Evidence map›Paper›PMID 39468897›Full record

SynthesisEuropean journal of neurology2025

Deep brain stimulation for severe dystonia associated with Wilson disease: A prospective multicenter meta-analysis of an N-of-1 trial.

Chloé Laurencin, Aurelia Poujois, Maxime Bonjour, Caroline Demily, Hélène Klinger, Emmanuel Roze, Victoire Leclert, Teodor Danaila, Carole Langlois-Jacques, Eduardo Couchonnal and 14 more

Registry-linked trialAbstract readMeta-Analysis
PubMed Full text at PMC Publisher
In one paragraph

Synthesis in European journal of neurology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT02552628 (DEEP BRAIN STIMULATION FOR SEVERE DYSTONIA ASSOCIATED WITH WILSON'S DISEASE. A Prospective Multicenter Meta-analysis of Nof1 Trials), which is not on this map. Cited by 1 paper, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed, 1 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT02552628 nacompletednot on this map

DEEP BRAIN STIMULATION FOR SEVERE DYSTONIA ASSOCIATED WITH WILSON'S DISEASE. A Prospective Multicenter Meta-analysis of Nof1 Trials

TypeinterventionalSponsorHospices Civils de LyonRan2016 to 2022Enrolled5ConditionsSevere Dystonia, Wilson's DiseaseArmsMedtronic, Activa® PC "on", Medtronic, Activa® PC "off"
3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

24 authors.

Chloé LaurencinDepartment of Neurology C, Parkinson Expert Center, Pierre Wertheimer Neurological Hospital, Hospices Civils de Lyon, Bron, France.ORCID 0000-0002-1516-5401
Aurelia PoujoisNational Reference Center for Wilson Disease and Other Copper-Related Rare Diseases, Neurology Department, Rothschild Foundation Hospital, Paris, France.
Maxime BonjourDepartment of Biostatistics, Hospices Civils de Lyon, Lyon, France.
Caroline DemilyReference Center for Rare Diseases With Psychiatric Phenotype Génopsy, Le Vinatier Hospital, Bron, France.
Hélène KlingerDepartment of Neurology C, Parkinson Expert Center, Pierre Wertheimer Neurological Hospital, Hospices Civils de Lyon, Bron, France.
Emmanuel RozeSorbonne University, INSERM, CNRS, Paris, France.
Victoire LeclertDepartment of Neurology C, Parkinson Expert Center, Pierre Wertheimer Neurological Hospital, Hospices Civils de Lyon, Bron, France.
Teodor DanailaDepartment of Neurology C, Parkinson Expert Center, Pierre Wertheimer Neurological Hospital, Hospices Civils de Lyon, Bron, France.ORCID 0000-0001-5072-4486
Carole Langlois-JacquesDepartment of Biostatistics, Hospices Civils de Lyon, Lyon, France.
Eduardo CouchonnalNational Reference Center for Wilson Disease and Other Copper-Related Rare Diseases, Femme Mère Enfant Hospital, Hospices Civils de Lyon, Bron, France.
France WoimantNational Reference Center for Wilson Disease and Other Copper-Related Rare Diseases, Neurology Department, Rothschild Foundation Hospital, Paris, France.
Mickael Alexandre ObadiaNational Reference Center for Wilson Disease and Other Copper-Related Rare Diseases, Neurology Department, Rothschild Foundation Hospital, Paris, France.
Gwennaelle PerezNational Reference Center for Wilson Disease and Other Copper-Related Rare Diseases, Neurology Department, Rothschild Foundation Hospital, Paris, France.
Michaela PernonNational Reference Center for Wilson Disease and Other Copper-Related Rare Diseases, Neurology Department, Rothschild Foundation Hospital, Paris, France.
Laurianne BlanchetDepartment of Neurology C, Parkinson Expert Center, Pierre Wertheimer Neurological Hospital, Hospices Civils de Lyon, Bron, France.
Emmanuel BroussolleDepartment of Neurology C, Parkinson Expert Center, Pierre Wertheimer Neurological Hospital, Hospices Civils de Lyon, Bron, France.
Marie VidailhetSorbonne University, INSERM, CNRS, Paris, France.
Behrouz KassaiCentre d'Investigation Clinique 1407, Hospices Civils de Lyon, Louis Pradel Hospital, Bron, France.
Elena MoroDivision of Neurology CHU Grenoble Alpes, Grenoble Institute of Neurosciences, INSERM U1216, Grenoble Alpes University, Grenoble, France.
Carine KarachiNeurosurgery Department, Hôpital de la Salpêtrière, Groupe Hospitalier Pitié-Salpêtrière, Assistance Publique-Hôpitaux de Paris, Paris, France.
Gustavo PoloNeurosurgery Department A, Hospices Civils de Lyon, Pierre Wertheimer Neurological Hospital, Bron, France.
David GrabliSorbonne University, INSERM, CNRS, Paris, France.
Aurélie PortefaixCentre d'Investigation Clinique 1407, Hospices Civils de Lyon, Louis Pradel Hospital, Bron, France.
Stéphane ThoboisDepartment of Neurology C, Parkinson Expert Center, Pierre Wertheimer Neurological Hospital, Hospices Civils de Lyon, Bron, France.

Funding

Ministère de l'Education Nationale, de l'Enseignement Superieur et de la Recherche 69HCL14_0448_1
6 · The paper itself

Abstract

background and purposeDisabling dystonia despite optimal medical treatment is common in Wilson disease (WD). No controlled study has evaluated the effect of deep brain stimulation (DBS) on dystonia related to WD. This study was undertaken to evaluate the efficacy of DBS on dystonia related to WD.

methodsA meta-analysis of an N-of-1 prospective, randomized, double-blind, multicenter DBS study was conducted at two French WD reference centers. Main inclusion criteria were patients with WD, stabilized for at least 6 months with significant disability due to dystonia despite optimized medical treatment. The subthalamic nucleus (STN) was targeted for bradykinetic patients with tonic dystonia, and the internal globus pallidus (GPi) was chosen for patients with hyperkinetic dystonia. Each patient underwent two periods of DBS "on" and two periods of DBS "off," each lasting 4 months. The order of stimulation conditions was randomized. The primary outcome was the change in the Canadian Occupational Performance Measure Performance (COPM-P) and Satisfaction scores after each 4-month period. Secondary outcomes were changes in the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) severity and disability scores and Unified Wilson's Disease Rating Scale (UWDRS) scores.

resultsBetween 12 May 2016 and 7 October 2022, three patients were included. Two patients received bilateral GPi DBS, and one received bilateral STN DBS. There was no change of COPM-P (p = 0.956), BFMDRS, and UWDRS scores. No serious adverse events were reported.

conclusionsSTN or GPi DBS are ineffective on dystonia related to WD.

Indexed as

Deep Brain StimulationDystoniaHepatolenticular DegenerationDouble-Blind MethodGlobus PallidusHumansMulticenter Studies as TopicProspective StudiesRandomized Controlled Trials as TopicSubthalamic NucleusTreatment Outcomedeep brain stimulationdystoniaWilson disease

Identifiers

PMID39468897
PMCPMC11622510

What OpenQuestion holds

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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.