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

Bruggemann, N.; Kuhn, 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, S.; Wachter, T.; Deuschl, G.; Krüger, Rejko; Kupsch, A.; Munchau, A.; Lohmann, K.; Volkmann, J.; Klein, C.

doi:10.1212/WNL.0000000000001312

Reference : Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolat...


	Document type :	Scientific journals : Article
	Discipline(s) :	Human health sciences : Neurology
	To cite this reference:	http://hdl.handle.net/10993/22700

Title :	Short- and long-term outcome of chronic pallidal neurostimulation in monogenic isolated dystonia
Language :	English
Author, co-author :	Bruggemann, N. [> >]
	Kuhn, 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, S. [> >]
	Wachter, T. [> >]
	Deuschl, G. [> >]
	Krüger, Rejko [University of Luxembourg > Faculty of Science, Technology and Communication (FSTC) > Life Science Research Unit]
	Kupsch, A. [> >]
	Munchau, A. [> >]
	Lohmann, K. [> >]
	Volkmann, J. [> >]
	Klein, C. []
Publication date :	2015
Journal title :	Neurology
Publisher :	American Academy of Neurology
Volume :	84
Issue/season :	9
Pages :	895-903
Peer reviewed :	Yes (verified by ORBi^lu)
ISSN :	0028-3878
e-ISSN :	1526-632X
Country :	United States
Abstract :	[en] 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.
Research centres :	Luxembourg Centre for Systems Biomedicine (LCSB): Clinical & Experimental Neuroscience (Krüger Group)
Permalink :	http://hdl.handle.net/10993/22700
DOI :	10.1212/WNL.0000000000001312

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