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

Nieuwenhuys A, Papageorgiou E, Pataky T, De Laet T, Molenaers G, Desloovere K - PLoS ONE (2016)

Bottom Line: 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.Part 2--Post-BTX-A, both statistical approaches found increased ankle dorsiflexion throughout the gait cycle.However, different statistical approaches may lead to different interpretations of treatment outcome.

View Article: PubMed Central - PubMed

Affiliation: Department of Rehabilitation Sciences, KU Leuven, Leuven, Belgium.

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.

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Related in: MedlinePlus

Workflow literature review.This figure describes the workflow which was followed to identify the 26 papers that were included in the literature review. Based on title and abstract, 2008 papers were excluded. After assessing the full-texts, 300 additional papers were excluded using the a priori defined inclusion criteria. In the end, we identified 223 papers that reported on the outcome of treatment in children with CP by means of 3DGA evaluations. Of those 223 papers, 26 reported on the outcome of BTX-A treatment and were included for this study.
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pone.0152697.g001: Workflow literature review.This figure describes the workflow which was followed to identify the 26 papers that were included in the literature review. Based on title and abstract, 2008 papers were excluded. After assessing the full-texts, 300 additional papers were excluded using the a priori defined inclusion criteria. In the end, we identified 223 papers that reported on the outcome of treatment in children with CP by means of 3DGA evaluations. Of those 223 papers, 26 reported on the outcome of BTX-A treatment and were included for this study.

Mentions: The literature search yielded a total of 2531 titles and abstracts, which was reduced to a selection of 26 papers that evaluated BTX-A treatment using 3DGA in children with CP (Fig 1)[5], [6], [9], [18], [26–47]. Fifteen papers reported the effect of BTX-A treatment to the gastrocnemius muscle, sometimes in combination with the soleus muscle [5], [6], [26–28], [30], [31], [35], [37–39], [41–43], [46]. Besides BTX-A injections to the gastrocnemius, nine papers also included the hamstrings in a multi-level treatment [9], [18], [29], [33], [34], [36], [40], [44], [47]. Two papers focused solely on BTX-A injections to the hamstrings [32], [45]. The papers reported a median of five features (range 2–49). After features with a similar meaning were grouped together, 53 kinematic features (S1–S5 Tables) and 33 kinetic features (S6–S8 Tables) were identified. Eleven kinematic features, which were ambiguously defined, could not be included in the statistical analysis of the experimental outcome study (part 2). Figs 2–6 list all 42 features that were selected for statistical analysis in the experimental outcome study and show for each of those features the number of papers that have reported it to be responsive to BTX-A treatment in children with CP.


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

Nieuwenhuys A, Papageorgiou E, Pataky T, De Laet T, Molenaers G, Desloovere K - PLoS ONE (2016)

Workflow literature review.This figure describes the workflow which was followed to identify the 26 papers that were included in the literature review. Based on title and abstract, 2008 papers were excluded. After assessing the full-texts, 300 additional papers were excluded using the a priori defined inclusion criteria. In the end, we identified 223 papers that reported on the outcome of treatment in children with CP by means of 3DGA evaluations. Of those 223 papers, 26 reported on the outcome of BTX-A treatment and were included for this study.
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4816309&req=5

pone.0152697.g001: Workflow literature review.This figure describes the workflow which was followed to identify the 26 papers that were included in the literature review. Based on title and abstract, 2008 papers were excluded. After assessing the full-texts, 300 additional papers were excluded using the a priori defined inclusion criteria. In the end, we identified 223 papers that reported on the outcome of treatment in children with CP by means of 3DGA evaluations. Of those 223 papers, 26 reported on the outcome of BTX-A treatment and were included for this study.
Mentions: The literature search yielded a total of 2531 titles and abstracts, which was reduced to a selection of 26 papers that evaluated BTX-A treatment using 3DGA in children with CP (Fig 1)[5], [6], [9], [18], [26–47]. Fifteen papers reported the effect of BTX-A treatment to the gastrocnemius muscle, sometimes in combination with the soleus muscle [5], [6], [26–28], [30], [31], [35], [37–39], [41–43], [46]. Besides BTX-A injections to the gastrocnemius, nine papers also included the hamstrings in a multi-level treatment [9], [18], [29], [33], [34], [36], [40], [44], [47]. Two papers focused solely on BTX-A injections to the hamstrings [32], [45]. The papers reported a median of five features (range 2–49). After features with a similar meaning were grouped together, 53 kinematic features (S1–S5 Tables) and 33 kinetic features (S6–S8 Tables) were identified. Eleven kinematic features, which were ambiguously defined, could not be included in the statistical analysis of the experimental outcome study (part 2). Figs 2–6 list all 42 features that were selected for statistical analysis in the experimental outcome study and show for each of those features the number of papers that have reported it to be responsive to BTX-A treatment in children with CP.

Bottom Line: 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.Part 2--Post-BTX-A, both statistical approaches found increased ankle dorsiflexion throughout the gait cycle.However, different statistical approaches may lead to different interpretations of treatment outcome.

View Article: PubMed Central - PubMed

Affiliation: Department of Rehabilitation Sciences, KU Leuven, Leuven, Belgium.

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.

Show MeSH
Related in: MedlinePlus