Mapping efficacious deep brain stimulation for pediatric dystonia

Ailish Coblentz, Gavin J.B. Elias, Alexandre Boutet, Jurgen Germann, Musleh Algarni, Lais M. Oliveira, Clemens Neudorfer, Elysa Widjaja, George M. Ibrahim, Suneil K. Kalia, Mehr Jain, Andres M. Lozano, Alfonso Fasano*

*Corresponding author for this work

Research output: Contribution to journalArticlepeer-review

7 Scopus citations


Objective: The objective of this study was to report the authors' experience with deep brain stimulation (DBS) of the internal globus pallidus (GPi) as a treatment for pediatric dystonia, and to elucidate substrates underlying clinical outcome using state-of-the-art neuroimaging techniques. Methods: A retrospective analysis was conducted in 11 pediatric patients (6 girls and 5 boys, mean age 12 ± 4 years) with medically refractory dystonia who underwent GPi-DBS implantation between June 2009 and September 2017. Using pre- and postoperative MRI, volumes of tissue activated were modeled and weighted by clinical outcome to identify brain regions associated with clinical outcome. Functional and structural networks associated with clinical benefits were also determined using large-scale normative data sets. Results: A total of 21 implanted leads were analyzed in 11 patients. The average follow-up duration was 19 ± 20 months (median 5 months). Using a 7-point clinical rating scale, 10 patients showed response to treatment, as defined by scores < 3. The mean improvement in the Burke-Fahn-Marsden Dystonia Rating Scale motor score was 40% ± 23%. The probabilistic map of efficacy showed that the voxel cluster most associated with clinical improvement was located at the posterior aspect of the GPi, comparatively posterior and superior to the coordinates of the classic GPi target. Strong functional and structural connectivity was evident between the probabilistic map and areas such as the precentral and postcentral gyri, parietooccipital cortex, and brainstem. Conclusions: This study reported on a series of pediatric patients with dystonia in whom GPi-DBS resulted in variable clinical benefit and described a clinically favorable stimulation site for this cohort, as well as its structural and functional connectivity. This information could be valuable for improving surgical planning, simplifying programming, and further informing disease pathophysiology.

Original languageEnglish (US)
Pages (from-to)346-356
Number of pages11
JournalJournal of Neurosurgery: Pediatrics
Issue number3
StatePublished - Mar 2021


  • Connectivity
  • Deep brain stimulation
  • Dystonia
  • Functional neurosurgery
  • Probabilistic mapping

ASJC Scopus subject areas

  • Surgery
  • Pediatrics, Perinatology, and Child Health
  • Clinical Neurology


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