BMJ neurology open · 2026
Deep brain stimulation for the treatment of paediatric dystonia due to neuroanatomical abnormalities: a systematic review of efficacy and future perspectives.
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Soliman Yasmin, Figliomeni Abbey, Lee Hyunchul, Thorburn Megan, Ghosh Soumya, Ramakonar Hari, Yau Wai Yan
Аннотация
BACKGROUND: Paediatric dystonia due to neuroanatomical brain abnormalities causes significant morbidity in affected children. Medical treatments have limited efficacy and deep brain stimulation (DBS) surgery is a potential therapy that may be underused.
OBJECTIVES: This review aims to map available literature regarding the efficacy of DBS in children with dystonia due to neuroanatomical abnormalities to support clinicians' decision in patient selection.
METHODS: A systematic review was conducted following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Studies were selected from public databases using predefined inclusion criteria, assessing DBS outcomes on Burke Fahn Marsden Dystonia Rating Scale motor, quality of life, DBS targets, underlying aetiologies and adverse events.
RESULTS: Of the 58 articles reviewed, 279 patients were included. All studies were individual case reports or small retrospective cohorts with variable indications, surgical targets, outcome measures and follow-up duration. We summarised the mean symptom improvements in subgroups of dyskinetic cerebral palsy, neurodegeneration with brain iron accumulation, glutaric aciduria type 1 and Lesch-Nyhan syndrome. We proposed a workflow for patient selection and discussed future directions in this field.
CONCLUSIONS: DBS provides mild to moderate motor benefits and may improve quality of life in carefully selected patients with dystonia due to neuroanatomical abnormalities. However, a higher rate of surgical complications may negate the benefits in progressive disorders. Knowledge gaps remain in optimal surgical target selection, the role of adaptive DBS and post-surgical outcomes in children with rarer heredodegenerative disorders treated with DBS.
OBJECTIVES: This review aims to map available literature regarding the efficacy of DBS in children with dystonia due to neuroanatomical abnormalities to support clinicians' decision in patient selection.
METHODS: A systematic review was conducted following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Studies were selected from public databases using predefined inclusion criteria, assessing DBS outcomes on Burke Fahn Marsden Dystonia Rating Scale motor, quality of life, DBS targets, underlying aetiologies and adverse events.
RESULTS: Of the 58 articles reviewed, 279 patients were included. All studies were individual case reports or small retrospective cohorts with variable indications, surgical targets, outcome measures and follow-up duration. We summarised the mean symptom improvements in subgroups of dyskinetic cerebral palsy, neurodegeneration with brain iron accumulation, glutaric aciduria type 1 and Lesch-Nyhan syndrome. We proposed a workflow for patient selection and discussed future directions in this field.
CONCLUSIONS: DBS provides mild to moderate motor benefits and may improve quality of life in carefully selected patients with dystonia due to neuroanatomical abnormalities. However, a higher rate of surgical complications may negate the benefits in progressive disorders. Knowledge gaps remain in optimal surgical target selection, the role of adaptive DBS and post-surgical outcomes in children with rarer heredodegenerative disorders treated with DBS.