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Dev Med Child Neurol. 2019 Jan;61(1):49-56. doi: 10.1111/dmcn.14063. Epub 2018 Oct 15.

Deep brain stimulation for pediatric dystonia: a meta-analysis with individual participant data.

Author information

1
Faculty of Medicine, Université de Montréal, Montreal, Canada.
2
Division of Neurosurgery, Department of Surgery, University of Toronto, Toronto, Canada.
3
College of Medicine, King Saud University, Riyadh, Saudi Arabia.
4
Division of Neurosurgery, Sunnybrook Health Sciences Centre, Toronto, Canada.
5
Division of Neurosurgery, Krembil Neuroscience Centre, Toronto Western Hospital, Toronto, Canada.
6
Child Development Program, Holland Bloorview Rehabilitation Hospital, University of Toronto, Toronto, Canada.
7
Division of Neurosurgery, The Hospital for Sick Children, Program in Neuroscience and Mental Health, The Hospital for Sick Children Research Institute, Toronto, Canada.
8
Department of Surgery, Institute of Biomaterials and Biomedical Engineering, Institute of Medical Science, University of Toronto, Toronto, Canada.

Abstract

AIM:

We performed a meta-analysis with individual participant data of deep brain stimulation (DBS) for dystonia in children and young people.

METHOD:

Three databases (PubMed, Embase, and Web of Science) were queried from January 1999 to August 2017 with no language restrictions to identify case studies and cohort studies reporting on pediatric patients (age ≤21y) with dystonia. The primary outcomes were changes in Burke-Fahn-Marsden (BFM) or Barry-Albright Dystonia Scale scores. A mixed-effects regression was used to identify associations between clinical covariates and outcomes.

RESULTS:

Of 2509 citations reviewed, 72 articles (321 children) were eligible. At last follow-up (median 12mo, 25th centile=9.0; 75th centile=32.2), 277 (86.3%) patients showed improvement in dystonia, while 66.1 percent showed clinically significant (>20%) BFM Dystonia Rating Scale-motor improvement. On multivariable hierarchical regression, older age at dystonia onset, inherited dystonia without nervous system pathology and idiopathic dystonia (vs inherited with nervous system pathology or acquired dystonia), and truncal involvement indicated a better outcome (p<0.05).

INTERPRETATION:

The data suggest that DBS is effective and should be considered in selected children with inherited or idiopathic dystonia.

WHAT THIS PAPER ADDS:

Deep brain stimulation is effective in selected children with inherited or idiopathic dystonia.

PMID:
30320439
DOI:
10.1111/dmcn.14063
[Indexed for MEDLINE]
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