Impact of intensive upper limb rehabilitation on quality of life: a randomized trial in children with unilateral cerebral palsy

Authors

  • LEANNE SAKZEWSKI,

    1.  Queensland Cerebral Palsy and Rehabilitation Research Centre, The University of Queensland, Brisbane, QLD.
    2.  Queensland Children’s Medical Research Institute, Brisbane, QLD.
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  • STACEY CARLON,

    1.  Department of Physiotherapy, La Trobe University, Melbourne, Vic.
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  • NORA SHIELDS,

    1.  Queensland Children’s Medical Research Institute, Brisbane, QLD.
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  • JENNY ZIVIANI,

    1.  Queensland Children’s Medical Research Institute, Brisbane, QLD.
    2.  School of Health and Rehabilitation Sciences, The University of Queensland, Brisbane, QLD.
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  • ROBERT S WARE,

    1.  Queensland Children’s Medical Research Institute, Brisbane, QLD.
    2.  School of Population Health, The University of Queensland, Brisbane, QLD, Australia.
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  • ROSLYN N BOYD

    1.  Queensland Cerebral Palsy and Rehabilitation Research Centre, The University of Queensland, Brisbane, QLD.
    2.  Queensland Children’s Medical Research Institute, Brisbane, QLD.
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  • This article is commented on by Majnemer on pages 388389 of this issue.

Dr Leanne Sakzewski at Queensland Cerebral Palsy and Rehabilitation Research Centre, Department of Paediatrics and Child Health, Level 7 Block 6, Royal Brisbane and Women’s Hospital, Herston Road, Herston, QLD 4029, Australia. E-mail: leanne.sakzewski@uqconnect.edu.au

Abstract

Aim  The aim of this study was to determine whether constraint-induced movement therapy is more effective than bimanual training in improving the quality of life of children with unilateral cerebral palsy (CP).

Method  Sixty-three children (mean age 10y 2mo [SD 2y 6mo]; 33 males, 30 females) with CP of the spastic motor type (n=59) or with spasticity and dystonia (n=4) were randomly allocated to two groups. The children were assessed as Manual Ability Classification System level I (n=16), II (n=46), or III (n=1). Each group received 6 hours of daily intervention (either constraint-induced movement therapy [CIMT] or bimanual training [BIM]) for 10 days over a 2-week period (total intervention time 60h). Children aged 9 years and older completed the Cerebral Palsy Quality of Life Questionnaire for Children (CPQOL-Child) and those aged 8 years and older completed the KIDSCREEN-52. All parents completed proxy versions of each measure. Assessments were made at baseline and at 3, 26, and 52 weeks after the end of the intervention.

Results  Thirty-five children completed the CPQOL-Child and 41 completed the KIDSCREEN-52. No changes in social or emotional well-being were reported by children in either group. Children and parents from both groups reported a significant improvement in their or their child’s feelings about functioning as well as participation and physical health on the CPQOL-Child. The parents of children receiving CIMT reported positive and sustained changes in their child’s social well-being (CPQOL-Child). The CIMT group showed significant improvements in physical well-being, psychological well-being, and moods and emotions (KIDSCREEN-52) at 3 weeks post intervention, which were maintained over the study period.

Interpretation  Intensive goal-directed upper limb training programmes using either CIMT or BIM achieved domain-specific changes in quality of life relating to feelings about functioning and participation and physical health. A condition-specific quality of life compared with a generic measure may be better able to detect changes in quality of life in children with unilateral CP.

Abbreviations
BIM

Bimanual training

CIMT

Constraint-induced movement therapy

CPQOL-Child

Cerebral Palsy Quality of Life Questionnaire for Children

QOL

Quality of life

What this paper adds

  •  Intensive goal-directed upper limb training using either a unimanual or bimanual approach leads to changes in specific domains of quality of life.
  •  A condition-specific QOL outcome measure such as the CPQOL-Child may better capture long-term changes in QOL domains following intervention compared with a generic QOL measure.
  •  Evaluation of QOL following intensive upper limb training programmes provides valuable information on the translation of functional improvements to the broader context of children’s sense of well-being.

