Abstract
Objective:
To systematically review the evidence on the effect of constraint-induced movement therapy compared with bimanual intensive training in children with unilateral cerebral palsy.
Data sources:
Seven electronic databases (Cinahl, Cochrane Library, EMBASE, Ovid MEDLINE, PEDro, PsycINFO, PubMed) were searched from database inception through December 2016.
Methods:
A systematic review was performed using the American Academy of Cerebral Palsy and Developmental Medicine and Preferred Reporting Items for Systematic Review and Meta-Analysis guidelines. Standardised mean differences (effect sizes) were calculated for each study and outcome.
Results:
Nine studies met the eligibility criteria. All studies provided level II evidence. Methodological quality was high in two studies, moderate in four studies and low in three studies. The methodology, participant and intervention characteristics were heterogeneous. The participants’ ages ranged from 1.5 to 16 years. Their initial hand function ranged from Manual Ability Classification System Level I to Level III. The total intervention dose ranged from 24 to 210 hours and duration from one week to ten weeks. The studies measured outcomes assessing unimanual and bimanual hand and arm function, participation and attainment of individualised goals. Overall, the effect sizes did not favour one of the interventions at short- or long-term follow-up. The 95% confidence intervals were broad, indicating inaccurate precision of the effect sizes. Pooling of the data for a meta-analysis was judged to be of little clinical value owing to heterogeneity.
Conclusion:
It is not possible to conclude whether constraint-induced movement therapy or bimanual intensive training is more effective than the other in children with unilateral cerebral palsy.
Keywords
Introduction
While most children with spastic unilateral cerebral palsy achieve independent walking and have the intellectual capacity to attend regular schools, about half of these children experience limitations in activities of daily living and restrictions in participation in education, leisure and occupational activities owing to impaired hand and arm function.1–3
This is particularly true for activities that require the use of the two hands to perform more complicated bimanual tasks.1,4 To meet the task demands, many children choose to forego challenging activities and/or adapt their behaviour at a young age.5–9 As a consequence, these children do not use the affected hand and arm to its full capacity, even when the limb is only mildly impaired. This discrepancy between capacity (the ability to execute meaningful tasks in daily life 10 ) and performance (spontaneous use of the hand and arm in daily life 10 ) is described as developmental disregard.8,9
A good base of research evidence now indicates that both constraint-induced movement therapy 11 and bimanual intensive training12,13 are more effective than no treatment or ‘very basic treatment’ in improving different aspects of hand and arm function.2,9,14,15 The interventions have also been proposed as efficacious for decreasing developmental disregard.8,9 Although the results are promising, it remains unclear whether one of these intervention protocols is more effective than the other, and if so, to what degree.2,6,16
To answer these questions, this review was conducted to systematically and critically evaluate the research evidence of studies comparing constraint-induced movement therapy to bimanual intensive training. Based on the theory of training specificity, it was hypothesised that constraint-induced movement therapy is more effective in improving unimanual hand and arm function, while bimanual intensive training has a greater impact on bimanual function.
Method
A systematic review was conducted based on the guidelines of the Preferred Reporting Items for Systematic Review and Meta-Analysis protocols (PRISMA-P) 17 and the American Academy of Cerebral Palsy and Developmental Medicine (AACPDM). 18 Two of the authors independently performed a systematic literature search in seven electronic databases, including Cinahl, Cochrane Library, EMBASE, Ovid MEDLINE, PEDro, PsycINFO and PubMed, from database inception through December 2016. The search strategy included index terms and keywords listed in Appendix S1 (available online). The reference lists of the relevant articles were reviewed for additional records. The titles and abstracts of the identified articles were screened against the eligibility criteria. The full text was retrieved when title and abstract did not provide adequate information.
Articles were included if they met the following criteria: (1) the participants were aged 17 years or younger and diagnosed with ‘spastic unilateral cerebral palsy (hemiplegia)’ or ‘congenital hemiplegia’; (2) constraint-induced movement therapy was compared with bimanual intensive training; (3) a randomized controlled group design was used; and (4) the article was published in English or Norwegian peer-reviewed scientific journals. Articles were excluded if the intervention consisted of a combination of constraint-induced movement therapy and bimanual intensive training (hybrid-models) and if the intervention was augmented with computer-based training or gaming or combined with Botulinum toxin-A injections.
