Abstract
Objective:
To systematically examine the research literature on the effectiveness of constraint-induced movement therapy on improving arm function in children with cerebral palsy, and to assess the association between the study effect size and the characteristics of the patients and intervention protocol.
Data sources:
A systematic literature search was conducted in PubMed, PsycINFO, Cochrane, CINAHL, Web of Science, and TRIP Database up to May 2014.
Review methods:
Studies employing randomized controlled trial design, children with cerebral palsy, comparing constraint-induced movement therapy with another intervention with a focus on arm function, and upper-extremity measures were included in this review. Methodological quality was evaluated using the Physiotherapy Evidence-based Database (PEDro) scale.
Results:
The literature search resulted in 27 randomized controlled trial studies with good methodological quality that compared constraint-induced movement therapy with other intervention therapy. Overall, constraint-induced movement therapy provided a medium beneficial effect (d = 0.546; p < 0.001) when compared with conventional therapy. For the subgroup analyses, presence of a dose-equivalent comparison group, intervention location, and time of follow-up were significant factors. Studies examining constraint-induced movement therapy effect without a dose-equivalent comparison group showed a large effect in children with cerebral palsy, but studies with a dose-equivalent group only showed a small effect. Children who received home-based constraint-induced movement therapy had a better improvement in arm function than those who received constraint-induced movement therapy elsewhere.
Conclusion:
The research literature supports constraint-induced movement therapy as an effective intervention to improve arm function in children with cerebral palsy.
Keywords
Introduction
Constraint-induced movement therapy has recently become a popular intervention for arm function training in children with hemiplegic cerebral palsy.1–3 There have been some attempts at reviewing the efficacy of constraint-induced movement therapy in improving arm function in children with hemiplegic cerebral palsy,4–6 but included very few randomized controlled trials (RCTs) (3–7 RCTs only). To the best of our knowledge, no systematic reviews using a meta-analytic method to examine the effectiveness of constraint-induced movement therapy in children with cerebral palsy have been published. In addition, 35 studies using RCT design have been published since the databases were accessed for the two most recently published systematic reviews.5,6 Thus, this review was designed to clarify the effects of constraint-induced movement therapy on arm function in children with cerebral palsy using a rigorous systematic review and meta-analytic approach. We expanded upon previous systematic reviews by: (1) adding more studies; (2) quantifying effect sizes (Cohen’s d) of constraint-induced movement therapy; and (3) using the International Classification of Functioning, Disability, and Health levels (ICF) model to classify outcome variables and then combine the outcome measures. 7 We also attempted to explain the between-study variance in effect size by examining the effects of various factors, including key characteristics of the children (e.g. age) and aspects of the intervention protocol (e.g. constraint-induced movement therapy setting, restraint type, restraint duration, constraint-induced movement therapy dosage).
Methods
Two of the authors independently conducted systematic literature searches in January 2012, January 2013, October 2013, and again in May 2014, using the electronic databases: PubMed, PsycINFO, Cochrane Central Register of Controlled Trials, CINAHL, Web of Science and Trip Database, as well as a manual search of the reference lists of each article. The keywords or mesh terms (if applicable) used for the search were: constraint-induced movement therapy, constraint induced therapy, forced use therapy, and cerebral palsy. References found in publications such as review articles were also considered for inclusion.
Studies meeting the following criteria were considered for review: (1) the study was conducted in children with cerebral palsy (aged between birth and 21 years old); (2) the study compared constraint-induced movement therapy with another therapy, such as bimanual intensive therapy, or conventional rehabilitation, which emphasized upper-extremity training; (3) the outcome measures used in the study were related to arm function; (4) the study was an RCT; and (5) the study was written in English. Studies were excluded if the study did not compare constraint-induced movement therapy with another therapy (e.g. only compared three-week constraint-induced movement therapy against six-week constraint-induced movement therapy) or the study did not provide sufficient data to compute the effect size (e.g. no standard deviations).
A data extraction form was used to code the demographic, methodological, medical, and miscellaneous variables extracted from each RCT included in the review. Demographic and medical variables included children’s age, gender, diagnosis, severity, cognitive status, and lesion side. Sample size, instruments used as outcome measures, restraint type, restraint duration, constraint-induced movement therapy dosing (session intensity, duration, frequency, and total accumulated constraint-induced movement therapy duration), comparison therapy type, dosing in comparison therapy, and time of follow-up were coded as methodological characteristics. Year and type of publication, names of authors, and country and affiliation of the authors were included in the miscellaneous section.
