Deep brain stimulation reduces pain in children with dystonia, including in dyskinetic cerebral palsy

Abstract

Aim To establish the prevalence of dystonic pain in children and their response to deep brain stimulation (DBS). Method Dystonic pain was assessed in a cohort of 140 children, 71 males and 69 females, median age 11 years 11 months (range 3y–19y 1mo), undergoing DBS in our centre over a period of 10 years. The cohort was divided into aetiological dystonia groups: 1a, inherited; 1b, heredodegenerative; 2, acquired; and 3, idiopathic. Motor responses were measured with the Burke–Fahn–Marsden Dystonia Rating Scale (BFMDRS). Results Dystonic pain was identified in 63 (45%) patients, 38% of whom had a diagnosis of cerebral palsy (CP). Dystonic pain improved in 90% of children and in all aetiological subgroups 1 year after DBS, while the BFMDRS motor score improved in 70%. Statistically significant improvement (p<0.01) was noted for the whole cohort on the Numerical Pain Rating Scale (n=27), Paediatric Pain Profile (n=17), and Caregivers Priorities and Child Health Index of Life with Disabilities questionnaire (n=48). There was reduction of pain severity, frequency, and analgesia requirement. Findings were similar for the whole cohort and aetiological subgroups other than the inherited heredodegenerative group where the improvement did not reach statistical significance. Interpretation Dystonic pain is frequent in children with dystonia, including those with CP, who undergo DBS; this can be an important, realizable goal of surgery irrespective of aetiology. We encourage the use of multimodal approach in pain research to reduce the risk of bias.

Pharmacological and neurosurgical interventions for individuals with cerebral palsy and dystonia: a systematic review update and meta‐analysis

“…Nineteen non‐randomized studies 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 , 66 , 67 evaluating DBS were identified, including six new studies (Appendix S3 ). Evidence certainty was assessed as very low for all outcomes (Appendix S4 ).…”

Application of Machine Learning Using Decision Trees for Prognosis of Deep Brain Stimulation of Globus Pallidus Internus for Children With Dystonia

“…We have evaluated cognitive abilities before and after DBS across the etiological spectrum in childhood dystonia and found overall no adverse consequences of GPi DBS in genetic (33) or acquired dystonias (including dystonic cerebral palsy) (34) or neurodegeneration with brain iron accumulation (NBIA) (35). Recent work also demonstrates the benefit of DBS in reducing pain in children with dystonia and dystonic cerebral palsy (36). Further work is currently ongoing to assess non-motor functioning pre-and post-DBS in more detail.…”

Deep Brain Stimulation and Hypoxemic Perinatal Encephalopathy: State of Art and Perspectives

“…Dysfunctional cerebellar outputs can lead to movement disorders. In addition to the motor improvement by DBS in CP, Perides S et al [21] demonstrated that DBS decrease significantly dystonic pain in children with CP. Given that pain plays an important role in quality of life of these children, it can be an important goal for DBS surgery in CP, and a main criterion for selection.…”