When multiple articles reporting on the same trial and same measurement instruments were traced, only the article with the longest follow-up was included. When one research group gathered a cohort of participants for one main trial, but also used the same cohort in smaller substudies with focus on different outcome measures than those used in the main trial and published the results in more than one article, the articles were treated independently acknowledging that the same cohort was involved.
The level of evidence and the methodological quality were assessed based on the AACPDM criteria. 18 Studies were rated as strong when they scored ‘yes’ for six or seven items on the quality of conduct scale, moderate for four or five items, and low for three or less item. Any disagreements were resolved by consensus of the three authors of this review. Authors of the included studies were not contacted for additional information.
The two authors independently extracted data from the included studies. The measurement instruments were reviewed for psychometric properties preferring recent systematic reviews.
Adverse events were extracted and analysed. Standardised mean differences (SMDs) with 95% confidence intervals (95% CIs) were calculated for all outcomes with Hedges’ adjusted g, 19 which is adjusted for small sample sizes. The term ‘effect size’ is used to refer to SMD in this article. Review Manager software version 5.3 (RevMan; Cochrane Information Management System) was used. The effect sizes were calculated from the postintervention scores, and for the purposes of this review, an effect size of 0.2 was considered small, 0.5 moderate and 0.8 large. 20 When a measurement instrument was constructed so that a lower value indicated improved outcome, the resultant effect sizes were multiplied by −1 to adjust for effect direction. 21 Clinical and methodological heterogeneity of the studies was assessed to determine the feasibility of pooling the data in meta-analyses.
Results
The results of the literature search are presented in Figure 1. A total of 29 articles were assessed for eligibility, of which nine met the inclusion criteria. The excluded articles are listed in Appendix S2 (available online). No additional records were identified from the reference lists of the relevant articles.

Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) flow diagram.
The design, participant and intervention characteristics are summarised in Table 1. In total 225 children participated in the studies. The group size ranged from six 22 to 39 6 participants. A total of 36 children who participated in the larger Gordon et al. 16 study were also included in the smaller studies authored by Hung et al. 23 or de Brito Brandao et al. 24 The Sakzewski et al. (2015) 25 study that compared the effect of 30 and 60 hours of constraint-induced movement therapy to the same amount of bimanual intensive training used data from two separate randomized controlled trials. The data for the groups receiving 60 hours of intervention is derived from the Sakzewski et al. (2011) 26 article, which is included as an independent study in our review. Therefore, we only included the data for the groups receiving 30 hours of intervention from the Sakzewski et al. (2015) 25 article.
Study and participant characteristics, outcomes of interest and the major outcome findings. An effect size 0.0 represents no difference between the groups. Positive effect size favours constraint-induced movement therapy and negative effect size bimanual intensive training.
95% CI: 95% confidence interval; ADL: activities of daily living; AHA: Assisting Hands Assessment; BIT: bimanual intensive training; CIMT: constraint-induced movement therapy; COPM: Canadian Occupational Performance Measure; CP QOL-Child: Cerebral Palsy Quality of Life – Child; GAS: Goal Attainment Scale; GMFCS: Gross Motor Function Classification Scale; JTTHF: Jebsen-Taylor Test of Hand Function; LIFE-H: Assessment of Life Habits for Children; MACS: Manual Ability Classification Scale; MAS: Modified Ashworth Scale; MUUL: Melbourne assessment of Unimanual Upper Limb function; PEDI: Paediatric Evaluation of Disability Inventory; RCT: randomized controlled trial; N: total number; N/A: data not available/not possible to calculate SMD from the available data; OT: occupational therapist; PT: physical therapist; QUEST: Quality of Upper Extremity Skills Test; ROM; range of motion; SD: standard deviation.
The age of the participants ranged from 1.5 years22,27 to 16 years.25,26,28 Children whose hand function was classified on the Manual Ability Classification System (MACS) 29 Level I and II were included in two studies23,25 and children classified on MACS Levels I through III in five studies.16,22,24,26,28 Two studies6,27 did not report the children’s MACS level.
