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

Marie Vidailhet 1 Jerome Yelnik 1, 2 Christelle Lagrange 3 Valerie Fraix 4, 5 David Grabli 1, 6 Stephane Thobois 7 Pierre Burbaud 8 Marie-Laure Welter 1, 2, 9 Jin Xie-Brustolin 10 Maria-Clara Coelho Braga 4 Claire Ardouin 3 Virginie Czernecki 1, 11 Hélène Klinger 12, 13 Stephan Chabardes 4 Eric Seigneuret 4, 14 Patrick Mertens 15 Emmanuel Cuny 8 Soledad Navarro 6 Philippe Cornu 6 Alim-Louis Benabid 16 Jean-Francois Le Bas 17, 4 Didier Dormont 18, 19 Marc Hermier 20 Kathy Dujardin 21 Serge Blond 22 Pierre Krystkowiak 23 Alain Destée 21 Eric Bardinet 1, 19, 24 Yves Agid 25 Paul Krack 4, 5 Emmanuel Broussolle 26 Pierre Pollak 4, 5 Non Renseigné 
19 NEMESIS-CRICM - Equipe NEMESIS - Centre de Recherches de l'Institut du Cerveau et de la Moelle épinière
CRICM - Centre de Recherche de l'Institut du Cerveau et de la Moelle épinière
Abstract : 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-choreoathetosis 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. FUNDING: National PHRC; Cerebral Palsy Foundation: Fondation Motrice/APETREIMC; French INSERM Dystonia National Network; Medtronic.
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Marie Vidailhet, Jerome Yelnik, Christelle Lagrange, Valerie Fraix, David Grabli, et al.. Bilateral pallidal deep brain stimulation for the treatment of patients with dystonia-choreoathetosis cerebral palsy: a prospective pilot study.. The Lancet Neurology, Elsevier, 2009, 8 (8), pp.709-17. ⟨10.1016/S1474-4422(09)70151-6⟩. ⟨hal-00805447⟩



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