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Constraint-Induced Movement Therapy in Children Aged 5 to 9 Years With Cerebral Palsy

A Day Camp Model

Thompson, Ashley M.E. MHSc; Chow, Serena MScOT; Vey, Cathy BScOT; Lloyd, Meghann PhD

doi: 10.1097/PEP.0000000000000111
RESEARCH ARTICLE
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Purpose: To examine the effectiveness of a modified form of constraint-induced movement therapy (mCIMT) in the context of a day camp model in 6 children aged 5–9 years with spastic hemiplegic cerebral palsy.

Methods: Before, 1 week after, and 3 months after 9 consecutive days of mCIMT, participants were assessed using the Quality of Upper Extremity Skills Test (QUEST) and assessments of range of motion and grip strength. Caregiver perceptions were assessed using the Pediatric Evaluation of Disability Inventory (PEDI) and a parent questionnaire.

Results: Significant improvements were observed on the “grasps” and “protective extension” subsections of the QUEST after the intervention. Increased social function was also observed as measured by the PEDI. All improvements were maintained at the 3-month follow-up assessment. Analysis of individual participants yielded additional information on clinically significant improvements as a result of the mCIMT intervention.

Conclusions: The day camp model of mCIMT was effective in inducing lasting and meaningful changes in the children with hemiplegic cerebral palsy.

The authors report that the day camp model of a modified CIMT program was effective in inducing lasting and meaningful changes in the children with hemiplegic CP.

Carleton University (Ms Thompson), Ottawa, Ontario, Canada; University of Ontario Institute of Technology (Dr Lloyd), Oshawa, Ontario, Canada; Grandview Children's Centre (Ms Chow and Ms Vey), Oshawa, Ontario, Canada.

Correspondence: Ashley M.E. Thompson, MHSc, Carleton University, 1125 Colonel By Dr, Ottawa, ON K1S 5B6, Canada (Ashley.Thompson3@carleton.ca).

Grant Support: This study was supported by grants from the Grandview Children's Centre Foundation, Oshawa, Ontario, Canada.

At the time this article was written Ashley Thompson was a student at the University of Ontario Institute of Technology, Master of Health Sciences, Oshawa, Ontario, Canada.

The authors declare no conflict of interest.

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INTRODUCTION AND PURPOSE

Cerebral palsy (CP) encompasses a group of movement and posture disorders caused by a nonprogressive but permanent abnormality in the fetal or infant brain.1 The development of the brain is interrupted by an interfering event that damages or otherwise influences the expected patterns of maturation; the result may be a lesion or a malformation in the immature brain.1–3 Although the causes of these abnormalities vary,4 CP occurs on average in 1 of 500 children,5 making it the most common pediatric physical disability.6,7 The most common form of CP is spastic hemiplegic CP, which is characterized by rigid movements as well as asymmetric motor impairment.7–9 In hemiplegia, one side of the body is more impaired than the other; the upper limb is typically more affected than the lower limb.10 The impairments experienced by children with hemiplegic CP affect many aspects of their daily lives; these limitations ultimately interfere with proper motor development in multiple ways. Independence while performing self-care tasks, ability to fully participate in play and other group settings, and overall daily function are some of the areas in which children with hemiplegic CP experience difficulties.11

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CONSTRAINT-INDUCED MOVEMENT THERAPY

Constraint-induced movement therapy (CIMT) is a form of rehabilitative therapy that involves constraining the less-affected limb, while simultaneously training the more-affected limb.12 The originally proposed recommendations were to restrain the less-affected limb for 90% of the individual's waking hours, and to perform intensive movement therapy with a trained therapist for 6 hours per day over a 2-week period.12 However, in the population of children with spastic hemiplegic CP, researchers must contend with unique challenges; the intensity of the traditional CIMT therapy may be too great to retain the attention of young children.13,14 The therapy should ideally include a variety of child-geared activities as well as intermittent breaks to maximize compliance and effectiveness.13–17 In addition, original guidelines for CIMT would require between 60 and 84 hours of physical therapy per week, rendering traditional CIMT costly, both financially and in terms of therapists' time. Thus, the original form of CIMT may not be an economically feasible and sustainable therapeutic model for children with CP. As such, researchers have been challenged to modify CIMT for children affected by spastic hemiplegic CP. Many studies have used modified forms of CIMT, using alterations in the intensity and/or duration of the therapeutic technique.15,17–20

