Darcy Fehlings1, Leah Brown1, Adrienne Harvey2, Kate Himmelmann3, Jean-Pierre Lin4, Alexander Macintosh1, Jonathan W Mink5, Elegast Monbaliu6, James Rice7, Jessica Silver1, Lauren Switzer1, Ilana Walters1. 1. Department of Paediatrics, Bloorview Research Institute, Holland Bloorview Kids Rehabilitation Hospital, University of Toronto, Toronto, ON, Canada. 2. Developmental Disability and Rehabilitation Research, Murdoch Childrens Research Institute, Parkville, Vic, Australia. 3. Department of Pediatrics, Clinical Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden. 4. Complex Motor Disorders Service, Evelina London Children's Hospital, Guy's and St Thomas', NHS Foundation Trust, Kings' Health Partners, London, UK. 5. Department of Neurology, University of Rochester, Rochester, NY, USA. 6. Department of Rehabilitation Sciences, KU Leuven, Leuven, Belgium. 7. Paediatric Rehabilitation Department, Women's and Children's Hospital, North Adelaide, South Australia, Australia.
Abstract
AIM: To systematically review evidence for pharmacological/neurosurgical interventions for managing dystonia in individuals with cerebral palsy (CP) to inform a care pathway. METHOD: Searches included studies with a minimum of five participants with dystonia in CP receiving oral baclofen, benzodiazepines (clonazepam, diazepam, lorazepam), clonidine, gabapentin, levodopa, trihexyphenidyl, botulinum toxin, intrathecal baclofen (ITB), or deep brain stimulation (DBS). Evidence was classified according to American Academy of Neurology guidelines. RESULTS: Twenty-eight articles underwent data extraction: one levodopa, five trihexyphenidyl, three botulinum toxin, six ITB, and 13 DBS studies. No articles for oral baclofen, benzodiazepines, clonidine, or gabapentin met the inclusion criteria. Evidence for reducing dystonia was level C (possibly effective) for ITB and DBS; level C (possibly ineffective) for trihexyphenidyl; and level U (inadequate data) for botulinum toxin. INTERPRETATION: For dystonia reduction, ITB and DBS are possibly effective, whereas trihexyphenidyl was possibly ineffective. There is insufficient evidence to support oral medications or botulinum toxin to reduce dystonia. There is insufficient evidence for pharmacological and neurosurgical interventions to improve motor function, decrease pain, and ease caregiving. The majority of the pharmacological and neurosurgical management of dystonia in CP is based on clinical expert opinion. WHAT THIS PAPER ADDS: Intrathecal baclofen and deep brain stimulation are possibly effective in reducing dystonia. Current evidence does not support effectiveness of oral medications or botulinum toxin to reduce dystonia. Evidence is inadequate for pharmacological/neurosurgical interventions impact on improving motor function, pain/comfort, and easing caregiving. The majority of the care pathway rests on expert opinion.
AIM: To systematically review evidence for pharmacological/neurosurgical interventions for managing dystonia in individuals with cerebral palsy (CP) to inform a care pathway. METHOD: Searches included studies with a minimum of five participants with dystonia in CP receiving oral baclofen, benzodiazepines (clonazepam, diazepam, lorazepam), clonidine, gabapentin, levodopa, trihexyphenidyl, botulinum toxin, intrathecal baclofen (ITB), or deep brain stimulation (DBS). Evidence was classified according to American Academy of Neurology guidelines. RESULTS: Twenty-eight articles underwent data extraction: one levodopa, five trihexyphenidyl, three botulinum toxin, six ITB, and 13 DBS studies. No articles for oral baclofen, benzodiazepines, clonidine, or gabapentin met the inclusion criteria. Evidence for reducing dystonia was level C (possibly effective) for ITB and DBS; level C (possibly ineffective) for trihexyphenidyl; and level U (inadequate data) for botulinum toxin. INTERPRETATION: For dystonia reduction, ITB and DBS are possibly effective, whereas trihexyphenidyl was possibly ineffective. There is insufficient evidence to support oral medications or botulinum toxin to reduce dystonia. There is insufficient evidence for pharmacological and neurosurgical interventions to improve motor function, decrease pain, and ease caregiving. The majority of the pharmacological and neurosurgical management of dystonia in CP is based on clinical expert opinion. WHAT THIS PAPER ADDS: Intrathecal baclofen and deep brain stimulation are possibly effective in reducing dystonia. Current evidence does not support effectiveness of oral medications or botulinum toxin to reduce dystonia. Evidence is inadequate for pharmacological/neurosurgical interventions impact on improving motor function, pain/comfort, and easing caregiving. The majority of the care pathway rests on expert opinion.
Authors: Andrew T Hale; Meredith A Monsour; John D Rolston; Robert P Naftel; Dario J Englot Journal: Neurosurg Rev Date: 2018-11-05 Impact factor: 3.042
Authors: Jacquelin Peck; Ivan Urits; Hisham Kassem; Christopher Lee; Wilton Robinson; Elyse M Cornett; Amnon A Berger; Jared Herman; Jai Won Jung; Alan D Kaye; Omar Viswanath Journal: Psychopharmacol Bull Date: 2020-10-15
Authors: Laura A Bonouvrié; Jules G Becher; Johan S H Vles; R Jeroen Vermeulen; Annemieke I Buizer Journal: Ann Neurol Date: 2019-05-21 Impact factor: 10.422