Evidence map›Paper›PMID 30397842›Full record

SynthesisNeurosurgical review2020

Deep brain stimulation in pediatric dystonia: a systematic review.

Andrew T Hale, Meredith A Monsour, John D Rolston, Robert P Naftel, Dario J Englot

Abstract readSystematic Review
In one paragraph

Synthesis in Neurosurgical review, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 37 papers.

0numbers the graph read from it
0cells of the map it votes in
37citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

37 citing papers in PubMed.

  1. Article
  2. Long-Term Outcomes of Deep Brain Stimulation in Woodhouse-Sakati Syndrome.Movement disorders : official journal of the Movement Disorder Society · 2026
    Article
  3. Article
  4. Review
  5. Movement Disorders in Children: Treatment Algorithm and Surgical Options.Advances and technical standards in neurosurgery · 2026
    Review
  6. Review
  7. Article
  8. Simultaneous, dual-target, bilateral deep brain stimulation for treatment of galactosemia-induced dystonia and tremor in a pediatric patient.Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery · 2025
    Article
  9. Review
  10. Article
  11. Review
  12. Article
  13. Review
  14. Article
  15. Deep brain stimulation in pediatric dystonia: calls for therapeutic realism over nihilism.Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery · 2024
    Article
  16. Review
  17. Article
  18. The effect of GPi-DBS assessed by gait analysis in DYT11 dystonia: a case study.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2024
    Article
  19. Article
  20. Neurosurgical management of non-spastic movement disorders.Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery · 2023
    Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

5 authors.

Andrew T HaleDepartment of Neurological Surgery, Vanderbilt University Medical Center, Nashville, TN, USA. Andrew.hale@vanderbilt.edu.ORCID http://orcid.org/0000-0003-3182-2966
Meredith A MonsourDepartment of Neurological Surgery, Vanderbilt University Medical Center, Nashville, TN, USA.
John D RolstonDepartment of Neurosurgery, University of Utah, Salt Lake City, UT, USA.
Robert P NaftelDepartment of Neurological Surgery, Vanderbilt University Medical Center, Nashville, TN, USA.
Dario J EnglotDepartment of Neurological Surgery, Vanderbilt University Medical Center, Nashville, TN, USA.

Funding

MEDICAL SCIENTIST TRAINING PROGRAMT32GM007347 · NIGMS · VANDERBILT UNIVERSITY · PI WILLIAMS, CHRISTOPHER S. · 1985 to 2023
$26.3M
Medical Scientist Training ProgramT32GM152284 · NIGMS · VANDERBILT UNIVERSITY · PI Christopher S. Williams · 2024 to 2026
$4.8M
MULTIMODAL MAPPING OF SUBCORTICAL AND CORTICAL FUNCTIONAL NETWORK DISTURBANCES IN FOCAL EPILEPSYR00NS097618 · NINDS · VANDERBILT UNIVERSITY MEDICAL CENTER · PI ENGLOT, DARIO J · 2017 to 2019
$743k
Alterations in the sulfur assimilation pathway mediates iron metabolismF30HL143826 · NHLBI · VANDERBILT UNIVERSITY · PI HALE, ANDREW T · 2018 to 2020
$99k
NIGMS NIH HHS T32 GM007347NIGMS NIH HHS T32 GM152284NIH HHS F30HL143826, T32GM007347NIH HHS R00 NS097618NINDS NIH HHS R00 NS097618
6 · The paper itself

Abstract

While deep brain stimulation (DBS) treatment is relatively rare in children, it may have a role in dystonia to reduce motor symptoms and disability. Pediatric DBS studies are sparse and limited by small sample size, and thus, outcomes are poorly understood. Thus, we performed a systematic review of the literature including studies of DBS for pediatric (age < 21) dystonia. Patient demographics, disease causes and characteristics, motor scores, and disability scores were recorded at baseline and at last post-operative follow-up. We identified 19 studies reporting DBS outcomes in 76 children with dystonia. Age at surgery was 13.8 ± 3.9 (mean ± SD) years, and 58% of individuals were male. Post-operative follow-up duration was 2.8 ± 2.8 years. Sixty-eight percent of patients had primary dystonia (PD), of whom 56% had a pathological mutation in DYT1 (DYT1+). Across all patients, regardless of dystonia type, 43.8 ± 36% improvement was seen in Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) motor (-M) scores after DBS, while 43.7 ± 31% improvement was observed in BFMDRS disability (-D) scores. Patients with PD were more likely to experience ≥ 50% improvement (56%) in BFMDRS-M scores compared to patients with secondary causes of dystonia (21%, p = 0.004). DYT1+ patients were more likely to achieve ≥ 50% improvement (65%) in BFMDRS-D than DTY1- individuals (29%, p = 0.02), although there was no difference in BFMDRS-M ≥ 50% improvement rates between DYT1+ (66%) or DYT1- (43%) children (p = 0.11). While DBS is less common in pediatric patients, individuals with severe dystonia may receive worthwhile benefit with neuromodulation treatment.

Indexed as

AdolescentChildChild, PreschoolDeep Brain StimulationDystoniaHumansNeurosurgeryNeurosurgical ProceduresPediatricsTreatment OutcomeDeep brain stimulationDystoniaFunctional neurosurgeryPediatric neurosurgeryPediatrics

Identifiers

PMID30397842
PMCPMC6500764

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.