Modified CIMT (mCIMT) has yielded improved functional outcomes for children with CP21 that are at least as good as traditional CIMT.22 In children with hemiplegic CP, mCIMT has been shown to increase the spontaneous use of the affected limb in day-to-day tasks and self-care activities (eg, dressing, feeding, and toileting),17,19 and improve motor performance measured by standardized motor assessments.21,23 In many studies, the improvements were maintained several months later.15–17,19,20,21 This suggests that a less intensive form of CIMT may produce positive effects in children and be more economical and less time-intensive for families.

In addition, many recent studies have successfully implemented CIMT in nonclinical settings.15–17,22,24 The importance of transferring the shaping and practice of CIMT to more natural settings is gaining attention in the field, as many recent studies have implemented home exercise programs into the interventions.24–27 These studies have demonstrated that CIMT implemented in a natural setting can improve the spontaneous use of the affected limb, improve coordination and precision, and improve participants' abilities to perform self-care activities. In one study the home and clinic environments were specifically compared for providing CIMT, and although participants in both groups improved, the participants in the home group improved more.24 When compared with a home setting, a day camp model of CIMT additionally offers peer interaction, which has yielded positive social outcomes for youth.14 More research is needed to support the effectiveness of mCIMT applied in natural settings for children with spastic hemiplegic CP. Furthermore, the importance of implementing CIMT in a natural setting is supported by the World Health Organization's International Classification of Functioning, Disability and Health (ICF),28 which describes the reciprocal relationship between the individual and the environment, and the way in which this interaction contributes to the individual's overall health condition and functional status.

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ICF and CIMT

According to the ICF, disability must be considered in the context of body structures and functions, personal and environmental factors, activity, and participation.28 These factors interact with one another and contribute to the overall health condition and function of an individual. The activity of the child refers to what he or she is able to do, and participation refers to what the child actually does outside the therapeutic setting.28 The goal of any intervention is to increase the activity of the child, such that participation may improve. By improving the ability to perform upper limb tasks in a lasting way, CIMT is promoting functional changes that permit children with hemiplegic CP to increase their participation in various tasks outside of therapy. Intervention in a natural setting accounts for the importance of the individual's environment and might facilitate the transfer of any learned skills into daily functioning.

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Purpose

The objective of the current study was to determine whether a 2-week day camp model of CIMT, delivered by highly trained camp counselors supported by occupational therapists (OTs), was effective in improving functional outcomes for children aged 5 to 9 years with spastic hemiplegic CP. We hypothesized that restraining the less-affected upper limb for 7 hours per day during a day camp taking place over 9 days would induce functional benefits for the more-affected limb after the intervention. We also hypothesized that the improvements observed in the more-affected limb as a result of mCIMT would persist at the 3-month follow-up assessment. Finally, we hypothesized that conducting mCIMT in a day camp setting would yield positive social outcomes for participants.

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METHODS

The current study involved a preassessment that took place 1 week before the intervention, a 9-day intervention during which participants wore a splint on the unaffected arm for 7 hours per day and participated in activities that promoted the use of the affected arm, a postassessment that took place 1 week after the intervention, and a 3-month follow-up assessment. Children with hemiplegic CP between 5 and 9 years of age were recruited through fliers that advertised the camp on bulletin boards at the Children's Treatment Centre (CTC) where the camp took place; the camp was also advertised to other CTCs in the area. Potential participants were also directly informed of the study by OTs at the CTC.

