, F, Wittstock, M, Tronnier, V, Lozano, AM, Hamani, C, Poon, Y-Y, Zittel-Dirks, S, Wächter, T, Deuschl, G, Krüger, R, Kupsch, A, Münchau, A, Lohmann, K, Volkmann, J & Klein, C 2015, 'Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia', NEUROLOGY, vol. 84, no. 9, pp. 895-903. https://doi.org/10.1212/WNL.0000000000001312

APA

Brüggemann, N., Kühn, A., Schneider, S. A., Kamm, C., Wolters, A., Krause, P., Moro, E., Steigerwald, F., Wittstock, M., Tronnier, V., Lozano, A. M., Hamani, C., Poon, Y-Y., Zittel-Dirks, S., Wächter, T., Deuschl, G., Krüger, R., Kupsch, A., Münchau, A., ... Klein, C. (2015). Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia. NEUROLOGY, 84(9), 895-903. https://doi.org/10.1212/WNL.0000000000001312

Vancouver

Brüggemann N, Kühn A, Schneider SA, Kamm C, Wolters A, Krause P et al. Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia. NEUROLOGY. 2015 Mar 3;84(9):895-903. https://doi.org/10.1212/WNL.0000000000001312

Bibtex

@article{1a6ad8f7622a474b850d9b50368060b5,
title = "Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia",
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.",
keywords = "Adolescent, Adult, Child, Child, Preschool, Deep Brain Stimulation, Dystonia, Electrodes, Implanted, Female, Follow-Up Studies, Globus Pallidus, Humans, Male, Middle Aged, Retrospective Studies, Treatment Outcome, Young Adult, Case Reports, Journal Article, Multicenter Study, Research Support, N.I.H., Extramural, Research Support, Non-U.S. Gov't",
author = "Norbert Br{\"u}ggemann and Andrea K{\"u}hn and Schneider, {Susanne A} and Christoph Kamm and Alexander Wolters and Patricia Krause and Elena Moro and Frank Steigerwald and Matthias Wittstock and Volker Tronnier and Lozano, {Andres M} and Clement Hamani and Yu-Yan Poon and Simone Zittel-Dirks and Tobias W{\"a}chter and G{\"u}nther Deuschl and Rejko Kr{\"u}ger and Andreas Kupsch and Alexander M{\"u}nchau and Katja Lohmann and Jens Volkmann and Christine Klein",
note = "{\textcopyright} 2015 American Academy of Neurology.",
year = "2015",
month = mar,
day = "3",
doi = "10.1212/WNL.0000000000001312",
language = "English",
volume = "84",
pages = "895--903",
journal = "NEUROLOGY",
issn = "0028-3878",
publisher = "Lippincott Williams and Wilkins",
number = "9",

}

RIS

TY - JOUR

T1 - Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia

AU - Brüggemann, Norbert

AU - Kühn, Andrea

AU - Schneider, Susanne A

AU - Kamm, Christoph

AU - Wolters, Alexander

AU - Krause, Patricia

AU - Moro, Elena

AU - Steigerwald, Frank

AU - Wittstock, Matthias

AU - Tronnier, Volker

AU - Lozano, Andres M

AU - Hamani, Clement

AU - Poon, Yu-Yan

AU - Zittel-Dirks, Simone

AU - Wächter, Tobias

AU - Deuschl, Günther

AU - Krüger, Rejko

AU - Kupsch, Andreas

AU - Münchau, Alexander

AU - Lohmann, Katja

AU - Volkmann, Jens

AU - Klein, Christine

N1 - © 2015 American Academy of Neurology.

PY - 2015/3/3

Y1 - 2015/3/3

N2 - 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.

AB - 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.

KW - Adolescent

KW - Adult

KW - Child

KW - Child, Preschool

KW - Deep Brain Stimulation

KW - Dystonia

KW - Electrodes, Implanted

KW - Female

KW - Follow-Up Studies

KW - Globus Pallidus

KW - Humans

KW - Male

KW - Middle Aged

KW - Retrospective Studies

KW - Treatment Outcome

KW - Young Adult

KW - Case Reports

KW - Journal Article

KW - Multicenter Study

KW - Research Support, N.I.H., Extramural

KW - Research Support, Non-U.S. Gov't

U2 - 10.1212/WNL.0000000000001312

DO - 10.1212/WNL.0000000000001312

M3 - SCORING: Journal article

C2 - 25653290

VL - 84

SP - 895

EP - 903

JO - NEUROLOGY

JF - NEUROLOGY

SN - 0028-3878

IS - 9

ER -