The quality of RCTs was evaluated using the Physiotherapy Evidence-Based Database Scale (PEDro: www.pedro.fhs.usyd.edu.au). The PEDro scale includes 11 items, in which the first item assesses the external validity and the remaining 10 items assess the internal validity, examining random allocation, concealment of allocation, baseline equivalence, blinding procedure, ‘intention to treat’ analysis, adequacy of follow-up, between-group statistical analysis, and measurement of data variability. This scale yields a total possible score of 10 points for the evaluation of internal validity, with a score of 9–10 considered ‘methodologically excellent’, 6–8 ‘good’, 4–5 ‘fair’, and less than 4 ‘poor’. 8
Data analysis
The outcome measures of each RCT were converted to a standard format by calculating the standardized mean difference (Cohen’s d), which is referred to as effect size throughout this review.9,10 If the RCT reported Cohen’s d in the results, we used that value and calculated the study variance based on effect sizes and sample sizes. Because correlations of pre- and postintervention measurements were not reported in any study, we used the postintervention mean and standard deviation data to compute effect size using the equation: (mean of constraint-induced movement therapy – comparison mean) divided by the pooled standard deviation. If more than one outcome measure was used in an RCT, the effect size for each outcome measure was computed separately and then the multiple effect sizes were averaged together to represent the overall effect size of each RCT. In addition, the outcome measures of each RCT were also classified based on the ICF model, 7 and then the multiple effect sizes of the same ICF level within a study were averaged to represent the effect size of the specific ICF level in each RCT.
Meta-analyses were run with a random-effects model that accounted for true inter-study variation in effects, as well as for random error within studies. A random-effect model was chosen over a fixed-effect model, because of the wide variation in experimental factor levels (e.g. constraint-induced movement therapy dosage, restraint duration, children’s age) used in the included studies. We also sought to determine the role of experimental factors in explaining the considerable inter-study variation observed in effect size. These experimental factors can be treated as moderator variables in a meta-analysis. Meta-regressions (using a method-of-moments model) for continuous variables or subgroup meta-analyses for categorical variables were used to examine the following potential moderator variables: (1) presence or not of a dose-equivalent comparison group; (2) children’s age; (3) restraint type; (4) restraint duration; (5) constraint-induced movement therapy daily session intensity; (6) constraint-induced movement therapy frequency per week; (7) constraint-induced movement therapy length in weeks; (8) total accumulated constraint-induced movement therapy duration; (9) location of training (e.g. laboratory or home); and (10) time of follow-up.
Meta-analyses and meta-regressions were conducted using the Comprehensive Meta-analysis software (Version 2.2; Biostat Inc., Englewood, NJ). An α value of 0.05 was used for significance in all analyses. Effect sizes were interpreted using Cohen’s convention as small (0.2), medium (0.5), or large (0.8). The effect of publication bias on the primary meta-analyses was addressed by combining a funnel plot assessment with the Duval and Tweedie’s trim and fill correction. 11 This is a preferred method for assessing the extent of publication bias, as well as for making a correction to the overall effect size.
Results
A total of 141 published studies were found from the database searches. After reviewing abstracts and titles, 40 RCTs,2,3,12–49 16 reviews,4–6,50–62 and two clinical guidelines63,64 related to constraint-induced movement therapy use in cerebral palsy were selected for full-text review. One additional RCT study was added after reading previously published reviews. 65 Thus, a total of 41 RCT studies were retrieved for full evaluation (see Figure 1).

A flow diagram of the number of studies identified, the number excluded, and the final number of studies included in this meta-analysis.
Among the 41 studies, 12 studies were excluded: four studies3,43,47,48 reported the same participants and outcome measures as in other articles;26,31,32 four studies40,41,45,46 did not include a comparison group that received traditional therapy; and four studies2,39,42,65 did not provide sufficient data to compute an effect size, which did not meet our inclusion criteria. Therefore, a total of 29 articles were included for data extraction.12–38,44,49 During data extraction, two studies were found to report their outcome measures in multiple articles;12,13,31,32 therefore, their outcome measures were merged and treated as the same studies. The final number of RCTs included in this review was 27, published between 2004 and 2014.