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Participants

This study was approved by the University's Research Ethics Board and the CTC's Ethics Committee. Inclusion criteria for the study required that participants have a diagnosis of spastic hemiplegic CP and be between 5 and 9 years of age. Participants had to be able to walk independently without an ambulation aid, have the cognitive and social ability to participate in a camp setting, and be willing to travel to the CTC for the duration of the camp, for the intake assessment, and for the 3 assessment sessions. Exclusion criteria included (1) any orthopedic (correctional) surgery on the affected upper limb and/or (2) dorsal rhizotomy. Regardless of any secondary conditions or any other impairment or symptoms present, all interested children who satisfied the above criteria were included in the study. A total of 6 children took part in the study (4 boys and 2 girls; mean age = 6 years, 4 months). Informed consent from a parent/guardian of each child as well as child assent was obtained for all participants during an intake session; during this session, additional information about each participant was gathered on a supplemental form, and participants were fit for a splint.

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Procedures

Participants were enrolled in a day camp (“Cast Camp”) where mCIMT was administered for 7 hours per day; Cast Camp took place over a 2-week period during the summer, for a total of 9 days (a holiday took place on the Monday of the first week). Each participant was given a Benik© splint to be worn on the unaffected hand. The model selected was the W323 with Volar Pan extension; this splint effectively immobilizes the wrist, fingers, and thumb of the restrained hand (Figure 1). This splint was chosen because of its ability to prevent the children from flexing and extending the wrist, as well as to prevent finger extension and all thumb movements. The splint was worn only during camp hours and was applied and removed by camp staff each day.

Fig. 1

Fig. 1

All children who attended the camp were participants in the study (no children other than the 6 in the study attended camp for these 2 weeks); all children wore the splint and received mCIMT. The camp was led by 3 counselors, all of whom were university students (entering either the third or fourth year of undergraduate studies) and were required to undergo a 2-week intensive orientation and training session at the CTC; this training prepared the counselors to act as camp counselors for children with various physical and developmental disabilities. In addition, 2 of the 3 camp counselors, including the camp coordinator, were involved in Cast Camp in 2011; the camp coordinator was further involved in Cast Camp in 2010. The counselors for Cast Camp additionally underwent two 1-hour training sessions with OTs at the CTC to prepare to effectively deliver CIMT in a day camp setting. Occupational therapists at the CTC dropped in to the camp daily to ensure that CIMT was being effectively delivered by the camp counselors. The daily visits were arranged using sign-up sheets at the CTC; various OTs working at the CTC volunteered to drop in to the camp each day around 10 AM. The length of the visits varied depending on the needs of the camp (ie, questions or problems that arose), with visits lasting from 10 minutes to 1 hour. The OTs spent this time observing the staff, providing direction regarding how to give the children the correct amount of support, answering questions, and working with the children directly. In addition, 1 of the OTs directly involved with this project dropped in to the camp more informally each afternoon to observe and answer any questions. This OT was also available on-call at all times during the camp's duration in case any problems requiring the assistance of an OT arose. All activities performed during the camp were planned by camp counselors, supported by OTs, and specifically selected to promote the use of the affected hand. Gross manipulation tasks as well as precision tasks were emphasized in the daily activities; examples of the activities included finger painting, crafts, and team-building activities. Counselors also monitored all feeding times and provided a motivating environment in which children were encouraged to use their affected hands as much as possible. The activities changed from day to day but always included both free play in an outdoor setting (that included a park) and structured play (eg, sports, games, and crafts) that were all performed by the children using their affected hand (the splinted hand was allowed to be used as an assisting hand when tasks were more challenging or clearly bimanual in nature ie, stabilizing a lunch bag with the splinted hand to unzip the bag using the affected hand). Camp counselors used verbal cues to discourage the use of the splinted hand and to encourage the use of the affected hand during both structured and unstructured play. The splint restricted motion of the unaffected hand such that the unaffected arm could only be used for stability and gross movements; still, consistent reminders and encouragement to use the affected hand in a dominant manner were provided by the camp counselors throughout each day of the camp. In the current study, mCIMT was administered in a day camp setting for 7 hours per day for 9 consecutive weekdays, for a total of 63 hours of CIMT; as such, these conditions are fairly close to the originally proposed guidelines of 6 hours per day for 14 days, but represent a more cost-effective model of CIMT.

