Bilateral pallidal deep brain stimulation for the treatment of patients with dystonia-choreoathetosis cerebral palsy: a prospective pilot study

被引：230

作者：

Vidailhet, Marie ^{[1
,2
]}

Yelnik, Jerome ^{[1
,2
]}

Lagrange, Christelle ^{[3
]}

Fraix, Valerie ^{[3
]}

Grabli, David ^{[1
,2
]}

Thobois, Stephane ^{[4
,5
]}

Burbaud, Pierre ^{[6
]}

Welter, Marie-Laure ^{[1
,2
]}

Xie-Brustolin, Jin ^{[4
,5
]}

Braga, Maria-Clara Coelho ^{[3
]}

Ardouin, Claire ^{[3
]}

Czernecki, Virginie ^{[1
,2
]}

Klinger, Helene ^{[4
,5
]}

Chabardes, Stephan ^{[7
]}

Seigneuret, Eric ^{[7
]}

Mertens, Patrick ^{[8
]}

Cuny, Emmanuel ^{[9
]}

Navarro, Soledad ^{[10
]}

Cornu, Philippe

Benabid, Alim-Louis ^{[7
]}

Le Bas, Jean-Francois ^{[11
]}

Dormont, Didier ^{[12
,13
]}

Hermier, Marc ^{[14
]}

Dujardin, Kathy ^{[15
]}

Blond, Serge ^{[16
]}

Krystkowiak, Pierre ^{[17
]}

Destee, Alain ^{[15
]}

Bardinet, Eric ^{[2
]}

Agid, Yves ^{[1
,2
]}

Krack, Paul ^{[3
]}

Broussolle, Emmanuel ^{[4
,5
]}

Pollak, Pierre ^{[3
]}

机构：

[1] Grp Hosp Pitie Salpetriere, Federat Neurol, AP HP, F-75013 Paris, France

[2] Univ Paris 06, INSERM, UMRS 975, CNRS,UMR 7225, F-75013 Paris, France

[3] Univ Hosp Grenoble, Dept Neurol, Grenoble, France

[4] Hop Neurol & Neurochirurg P Wertheimer, Serv Neurol C, Hosp Civils Lyon, Lyon, France

[5] CNRS, UMR 52 29, Bron, France

[6] Univ Victor Segalen, CNRS, Hop Pellegrin, Dept Neurophysiol,UMR 5543, F-33076 Bordeaux, France

[7] Univ Grenoble 1, INSERM, Inst Neurosci, Univ Hosp Grenoble,Dept Neurosurg,U836, Grenoble, France

[8] Univ Lyon 1, Hop Neurol Pierre Wertheimer, Dept Neurosurg, Fac Med, F-69365 Lyon, France

[9] Univ Victor Segalen, CNRS, Dept Neurosurg, UMR 5543, Bordeaux, France

[10] Univ Paris 06, Grp Hosp Pitie Salpetriere, Dept Neurosurg, AP HP, Paris, France

[11] Univ Hosp Grenoble, Dept Neuroradiol, Grenoble, France

[12] Univ Paris 06, Dept Neuroradiol, Grp Hosp Pitie Salpetriere, AP HP, Paris, France

[13] LENA CNRS, Lab Neurosci Cognit & Imagerie Cerebrale, UMR S 975, Paris, France

[14] Hosp Civils Lyon, Hop Neurol, Dept Neuroradiol, Lyon, France

[15] Lille Univ Hosp, Dept Neurol, IFR114, Lille, France

[16] CHRU Lille, Hop Roger Salengro, Serv Neurochirurg Fonct & Stereotax, Lille, France

[17] Amiens Univ Hosp, Dept Neurol, Amiens, France

来源：

LANCET NEUROLOGY | 2009年 / 8卷 / 08期

关键词：

PRIMARY GENERALIZED DYSTONIA; GLOBUS-PALLIDUS; FOLLOW-UP; INTERNUS; DISEASE; ATLAS; SCALE;

D O I：

10.1016/S1474-4422(09)70151-6

中图分类号：

R74 [神经病学与精神病学];

学科分类号：

摘要：

Background Cerebral palsy (CP) with dystonia-choreoathetosis is a common cause of disability in children and in adults, and responds poorly to medical treatment. Bilateral pallidal deep brain stimulation (BP-DBS) of the globus pallidus internus (GPi) is an effective treatment for primary dystonia, but the effect of this reversible surgical procedure on dystonia-cboreoathetosis CP, which is a subtype of secondary dystonia, is unknown. Our aim was to test the effectiveness of BP-DBS in adults with dystonia-choreoathetosis CP. Methods We did a multicentre prospective pilot study of BP-DBS in 13 adults with dystonia-choreoathetosis CP who had no cognitive impairment, little spasticity, and only slight abnormalities of the basal ganglia on MRI. The primary endpoint was change in the severity of dystonia-choreoathetosis after 1 year of neurostimulation, as assessed with the Burke-Fahn-Marsden dystonia rating scale. The accuracy of surgical targeting to the GPi was assessed masked to the results of neurostimulation. Analysis was by intention to treat. Findings The mean Burke-Fahn-Marsden dystonia rating scale movement score improved from 44-2 (SD 21-1) before surgery to 34.7 (21-9) at 1 year post-operatively (p=0.009; mean improvement 24.4 [21.1]%, 95% CI 11.6-37.1). Functional disability, pain, and mental health-related quality of life were significantly improved. There was no worsening of cognition or mood. Adverse events were related to stimulation (arrest of the stimulator in one patient, and an adjustment to the current intensity in four patients). The optimum therapeutic target was the posterolateroventral region of the GPi. Little improvement was seen when the neurostimulation diffused to adjacent structures (mainly to the globus pallidus externus [GPe]). Interpretation Bilateral pallidal neurostimulation could be an effective treatment option for patients with dystonia-choreoathetosis CP. However, given the heterogeneity of motor outcomes and the small sample size, results should be interpreted with caution. The optimum placement of the leads seemed to be a crucial, but not exclusive, factor that could affect a good outcome.

引用

页码：709 / 717

页数：9

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