The intervention dose, duration and intensity varied across the studies. The total dose ranged from 24 27 to 210 6 hours. The intervention lasted between one week 25 and ten weeks. 6 Five studies16,23–25,28 employed a short-duration, high-intensity programme, two studies6,22 a less intensive, longer-duration programme and one study a less intensive, short-duration programme. 27 The use of the constraint ranged from two hours a day during therapy 22 to wearing the constraint all day.16,23,24
A total of 12 different measurement instruments were used to assess unimanual function, bimanual function, participation in daily activities, attainment of individual goals and quality of life. The instruments most often selected were the Assisting Hands Assessment (AHA), which was used in four studies,16,22,25,26 the Quality of Upper Extremity Skills Test (QUEST) dissociated movements and grasp domains were used in four studies each,6,16,22,27 the QUEST total score was used in three studies6,22,27 and the Jebsen-Taylor Test of Hand Function (JTTHF) was used in three studies.16,25,26 Descrip-tions and psychometric properties of the instruments are provided in Appendix S3 (available online). The timing of the final evaluation varied from immediately after the end of the intervention23,24,27 to one year postintervention.26,28
All nine studies provided Level II evidence. Two studies16,22 were considered to be of high quality, four6,26–28 of moderate quality and three23–25 of low quality, based on the AACPDM quality of conduct scale (Table 1; detailed quality of conduct scoring is provided in Appendix S4, available online). The effect sizes for each study at the final evaluation are presented in Table 1. The data per outcome of interest and measurement instrument at all evaluation points is presented in Appendix S5 (available online). One study 16 used the Goal Attainment Scale (GAS), but because the data generated from the GAS is at best ordinal-level data, 30 the effect sizes were not calculated for this instrument.
Of the nine studies, three were not included in quantitative analyses (Figure 1). Two articles25,28 did not provide data for the group mean and standard deviation, which are needed to calculate effect sizes. One article 23 used three-dimensional kinematics to measure hand and arm function, however, it was not possible to confirm the validity of the kinematic measures. Kinematic variables are task-specific and the validity and reliability must therefore be established for each measure. 31
Because the clinical and methodological heterogeneity across the studies was considerable, combining the data for a meta-analysis was judged to be of little clinical value.
The studies by Sakzewski et al., 26 Sakzewski et al. 28 and Gordon et al. 16 reported that there were no adverse events. The other six studies did not address adverse events (see Appendix S6, available online).
Discussion
The body of evidence in this systematic review consists of Level II evidence from nine randomized controlled trials comparing the effectiveness of constraint-induced movement therapy to bimanual intensive training in children with unilateral spastic cerebral palsy. Our findings highlight considerable clinical and methodological heterogeneity across the studies. In general, the effect sizes were small and inaccurate as indicated by the broad 95% CIs. It is therefore not possible to conclude whether one of the intervention protocols is more effective than the other in improving unimanual or bimanual hand and arm function, participation in daily activities and achievement of individualised goals at short-term or long-term time frames. Similar effect sizes were recorded regardless of whether the interventions were delivered as high-intensity, short-duration programmes or as less intensive, longer-duration programmes.
The findings do not support our hypothesis of training specificity where constraint-induced movement therapy was anticipated to be more effective in improving unimanual function and bimanual intensive training more effective in improving bimanual function. Our conclusion differs from that reported in the systematic review by Dong et al. 32 The difference in our review and the Dong et al. 32 review is likely related to the use of two different review methodologies. Whereas Dong et al. 32 conducted a narrative analysis, we evaluated the methodology of the included studies and calculated effect sizes, which allowed for detailed and systematic evaluation of the consistency of intervention effects across the studies over time. The benefit of calculating effect sizes can be seen in the analysis of the small study by Zafer et al. 27 The effect sizes in this study favoured constraint-induced movement therapy immediately postintervention (see Appendix 5: Effect sizes are presented by measurement instrument). However, large effect sizes were not found in the other studies making it difficult to generalise the findings from Zafer et al. 27 study or to make any statements about the impact of constraint-induced movement therapy.
Our conclusion on the intervention specificity agrees with what is reported in the systematic reviews by Sakzewski et al. 2 and Tinderholt Myrhaug et al. 33 However, these two reviews differed from ours because they included hybrid-models in addition to pure constraint-induced movement therapy and bimanual intensive training protocols in their meta-analysis.