Description of the studies
The characteristics of these studies are summarized in Tables 1 and 2 and Supplementary Table 1, available online. There were 894 participants in the 27 studies. The average age of participants ranged from 2.4 to 10.7 years. The restraint types used in the studies included slings, glove, mitten, and cast. The restraint duration over the entire study ranged from 16 to 1008 hours, whereas the daily dose ranged from 0.5 to 6 hours. Intervention length ranged from 2 to 10 weeks, with the majority of the studies having an intervention length around two to three weeks. Intervention frequency ranged from two to seven days a week, with the majority of the studies having a frequency of about five days a week. The PEDro quality scores for the studies ranged from 5 to 9 (with only one article with a score of 5), indicating that the quality of the RCTs included in this meta-analysis was good to excellent. There were 15 studies that had a matched-dose comparison group, while 12 studies did not. Several measures of arm function were used in the studies, including the Canadian Occupational Performance Measure (COPM), the Quality of Upper Extremity Skills Test (QUEST), the Jebsen-Taylor Hand Function Test (JTHF), the Melbourne Assessment of Unilateral Upper Limb Function (Melbourne), the Bruininks-Oseretsky Test of Motor Proficiency (BOTMP), the Assisting Hand Assessment (AHA), range of motion, and reaching kinematics (e.g. movement time, trajectory smoothness).
Characteristics of the constraint-induced movement therapy and cerebral palsy studies that met the inclusion criteria of this meta-analysis.
CIMT: constraint-induced movement therapy; NDT: neurodevelopmental therapy; HABIT: Hand-Arm Bimanual Intensive Therapy; OT: Occupational Therapy.
Restraint type, restraint duration, constraint-induced movement therapy intervention protocol (i.e. duration, intensity), and intervention location for individual studies.
CIMT: constraint-induced movement therapy.
We used the ICF model to group outcome variables (see Supplementary Table 1, available online). For example, COPM was categorized at ICF’s participation level; QUEST, Melbourne, AHA were at activity level; and reaching kinematics were at body structure and function level. Among the studies used in this meta-analysis, the majority (12 studies) included outcome variables in activity level, five studies contained activity and participation levels, five contained body structure and activity levels, one contained participation level only, one contained body structure level, and three included all three levels.
Overall effect of constraint-induced movement therapy
Across all studies, there was a medium effect (d = 0.546; p < 0.001) of constraint-induced movement therapy on arm function in children with cerebral palsy. The individual study and overall effect sizes are illustrated in the forest plot in Figure 2. We found moderate heterogeneity among the studies as the value of I2 was 53% (Q = 55.74, p = 0.0006).

Forest plot of effect sizes from the 27 studies that assessed the effect of constraint-induced movement therapy on arm function in children with cerebral palsy. A square represents the effect size for a given study, with the size of the square proportional to the weighting of that study in the meta-analysis. A horizontal line indicates the 95% confidence interval for an effect. The diamond at the bottom represents the overall effect size calculated using a random-effects model.
When effect sizes were broken down based on the classification of outcomes using the ICF model, a medium effect (d = 0.641) was found at posttest for activity level, and small effects were found for participation, and body structure and function levels (d = 0.306 for participation and d = 0.486 for body structure and function). During follow-up, a medium effect (d = 0.600) was found for participation level, and small effects were found for activity, and body structure and function levels (d = 0.394 for activity and d = 0.279 for body structure and function) (see Table 3 for details).
Average effect size when breaking down outcomes based on the classification by the ICF model. The numbers in parentheses are the lower and upper limit of 95% confidence interval for the effect size.
Publication bias was assessed by examining a funnel plot of standard error vs. effect size. Minor asymmetry was noted in the plot, and thus a Duval and Tweedie’s trim and fill correction to the overall effect size was calculated. This correction shifted from 0.546 to 0.365.
Subgroup meta-analyses and meta-regression analyses
Presence or absence of a dose-equivalent comparison group, constraint-induced movement therapy location, and time of follow-up were significant factors (p < 0.05) (see Supplementary Table 2, available online). Studies with a dose-equivalent comparison group had a smaller effect size than studies without a dose-equivalent comparison group (See Figure 3). Home-based constraint-induced movement therapy and clinic-based constraint-induced movement therapy had a larger effect size than camp-based constraint-induced movement therapy. Meta-regression analysis also showed a statistically significant negative linear relationship between time of follow-up and study effect size (p < 0.05): the longer the follow-up time, the smaller the effect size.

Subgroup analysis of whether the comparison group received a dose-equivalent treatment.
Discussion
In general, when combining all outcome measures and evaluation time points of all studies, constraint-induced movement therapy had a medium effect in improving arm function in children with cerebral palsy (d = 0.546). The effect was similar to the effect of constraint-induced movement therapy when used in adults with stroke (d = 0.44).66,67 When outcome measures were examined based on the ICF model, a medium effect size was found for activity level and a small effect size for the other two levels (body structure and function, and participation) immediately after the intervention; a medium effect size for participation level and a small effect size for the other two levels (body structure and function, and activity) were found during follow-up. This suggests that improvements in activity level may be prominent in a short period of time but may disappear without continuous practice, while improvement in participation level may require extended time, and therefore showed up only during the follow-up period. This finding is similar to findings on the use of constraint-induced movement therapy in adults with stroke.66,67
Previous systematic reviews of the effectiveness of constraint-induced movement therapy on children with cerebral palsy were unable to identify associations among study effect size, children’s characteristics, and intervention protocol (e.g. Huang et al.,; 5 Sakzewski et al.,; 55 Hoare et al., 4 ). Because a greater number of RCTs was included in this meta-analysis, we found that intervention location, time of follow-up, and presence of a dose-equivalent comparison group had significant associations with study effect size.