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Outcome Measures

Several assessments were used in this study, all selected to evaluate the function of the affected hand or to determine the parents' perceptions of the children's function. An OT, who was not the primary OT for any of the participants, conducted all assessments. Previous studies have used a wide variety of assessments to measure improvement after CIMT; however, a common aspect of most studies is the selection of assessments that directly measure motor function as well as those that examine qualitative improvements in daily life (eg, increased use of the limb, self-care, independence, and social ability).17,19–21,27,2930 The assessments used in the current study were chosen based on the variety of areas that they collectively assess (eg, gross motor, fine motor, social ability, and mobility), as well as for their use in previous studies using CIMT for children with CP.15,16,23,25,29,30

The primary assessment used in this study to evaluate upper limb function was the Quality of Upper Extremity Skills Test (QUEST).34 The QUEST is a standardized assessment of motor performance used to evaluate a child's ability to complete a variety of tasks; all items are graded as a yes/no, with a “yes” meaning that each child can perform the task completely.34 The QUEST is scored on a basis of 100 points, and includes items assessing upper limb movements, grasping and manipulating objects, balance, and protective extension skills.34

The assessments used to evaluate caregiver perceptions of each child's function included the Pediatric Evaluation of Disability Inventory (PEDI)35 and a questionnaire developed by the OTs at the CTC based on the Canadian Occupational Performance Measure.36,37 The PEDI is a questionnaire that is completed by the child's caregiver; this measure is also scored on a basis of 100 points. The PEDI is used to measure the ability of the child to participate in self-care tasks (eg, dressing and feeding), mobility capabilities (eg, indoor and outdoor movements), and social function abilities (eg, communication and social play).35 The CTC questionnaire is used to assess any changes observed in the frequency and/or spontaneity of the use of the affected limb; and furthermore to assess the child's ability to perform a variety of self-care tasks, and includes both the parents' evaluations of the importance of the child improving in each area and their levels of satisfaction with the child's current level of performance for each task. Each item is assessed on a scale of 1 to 10, with scores closer to 10 indicating a higher level of performance, importance, or satisfaction.

Additional tests included measures of range of motion, grip strength, and assessment of spasticity using the Modified Ashworth Scale,38 and the level of impairment using the Gross Motor Function Classification System (GMFCS)39 and the Manual Ability Classification System (MACS).40

Range of motion measures included elbow and wrist flexion/extension, finger and thumb flexion/extension, and shoulder rotation. Grip strength in both hands was measured using a modified sphygmomanometer (using a Baseline® sphygmomanometer); the device consists of a hand-held pump containing water connected to a pressure gauge that measures grip strength in pounds per square inch (psi) when squeezed (the maximum possible score is 15.5 psi). Participants were seated, with the shoulder in the neutral position and the elbow flexed at a 90° angle when grip strength measurements were taken. Three successive measurements were taken in the unaffected limb and then the affected limb; a rest period of about 15 seconds was given between trials, which was the time required to read and record the score.

The OT completed the Modified Ashworth Scale on the elbow joint of the affected limb during each of the 3 assessment sessions. The GMFCS level was established by the OT conducting the assessments, and the MACS level was established by asking the caregivers of each participant to select, from a list of 5 descriptions, the level that best described the child's overall function.

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Data Analysis

The Friedman test was used for the QUEST data as well for grip strength and the caregiver reports on the CTC questionnaire. Nonparametric post-hoc analyses were done using the Wilcoxon signed rank test, and Bonferroni corrections were used for multiple comparisons. The Wilcoxon signed rank test was further used to analyze results from the PEDI and the independent reports given from camp counselors and caregivers on the “performance” section of the CTC questionnaire (these assessments were only administered on 2 occasions). Observed power and effect sizes were calculated for all statistical tests, and are reported with the “results” section. All data handling and analyses were performed using SPSS version 19.0.41

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RESULTS

Characteristics of each participant can be found in Table 1. Considerable variability was present in the sample; therefore, each child was used as his or her own control and analyzed individually for many of the measures employed.