Systematic evaluation of the methodological quality of the included studies revealed major factors complicating the analysis of specific intervention effects and making recommendations for clinical practice difficult. First, the study intervention protocols varied in terms of dose, intensity and duration as well as timing of follow-up. Poor reporting of treatment compliance was the most prevalent methodological shortcoming, making it unclear to what extent the children actually received the intended intervention. These factors made pooling data for a meta-analysis of little clinical value.
Second, the age of the participants and the level of hand function varied within and across the studies. Two studies6,27 did not report the children’s MACS level, which is the most commonly used classification system of hand and arm function in children with cerebral palsy. Recent research has shown that children with unilateral cerebral palsy develop their hand and arm function rapidly between the ages of 18 months and eight years, followed by a plateau up to adolescence. 34 The level of skill acquisition varies depending of the MACS level. Children at MACS Level I and Level II develop faster and reach 90% of their developmental limit earlier than children at MACS Level III. 34 Therefore, studies that include heterogeneous groups of participants with regard to age and level of hand function make it difficult to assess how and to what extent the natural pattern of hand and arm development may impact the effect of the interventions.
Third, the psychometric properties of the measurement instruments used in the studies reviewed were variable, which calls for caution when analysing the findings. Both constraint-induced movement therapy and bimanual intensive training are based on task-oriented practice with a focus on completion of activities, rather than on correction of movement patterns and prevention of compensations. 35 Instruments to measure the effect of intervention on functional skills, (e.g. using the Canadian Occupational Performance Measure or GAS), would therefore be advisable. However, the most commonly used instrument in the studies reviewed was the QUEST, which was developed based on neuro-developmental theories. 36 Therefore, the theoretical construct differs fundamentally from the functional goals of constraint-induced movement therapy and bimanual intensive training protocols. Consequently, the extent to which task-oriented practice can improve the impairments underlying many QUEST items is questionable. 37
Both constraint-induced movement therapy and bimanual intensive training aim to improve spontaneous use of the affected hand and arm, decrease developmental disregard and ultimately improve participation. Therefore, both performance-based instruments that assess the actual use of the affected hand and arm in meaningful daily activities and capacity-based instruments that assess what the child is capable of doing in a standardised environment may be better suited to assess the effectiveness of the interventions with respect to this outcome of interest.
Fourth, the studies included in this review were conducted in different therapy environments; at home, at rehabilitation centres or a combination of the two. Unfortunately, because of the heterogeneity of the participants and intervention protocols, it remains unclear which of these intervention environments may be more beneficial.
Finally, a central aspect of constraint-induced movement therapy for adults is the ‘transfer package’, which focuses on improving participation by integrating the newly improved unimanual function in meaningful bimanual activities in the person’s habitual environment. 7 It is interesting to note that only one study 16 in our review included a ‘transfer package’ (i.e. a home training programme following the intervention). However, children in the constraint-induced movement therapy group practiced only unimanual tasks and children in the bimanual intensive training group practiced only bimanual tasks, which by definition does not fulfil the aim of a transfer package as defined in similar studies of adult populations.
To address the transfer of improved unimanual skills to bimanual activities, hybrid intervention models incorporating periods of constraint-induced movement therapy and bimanual intensive training have recently been investigated. Some studies38–40 have shown that intensive training based on the hybrid model is more effective than a lower dose of ‘general’ physical therapy and/or occupational therapy. However, in a study comparing an equal dose of the hybrid model and individualised occupational therapy, the effects were not reported to be significantly different. 41 To date, only one study 42 has compared the hybrid model to an equal dose of bimanual intensive training. The study concluded that, whereas the hybrid model led to better effects on unimanual function, both interventions led to similar effects on bimanual function. As additional studies on the hybrid models are published, it will be possible to assess the efficacy of the hybrid model compared with pure constraint-induced movement therapy and/or bimanual intensive training protocols.
Limitations
Our findings are based on a literature search that only included articles published in English or Norwegian, and excluded articles written in other languages, which may have implications on our findings. The validity, reliability and responsiveness of the measurement instruments were assessed based on findings in current systematic reviews. Our data analysis was complicated by the fact that baseline scores between the groups were not always equal. Also, none of the studies reported correlations of pre- and postintervention scores. Therefore, we could only calculate the effect sizes from postintervention scores. Because of incomplete reporting of group mean and standard deviation, we had to exclude two studies from our quantitative analyses.