In terms of intervention setting, home-based constraint-induced movement therapy had a larger effect size than other settings (clinic-based and camp-based). This finding was consistent with the proposed benefits of using a natural environment (home) as the intervention location.68,69 The natural environment (home) offered less distress during constraint-induced movement therapy practice for both children with cerebral palsy and their parents. Further, the training schedule can be tailored to fit into the family’s daily routine. A home-based intervention can also save the family time and money for commuting and parents can be more involved throughout the process, increasing opportunities for parent–child interaction.
The time of follow-up was negatively associated with study effect size: the longer the follow-up time, the smaller the study effect size. This finding is also consistent with the logical assumption that the constraint-induced movement therapy effect could not be maintained over time. After constraint-induced movement therapy ended, children with cerebral palsy might not practice with their affected arm as much as they did previously during constraint-induced movement therapy. From the motor learning perspective, repetitive practice advances motor skills. Research on patients with stroke suggested the patients with proper ‘transfer’ training from constraint-induced movement therapy to real-life functional tasks had a better follow-up performance than patients without ‘transfer’ training. 70 The majority of the studies reviewed here either did not include a ‘transfer’ training in their intervention or included a relatively short ‘transfer’ training to help these children apply to their daily living the new functional skill they learned using their affected hand. Consequently, the effect of this newly learned function could fade away without proper practice and reminding over time.
As expected, once we compared studies with and without a dose-equivalent comparison group, the overall effect size changed from large (d = 0.844 without a dose-equivalent comparison group) to small-to-moderate (d = 0.370 with a dose-equivalent group). The finding that constraint-induced movement therapy showed a large effect size when compared with a non-equivalent dose comparison group suggests the importance of intensive intervention regardless of the intervention type. Children in this meta-analysis received constraint-induced movement therapy intensively (a few hours per day, two to seven days per week), whereas children in the non-equivalent comparison group received conventional therapy only once per week or once every other week. Thus, the large effect was expected. In a meta-analysis done by Arpino et al., 71 intensive interventions, regardless of the treatment content, were compared with non-intensive rehabilitation treatment for children with cerebral palsy using the Gross Motor Function Measure score as the outcome measure. The authors found a large effect size with intensive intervention (d = 1.32) compared with the non-intensive therapy. Our finding is consistent with theirs.
When constraint-induced movement therapy was compared with a dose-equivalent comparison group, constraint-induced movement therapy was slightly better than the comparison group with a small-to-moderate effect size. That is, although small, constraint-induced movement therapy still showed an advantage in improving arm function in children with cerebral palsy.
We did not find any association between constraint-induced movement therapy dosage with study effect size. We believe this finding might come from a complex interaction from the study heterogeneity included in this review, especially if the intervention-related dosage in the comparison group was not equivalent with that of the constraint-induced movement therapy group. The effect of different constraint-induced movement therapy dosage remains inconclusive. Further investigations with vigorous research design are needed before a conclusion can be made.
Potential limitations of our study include: (1) inclusion of only published RCT studies; (2) publication bias; (3) exclusion of few studies because the information needed to compute effect size was unavailable; and (4) the varied outcome measures, intervention protocols, and children’s characteristics made the analysis and conclusions complicated, even though we used the ICF model as our theoretical framework.
In conclusion, our systematic review and meta-analysis found that constraint-induced movement therapy overall is an effective intervention to improve arm function in children with cerebral palsy. In contrast, when constraint-induced movement therapy is compared with a non-dose-equivalent comparison group, the overall effect of constraint-induced movement therapy appears smaller when compared with a dose-equivalent comparison group.
Clinical messages
Constraint-induced movement therapy provides a medium effect (d = 0.546) in improving arm function in children with cerebral palsy.
Studies examining constraint-induced movement therapy effect without a dose-equivalent comparison group showed a large effect in children with cerebral palsy; however, studies with a dose-equivalent comparison group only showed a small effect.
Constraint-induced movement therapy intervention settings were significantly associated with study effect size.
Footnotes
Conflict of interest
The authors declare that there is no conflict of interest.
Funding
This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
References
Supplementary Material
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