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Deep brain stimulation for Meige syndrome: a meta-analysis with individual patient data

  • Xin Wang
  • Zhibin Zhang
  • Zhiqi Mao
  • Xinguang YuEmail author
Original Communication

Abstract

Background

Deep brain stimulation (DBS) is an effective intervention for Meige syndrome, a type of dystonia characterized by blepharospasm, facial, and oromandibular dystonia. This individual patient-level data meta-analysis was to identify the potential outcome predictors, compare the stimulation targets and summarize the efficacy of DBS for Meige syndrome.

Methods

Three electronic databases (PubMed, Web of Science and Embase) were searched with no publication data restriction to identify studies regarding DBS for Meige syndrome. The primary outcome was the improvement in BFMDRS-M score. Pearson’s correlation coefficients and a stepwise multivariate regression analysis were used to identify the potential prognostic factors.

Results

Twenty-three studies (115 patients, 94 with pallidal stimulation and 21 with subthalamic stimulation) were eligible. Patients showed significant improvement in Burke–Fahn–Marsden Dystonia Rating Scale movement (BFMDRS-M) (21.5 ± 11.0 vs 8.6 ± 6.9, P < 0.001) and disability (BFMDRS-D) (6.4 ± 5.1 vs 2.9 ± 2.4, P < 0.001) scores at the last follow-up visit (31.9 ± 30.7 months), compared with scores at baseline. Preoperative BFMDRS-M and BFMDRS-D scores were positively correlated with the relative changes in BFMDRS-M score at the last follow-up visit. On the stepwise multivariate regression, only the preoperative BFMDRS remained significant in the best predictive model.

Conclusions

Based on the existing evidence, pallidal/subthalamic stimulation is an effective therapy for even the refractory Meige syndrome. Higher preoperative scores probably indicate larger improvement. Stimulation targets or other clinical factors do not constitute the outcome predictive factors.

Keywords

Meige syndrome Deep brain stimulation Globus pallidus internus Subthalamic nucleus Predictive factors Individual patient data Meta-analysis 

Abbreviations

DBS

Deep brain stimulation

GPi

Globus pallidus internus

STN

Subthalamic nucleus

BFMDRS

Burke–Fahn–Marsden Dystonia Rating Scale

BFMDRS-D

BFMDRS disability subscale

BFMDRS-M

BFMDRS movement subscale

IPD

Individual patient data

Notes

Compliance with ethical standards

Conflicts of interest

The authors have no conflict of interest to report.

Ethical standards

All studies in this review have been approved by the local ethics committee and have therefore been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.

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© Springer-Verlag GmbH Germany, part of Springer Nature 2019

Authors and Affiliations

  1. 1.School of MedicineNankai UniversityTianjinChina
  2. 2.Department of Neurosurgery, The First Medical Center of ChinesePLA General HospitalBeijingChina

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