Deep brain stimulation for Gilles de la Tourette syndrome in children and youth: a meta-analysis with individual participant data

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OBJECTIVE

Gilles de la Tourette syndrome (GTS) is a disorder characterized by motor and vocal tics. Although by definition the onset of GTS is before age 18 years, clinical trials of deep brain stimulation (DBS) have been conducted only in adults. Using individual participant data (IPD) meta-analysis methodology, the current study investigated the safety and efficacy of DBS as a treatment for GTS in children and youth.

METHODS

A systematic review with no date or language restrictions was performed according to the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) statement. Three electronic databases were searched: PubMed, EMBASE, and Web of Science. From 843 articles screened, the IPD of 58 children and youth (ages 12–21 years) extracted from 21 articles were collected and analyzed. A mixed-effects univariable analysis followed by multivariable hierarchical regression was performed using change in the Yale Global Tic Severity Scale (YGTSS) score as the primary outcome and reported measures of comorbidities as secondary outcomes.

RESULTS

The authors’ results showed an average improvement of 57.5% ± 24.6% across studies on the YGTSS. They also found that comorbid depression and stimulation pulse width each correlated negatively with outcome (p < 0.05). In patients with less severe GTS, greater improvements were evident following thalamic stimulation. More than one-quarter (n = 16, 27.6%) of participants experienced side effects, the majority of which were minor.

CONCLUSIONS

DBS in the pediatric population may be an effective option with a moderate safety profile for treatment of GTS in carefully selected children and youth. Large, prospective studies with long-term follow-up are necessary to understand how DBS influences tic symptoms and may alter the natural course of GTS in children.

ABBREVIATIONS ADHD = attention-deficit/hyperactivity disorder; DBS = deep brain stimulation; GPi = globus pallidus pars interna; GTS = Gilles de la Tourette syndrome; GTS-QOL = GTS Quality of Life; HDRS = Hamilton Depression Rating Scale; IPD = individual participant data; NA = nucleus accumbens; OCD = obsessive-compulsive disorder; SIB = self-injurious behavior; STAI = State-Trait Anxiety Inventory; Y-BOCS = Yale-Brown Obsessive Compulsive Scale; YGTSS = Yale Global Tic Severity Scale.

Article Information

Correspondence George M. Ibrahim: The Hospital for Sick Children, Toronto, ON, Canada. george.ibrahim@sickkids.ca.

INCLUDE WHEN CITING Published online October 26, 2018; DOI: 10.3171/2018.7.PEDS18300.

Disclosures Dr. Kalia: speaker’s fees and honorarium from Medtronic. Dr. Lozano: consultant for Medtronic, St. Jude Medical, and Boston Scientific.

© AANS, except where prohibited by US copyright law.

Headings

Figures

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    Search results and included articles as per the PRISMA guidelines.

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    Greater improvements in YGTSS score after thalamic DBS compared with GPi DBS in children and youth with less severe (excluding the most severe quartile, based on preoperative YGTSS score) GTS on univariate analysis. Bars indicate the mean and error bars the SEM.

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