Implications for practice
The clinical and methodological heterogeneity of the studies reviewed and the imprecise effect sizes do not allow us to suggest specific recommendations for clinical practice regarding the benefits of constraint-induced movement therapy vs. bimanual intensive training for improving function in children with unilateral cerebral palsy. In line with the definition of evidence-based practice, 43 when making a decision regarding which intervention protocol to select, the preferences of the child and his or her family should be considered, and the expertise and clinical skills of the clinician should also be taken into account.
Although limited qualitative studies in the areas of constraint-induced movement therapy and bimanual intensive training have been conducted, there is seminal information that clinicians could integrate with their clinical decision-making. For example, qualitative research has shown that a child’s intrinsic motivation may predict occupational performance.44,45 In addition, clinicians should consider the meaning children with unilateral cerebral palsy attach to improved hand and arm function,5,46 and the value children and caregivers attach to participating in constraint-induced movement therapy programs.47–49 Finally, how therapists perceive the feasibility and usefulness of constraint-induced movement therapy and bimanual intensive training should be a consideration when implementing such interventions.50,51
Implications for research
To improve the applicability of research to clinical practice, it is essential that future research in this area be conducted with more rigorous recruitment of participants, including greater homogeneity of age and level of hand function. This would allow for more in-depth analyses of which children may respond best to the specific intervention protocols. Multicentre and international cooperation would be beneficial to hone intervention protocols, increase group sizes, improve the power of the studies to detect meaningful differences and to systematically research what effect the intervention dose, intensity and duration may have on the intervention outcomes.
It is also imperative that reporting, particularly with respect to treatment compliance, be improved. Reporting of standardised effect sizes would make the evidence from clinical studies more meaningful and applicable to clinicians. To improve the validity and clinical application of future research, the most robust measurement instruments reflecting the aims of the intervention protocols and the individual goals of the child and family should be selected.52–54 The published guidelines 55 may be a valuable tool to improve reporting.
Studying the effect of the intervention environment, particularly in home, kindergarten and school settings, may generate valuable information to support the most effective implementation of the intervention protocols. Finally, additional studies that compare hybrid models to constraint-induced movement therapy and bimanual intensive training would reveal the possible benefits of combining these two intervention protocols. The use of mixed methods research would also contribute valuable evidence to the field of clinical rehabilitation. Mixed-method studies would expand our understanding of the complex and dynamic relationships between the physical, social and emotional factors that influence the acquisition of improved hand and arm function in children with unilateral cerebral palsy.
Clinical messages
It is not possible to conclude whether constraint-induced movement therapy or bimanual intensive training is more effective than the other in improving unimanual or bimanual function, participation in daily activities or achievement of individualised goals in children with unilateral cerebral palsy at short-term or long-term time frames.
Footnotes
Acknowledgements
We would like to thank in particular Dr Gyrd Thrane, PT, PhD, Associate Professor at the University of Tromsø, The Arctic University of Norway, for supervising the Master’s thesis of MHT that was used as a pilot study for this review.We also appreciate very much the assistance from Dr. Torstein Låg, the Senior Academic Librarian at the University Library of Tromsø at the Arctic University of Norway for discussions regarding the literature search in the Master’s thesis of MHT; Dr Robbin Hickman, PT, DSc, Associate Professor at the School of Allied Health Sciences, University of Nevada, Las Vegas, for discussions regarding the AACPDM methodology for assessing the quality of conduct of the studies; and Jenna Sainsbury from the Department of Physical Therapy, College of AHS, University of Illinois, Chicago, as well as Else Mari Larsen, PT, Eli Hereide, PT, and Kristine Smidt, OT, from the Stavanger University Hospital Child Rehabilitation Centre for reviewing the written document.
Contributors
MHT initiated the study, designed it, monitored progress and decided on the analytic strategy. She contributed to the method and data analysis of the study as well as to writing of the article. She is the guarantor of the study. GLG decided on the analytic strategy and contributed to the design, method and data analysis of the study, as well as to writing of the article. GKØ decided on the analytic strategy and contributed to the design, method and data analysis of the study as well as to writing of the article.
Conflict of interest
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
References
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