TABLE 1

TABLE 1

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Motor Outcomes

Table 2 shows the QUEST scores for all participants. Significant differences were observed on the “protective extension” subsection of the QUEST over time (χ2(2) = 8.316; P = .016; observed power = 0.833; partial η2 = 0.589) (Figure 2). Post-hoc analysis with Wilcoxon signed rank tests was conducted using a Bonferroni correction, resulting in a significance level set at α = 0.017. A trend toward improvement on the “protective extension” subsection observed immediately after the intervention (Z = −2.214; P = .027) was not statistically significant.

Fig. 2

Fig. 2

TABLE 2

TABLE 2

As a group, the participants experienced significant improvements in the “grasps” subsection of the QUEST (χ2(2) = 7.684; P = .021; observed power = 0.91; partial η2 = 0.712) (Figure 3). No statistically significant improvements were observed in post-hoc analyses using Wilcoxon signed rank tests. No significant changes were observed in the “dissociated movements” or “weight bearing” subsections of the QUEST, or in the overall QUEST scores.

Fig. 3

Fig. 3

No significant improvements in grip strength or range of motion of the affected or unaffected limb were observed after CIMT.

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Caregiver Reports

On the PEDI, caregivers' responses indicated a significant improvement in social function skills after the intervention (Z = −2.201; P = .028; observed power > 0.999; Cohen's d = −2.55). No significant changes were observed in any of the other categories of the PEDI.

On the CTC questionnaire, no significant changes were observed in the caregiver responses over time or on the “satisfaction” section of the questionnaire over time. In each individual category of the CTC questionnaire, however, at least 3 of the 6 caregivers' satisfaction responses increased between the initial assessment and the 3-month follow-up; the only exception to this was in the “frequency of use” subsection, where only 2 of the 6 caregivers reported increased satisfaction. In the “spontaneity of use” subsection, 5 of the 6 caregivers reported increased satisfaction after the intervention.

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DISCUSSION

The results from the current study support findings of prior clinical studies demonstrating mCIMT to be effective in children with hemiplegia.20,22,23 The results further support previous research demonstrating that mCIMT can be successfully applied outside a clinical setting, yielding positive outcomes for youth.15,17,24–26

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Motor Outcomes

The finding of improvement in the grasps area of the QUEST supports previous research findings of statistically significant improvements in grasping ability that persist months later.29,30 More efficient grasping capabilities would allow children to perform more efficiently a variety of functional tasks, including self-care tasks such as feeding and dressing, and might allow increased participation in other activities such as play.

Trends toward improvement in the “protective extension” sub-section of the QUEST occurred regardless of GMFCS or MACS level. As activities performed at the camp did not specifically address protective extension skills an increase in the spontaneous use of the affected limb could have reasonably contributed to improved “protective extension” scores. Children with CP often experience impairments in balance42; these impairments can lead to falls, further supporting the relevance of the current finding of improved protective extension skills.

The improvements observed on the QUEST are consistent with previous research, demonstrating improvements on standardized tests of motor performance16,17,19–21,23,30,43; however, those studies found varying degrees of improvement on various motor assessments.16–18,27,29 Ultimately, individual differences may affect the improvements observed on standardized tests of motor performance.

Like our findings, previous studies have consistently shown that grip strength and active range of motion in the unaffected limb are not hindered after CIMT.15,21,23,31 Previous studies have also reported nonsignificant results for change in grip strength in the affected limb after a CIMT intervention.33,44,45

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Caregiver Reports

Caregivers reported through the PEDI a significant increase in social function after the intervention, supporting our hypothesis related to positive social outcomes; previous studies have also reported increased social function after the CIMT intervention.28,46 Gilmore et al14 examined participants' opinions of a day-camp model of CIMT, and found that they enjoyed being in a place where others were like them; participants felt motivated as a result of this. Increased feelings of self-confidence, self-esteem, and motivation may have resulted from the camp setting, leading to improved social function. This represents a developmental gain that can positively influence many other areas of a child's life, including home, school, and community environments. Three of the 6 caregivers reported improved independence for their children in self-care tasks after the intervention. These results are consistent with previous research demonstrating caregiver perceptions of children's improved ability to use the affected limb after CIMT.16,18,20,22,32

