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, 6)
,
Jin Xie-Brustolin
(9)
,
Maria-Clara Coelho Braga
(4)
,
Claire Ardouin
(3)
,
Virginie Czernecki
(1, 6)
,
Hélène Klinger
(10, 11)
,
Stephan Chabardes
(4)
,
Eric Seigneuret
(4, 12)
,
Patrick Mertens
(13)
,
Emmanuel Cuny
(8)
,
Soledad Navarro
(6)
,
Philippe Cornu
(6)
,
Alim-Louis Benabid
(14)
,
Jean-Francois Le Bas
(15, 4)
,
Didier Dormont
(6, 16)
,
Marc Hermier
(17)
,
Kathy Dujardin
(18)
,
Serge Blond
(19)
,
Pierre Krystkowiak
(20)
,
Alain Destée
(18)
,
Eric Bardinet
(1, 16, 21)
,
Yves Agid
(6)
,
Paul Krack
(4, 5)
,
Emmanuel Broussolle
(22)
,
Pierre Pollak
(4, 5)
,
Non Renseigné
1
CRICM -
Centre de Recherche de l'Institut du Cerveau et de la Moelle épinière
2 CIC AP-HP (pitie-Salpetriere)/inserm
3 UM des troubles du mouvement
4 GIN - Grenoble Institut des Neurosciences
5 Département de neurologie
6 CHU Pitié-Salpêtrière [AP-HP]
7 ISC-MJ - Institut des sciences cognitives Marc Jeannerod - Centre de neuroscience cognitive - UMR5229
8 PPSC - Physiologie et physiopathologie de la signalisation cellulaire
9 Service de neurologie
10 Service de neurologie C [Hôpital Pierre Wertheimer - HCL]
11 UCBL - Université Claude Bernard Lyon 1
12 Département de neurochirurgie
13 Service de Neurochirurgie [Hopital Pierre Wertheimer - HCL]
14 CEA-LETI - Commissariat à l'énergie atomique et aux énergies alternatives - Laboratoire d'Electronique et de Technologie de l'Information
15 RBCH - RMN biomédicale : de la cellule à l'homme
16 NEMESIS-CRICM - Equipe NEMESIS - Centre de Recherches de l'Institut du Cerveau et de la Moelle épinière
17 Service de neuroradiologie [Lyon]
18 Département de neurologie [Lille]
19 JPArc - Centre de Recherche Jean-Pierre AUBERT Neurosciences et Cancer - U837
20 Service de neurologie [Amiens]
21 CENIR - Plateforme de neuroimagerie CENIR = Center for NeuroImaging Research-Human MRI Neuroimaging core facility for clinical research [ICM Paris]
22 Hôpital neurologique et neurochirurgical Pierre Wertheimer [CHU - HCL]
2 CIC AP-HP (pitie-Salpetriere)/inserm
3 UM des troubles du mouvement
4 GIN - Grenoble Institut des Neurosciences
5 Département de neurologie
6 CHU Pitié-Salpêtrière [AP-HP]
7 ISC-MJ - Institut des sciences cognitives Marc Jeannerod - Centre de neuroscience cognitive - UMR5229
8 PPSC - Physiologie et physiopathologie de la signalisation cellulaire
9 Service de neurologie
10 Service de neurologie C [Hôpital Pierre Wertheimer - HCL]
11 UCBL - Université Claude Bernard Lyon 1
12 Département de neurochirurgie
13 Service de Neurochirurgie [Hopital Pierre Wertheimer - HCL]
14 CEA-LETI - Commissariat à l'énergie atomique et aux énergies alternatives - Laboratoire d'Electronique et de Technologie de l'Information
15 RBCH - RMN biomédicale : de la cellule à l'homme
16 NEMESIS-CRICM - Equipe NEMESIS - Centre de Recherches de l'Institut du Cerveau et de la Moelle épinière
17 Service de neuroradiologie [Lyon]
18 Département de neurologie [Lille]
19 JPArc - Centre de Recherche Jean-Pierre AUBERT Neurosciences et Cancer - U837
20 Service de neurologie [Amiens]
21 CENIR - Plateforme de neuroimagerie CENIR = Center for NeuroImaging Research-Human MRI Neuroimaging core facility for clinical research [ICM Paris]
22 Hôpital neurologique et neurochirurgical Pierre Wertheimer [CHU - HCL]
Marie Vidailhet
- Fonction : Auteur
- PersonId : 758783
- ORCID : 0000-0002-2409-9143
- IdRef : 074466208
Valerie Fraix
- Fonction : Auteur
- PersonId : 761299
- ORCID : 0000-0003-4473-9993
David Grabli
- Fonction : Auteur
- PersonId : 760487
- ORCID : 0000-0001-6798-4567
Stephane Thobois
- Fonction : Auteur
- PersonId : 900551
Patrick Mertens
- Fonction : Auteur
- PersonId : 1298847
- ORCID : 0000-0001-5272-261X
Didier Dormont
- Fonction : Auteur
- PersonId : 1349450
- ORCID : 0000-0002-6080-1286
- IdRef : 075211238
Marc Hermier
- Fonction : Auteur
- PersonId : 1144595
- ORCID : 0000-0002-4461-2346
Pierre Krystkowiak
- Fonction : Auteur
- PersonId : 1149993
- IdRef : 059356502
Eric Bardinet
- Fonction : Auteur
Emmanuel Broussolle
- Fonction : Auteur
- PersonId : 1333391
- ORCID : 0000-0002-0410-9519
- IdRef : 032277261
Non Renseigné
- Fonction : Auteur
Résumé
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.