Natasha Yuan-Kim Liow1, Hortensia Gimeno2, Daniel Edward Lumsden1, Jennifer Marianczak3, Margaret Kaminska1, Stephen Tomlin3, Jean-Pierre Sao-Ming Lin4. 1. Complex Motor Disorders Service, Children's Neurosciences Centre, Evelina London Children's Hospital, Guy's and St Thomas' NHS Foundation Trust, London, UK. 2. Complex Motor Disorders Service, Children's Neurosciences Centre, Evelina London Children's Hospital, Guy's and St Thomas' NHS Foundation Trust, London, UK; Department of Psychology, Institute of Psychiatry, Psychology and Neurology, King's College London, UK. 3. Paediatric Pharmacy Evelina London Children's Hospital, Guy's and St Thomas NHS Foundation Trust, London, UK. 4. Complex Motor Disorders Service, Children's Neurosciences Centre, Evelina London Children's Hospital, Guy's and St Thomas' NHS Foundation Trust, London, UK. Electronic address: Jean-Pierre.Lin@gstt.nhs.uk.
Abstract
INTRODUCTION: Gabapentin has been used in the management of neuropathic pain, epilepsy and occasionally movement disorders. METHODS: A four-year retrospective, observational study analysed the use of gabapentin for severe dystonia in children at the Evelina London Children's Hospital. Motor severity was classified according to the Gross Motor Function Classification System (GMFCS), Dystonia Severity Assessment Plan (DSAP) and levels of impairment in activities of daily living were graded according to the WHO International Classification of Functioning, Disability and Health, Children & Youth version (ICF-CY) before and after gabapentin. RESULTS: The majority of the 69 children reported were level 5 GMFCS (non-ambulant). The DSAP grade fell significantly from grade 3 before to grade 1 after gabapentin. Significant improvements in median ICF-CY grades were seen following gabapentin in sleep quality, sleep amount, mood & agreeableness, pain, general muscle tone, involuntary muscle contractions and seating tolerance (p < 0.01 in all areas). A significantly higher mean dose of 18.1 mg/kg/dose (SD: 13.3) for dystonia, compared to 7.61 mg/kg/dose (SD: 4.14) for pain relief without dystonia (z = -2.54, p = 0.011) was noted. DISCUSSION & CONCLUSION: Gabapentin may significantly ameliorate dystonia severity and improve activities of daily living and quality of life in children with severe dystonia.
INTRODUCTION: Gabapentin has been used in the management of neuropathic pain, epilepsy and occasionally movement disorders. METHODS: A four-year retrospective, observational study analysed the use of gabapentin for severe dystonia in children at the Evelina London Children's Hospital. Motor severity was classified according to the Gross Motor Function Classification System (GMFCS), Dystonia Severity Assessment Plan (DSAP) and levels of impairment in activities of daily living were graded according to the WHO International Classification of Functioning, Disability and Health, Children & Youth version (ICF-CY) before and after gabapentin. RESULTS: The majority of the 69 children reported were level 5 GMFCS (non-ambulant). The DSAP grade fell significantly from grade 3 before to grade 1 after gabapentin. Significant improvements in median ICF-CY grades were seen following gabapentin in sleep quality, sleep amount, mood & agreeableness, pain, general muscle tone, involuntary muscle contractions and seating tolerance (p < 0.01 in all areas). A significantly higher mean dose of 18.1 mg/kg/dose (SD: 13.3) for dystonia, compared to 7.61 mg/kg/dose (SD: 4.14) for pain relief without dystonia (z = -2.54, p = 0.011) was noted. DISCUSSION & CONCLUSION: Gabapentin may significantly ameliorate dystonia severity and improve activities of daily living and quality of life in children with severe dystonia.
Authors: Nikolas Boy; Chris Mühlhausen; Esther M Maier; Jana Heringer; Birgit Assmann; Peter Burgard; Marjorie Dixon; Sandra Fleissner; Cheryl R Greenberg; Inga Harting; Georg F Hoffmann; Daniela Karall; David M Koeller; Michael B Krawinkel; Jürgen G Okun; Thomas Opladen; Roland Posset; Katja Sahm; Johannes Zschocke; Stefan Kölker Journal: J Inherit Metab Dis Date: 2016-11-16 Impact factor: 4.982
Authors: V M McClelland; A Valentin; H G Rey; D E Lumsden; M C Elze; R Selway; G Alarcon; J-P Lin Journal: J Neurol Neurosurg Psychiatry Date: 2016-02-04 Impact factor: 10.154