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Strengths/Limitations

Despite demonstrating a variety of benefits of mCIMT, this study also identifies some of the methodological issues of clinical studies employing CIMT in the pediatric population affected by CP. Inconsistent results have been reported in prior studies of mCIMT in children32,47; some authors suggested that personal factors such as age, level of impairment, and level of motivation ought to be considered when applying the CIMT intervention in this population.17,30,44,48,49 The current study included children aged 5 to 9 years, which to the knowledge of the researchers, is among the smallest age ranges reported for this type of study. Although the narrow age range led to a small sample size, we believe a smaller age range will ultimately yield more generalizable results for this age group. Another potential limitation of the current study was that parents enrolled their children into the study, and parents who seek out these types of positive rehabilitative opportunities for their children may create a biased sample.

ICF. The ultimate goal of CIMT is to improve the child's capacity to perform daily activities using the affected limb. In this study, these skills were intentionally practiced in a natural setting to facilitate the transfer of these skills to daily life. Although the children were undergoing a form of therapy, they were also engaged in fun activities while at the camp, and had the opportunity to interact with other children who were facing similar challenges. The ICF28 considers these environmental and personal factors important when providing treatment programs for children with CP. Although all children did not experience the same benefits from the CIMT intervention, not all children experienced difficulties in the same areas. Although the benefits varied within our sample, all children experienced some improvement in functional outcome. If children are better able to perform even one activity, they may participate in that activity more frequently.

Statistical Versus Clinical Significance: Individual Differences. Although several of the primary motor assessments yielded important clinical outcomes as a result of the intervention, many of the measures did not reach statistical significance. For example, when asked about their satisfaction with their child's overall function on the CTC questionnaire, at least 50% of the parents reported increased satisfaction at the 3-month follow-up on 6 of the 7 measures. In addition, 83.33% of the parents reported increased satisfaction with respect to their child's spontaneity of the use of the affected limb. This indicates that important clinical changes are being observed, as caregivers are reporting increased satisfaction in a variety of functional areas. Because the ultimate objective of the current study was to promote functional benefits for this population as a result of mCIMT, these parental reports are important and should not be ignored. Several explanations are possible for the lack of statistically significant results; in the current study, the limited sample size and the heterogeneity within the sample are the most likely explanations for the lack of statistically significant results on certain measures. Within the sample, participants experienced different levels of mobility, functioning of the hemiplegic limb, and comorbidities. Our results show the importance of considering the personal factors of each child, outlined by the ICF,28 that have the potential to influence the outcome of the therapy. The results further support the need to consider clinical changes in this population as opposed to exclusively examining statistically significant changes in group means. This is particularly important when examining interventions for children with spastic hemiplegic CP, as a large amount of variability exists within this population.1,3,4

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Suggestions for Future Research

In the future a control group should be included, and investigators should attempt to recruit larger numbers of children within a narrow age range. Studies employing CIMT in the future should consider the importance of applying such techniques in a natural environment, and should also consider conducting a cost analysis to determine the economic efficiency of the day camp model of mCIMT compared with mCIMT conducted in a clinical setting.

Questionnaires about daily functioning might be best in a semistructured interview as caregivers may interpret questions differently. For example, 5 parents in this study reported improved spontaneous use of the affected limb after 3 months, but only 2 reported an increase in frequency of use. This may or may not represent an incongruent set of responses.

Finally, several investigators have examined the combined effects of botulinum toxin-A and upper limb therapy for children with spastic hemiplegic CP.51,52 In the future investigators should consider examining the potential benefits of employing CIMT in conjunction with botulinum toxin A.53,54

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CONCLUSIONS

The day camp model of mCIMT used in the current study provides an effective setting for children with spastic hemiplegic CP. The current study demonstrated the effectiveness of a day camp model of CIMT in inducing lasting functional benefits for children with spastic hemiplegic CP. This intervention, considered within the context of the ICF model, improved the activity and participation levels of participants.

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    Keywords:

    cerebral palsy/hemiplegia; child; evaluation of disability; motor skills; upper limb

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