Literature Review and Comparison of Two Statistical Methods to Evaluate the Effect of Botulinum Toxin Treatment on Gait in Children with Cerebral Palsy

PLoS One. 2016 Mar 31;11(3):e0152697. doi: 10.1371/journal.pone.0152697. eCollection 2016.

Abstract

Aim: This study aimed at comparing two statistical approaches to analyze the effect of Botulinum Toxin A (BTX-A) treatment on gait in children with a diagnosis of spastic cerebral palsy (CP), based on three-dimensional gait analysis (3DGA) data. Through a literature review, the available expert knowledge on gait changes after BTX-A treatment in children with CP is summarized.

Methods: Part 1--Intervention studies on BTX-A treatment in children with CP between 4-18 years that used 3DGA data as an outcome measure and were written in English, were identified through a broad systematic literature search. Reported kinematic and kinetic gait features were extracted from the identified studies. Part 2--A retrospective sample of 53 children with CP (6.1 ± 2.3years, GMFCS I-III) received 3DGA before and after multilevel BTX-A injections. The effect of BTX-A on gait was interpreted by comparing the results of paired samples t-tests on the kinematic gait features that were identified from literature to the results of statistical parametric mapping analysis on the kinematic waveforms of the lower limb joints.

Results: Part 1-53 kinematic and 33 kinetic features were described in literature. Overall, there is no consensus on which features should be evaluated after BTX-A treatment as 49 features were reported only once or twice. Part 2--Post-BTX-A, both statistical approaches found increased ankle dorsiflexion throughout the gait cycle. Statistical parametric mapping analyses additionally found increased knee extension during terminal stance. In turn, feature analyses found increased outtoeing during stance after BTX-A.

Conclusion: This study confirms that BTX-A injections are a valuable treatment option to improve gait function in children with CP. However, different statistical approaches may lead to different interpretations of treatment outcome. We suggest that a clear, definite hypothesis should be stated a priori and a commensurate statistical approach should accompany this hypothesis.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Biomechanical Phenomena
  • Botulinum Toxins, Type A / therapeutic use*
  • Cerebral Palsy / drug therapy*
  • Cerebral Palsy / physiopathology*
  • Child
  • Child, Preschool
  • Female
  • Gait / drug effects*
  • Humans
  • Infant
  • Male

Substances

  • Botulinum Toxins, Type A

Associated data

  • figshare/10.6084/M9.FIGSHARE.2055729.V1

Grants and funding

AN is supported by an 'Onderzoekstoelage' (OT) of KU Leuven university (OT/12/100). EP is supported by the MD Paedigree project, a Model-Driven Paediatric European Digital Repository, partially funded by the European Commission under FP7 - ICT Programme (grant agreement no: 600932, http://www.md-paedigree.eu). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.