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

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.