Cerebral palsy (CP) is the leading cause of childhood physical disability, with an incidence of 1 in 500 live births.1 Around 35% (1 in 1300) of these children have congenital hemiplegia and usually present with more pronounced impairment in the upper limbs than in the lower limbs.2 Quality of life (QOL) is now considered an important outcome of interventions, as it relates to a person’s individual perception or feelings of well-being across a number of domains (e.g. physical, social, emotional, spiritual).3

Children with CP generally report lower QOL than their typically developing peers.4 There is no clear relationship between the level of functioning and overall QOL. Functioning tends to be more highly correlated with physical domains of QOL and weakly associated with psychological domains.5 Among children with unilateral CP, functional difficulties often manifest as problems with day-to-day activities, to which impaired upper limb skills contribute. It remains unclear whether interventions that aim to improve upper limb function, and more broadly functional independence, will impact on specific domains of QOL. Only a few upper limb rehabilitation studies have measured QOL; little evidence of changes following intervention has been found.6–8

The INCITE study9 was a single blind matched-pairs randomized clinical trial comparing the efficacy of constraint-induced movement therapy (CIMT) with bimanual training (BIM) of equal intensity and reported outcomes across all domains of the International Classification of Functioning, Disability and Health.10 There were no differences between groups immediately after the intervention of 60 hours over 2 weeks.11,12 By 26 weeks post intervention, the results for each group reflected the mode of training. The CIMT group showed significantly improved unimanual capacity (i.e. how the impaired hand/arm is used when asked) in terms of both quality and efficiency of movement. The bimanual performance of the BIM group improved, but there was no change in the quality of upper limb movement. The two groups achieved equal improvement in occupational performance and in personal care.11,12 We then evaluated subsequent changes in QOL. Given that differences in other outcomes were relatively few, we hypothesized that the two training approaches would result in similar changes in QOL; however, we hypothesised that both interventions would lead to domain-specific changes, in particular in domains that relate to physical functioning. Changes in the physical functioning domains of QOL were anticipated as both interventions aim to improve upper limb function and specifically address functional goal areas identified by children and their caregivers.

Method

Participants

The methods used in the INCITE trial have been widely reported.9,11,12 The study was approved by the Ethics in Human Research Committees at The Royal Children’s Hospital, Melbourne; La Trobe University, Melbourne; The Royal Children’s Hospital and Health Services District, Brisbane; and The University of Queensland, Brisbane. Written informed consent was obtained from parents and children aged 12 years or older and verbal assent was given by younger participants.

Children were included if they had unilateral CP (determined by medical and rehabilitation physicians) and were aged 5 to 16 years, could follow instructions, and had a Modified Ashworth Scale score of not more than three for the distal upper limb. Children were ineligible if they had predominant dystonia or contractures or had previously undergone upper limb surgery or serial casting, or had received intramuscular botulinum toxin A injections to the upper limbs in the previous 6 months. Children were recruited through public and private therapy, medical specialists in Victoria and Queensland, and the population register in Queensland, Australia.

Design and procedure

Following screening assessments, children were matched in pairs according to age (within 12mo), sex, side of hemiplegia, and upper limb function. Once matched, children were randomized in pairs using a computer-generated list of random numbers and concealed envelopes opened by non-study personnel.

The authors of the Cerebral Palsy Quality of Life Assessment for Children (CPQOL-Child) suggest that an effect size of 0.67SD is large enough to be of significance to the child and family.13 A sample size of 36 children in each group (total sample size of 72) was calculated to have at least 80% power to compare changes in QOL domains from baseline to 26 weeks follow-up between the two groups (two-sided test at 0.05 significance).

Interventions

The interventions used either a unimanual (CIMT) or bimanual intensive training (BIM) approach and have been described in detail elsewhere.9 Each group received the same dosage and overall content of intervention, provided in the same environment, which was delivered in groups of 9 to 13 children for 6 hours daily for 2 weeks (10d) using an intensive day camp model (total dosage 60h). Six day camps were run at community sporting facilities in Melbourne and Brisbane, Australia, using a novel circus theme to enhance children’s motivation for engagement and participation. Interventions used a goal-directed activity-based framework, employing principles of motor learning. All activities were analysed and selected to target specific movement components required for goal achievement and were novel, fun, and motivating, and fostered self-generated voluntary repetition of desired movements.9 For the BIM training group, we adopted the strategy used by Gordon et al.,14 which provided explicit instructions on how each hand should be used before each activity. For both CIMT and BIM training, the focus was on completion of activities in their entirety. A core team of four experienced occupational therapists and physiotherapists conducted the camps and coordinated and trained the therapist volunteers, therapy students, circus trainers, and community youth workers, with a ratio of one therapist per two children. Participants in the CIMT group wore a tailor-made glove on their unimpaired hand while attending the day camp, which was removed for no more than 15 minutes each day. The glove was constructed from breathable fabric with a volar plastic insert to prevent grasp.

Outcome measures

The QOL measures were secondary outcomes for the INCITE trial. For this paper, the primary outcome for QOL, the CPQOL-Child,15 is a condition-specific measure that evaluates the well-being of children with CP across seven broad domains (social well-being and acceptance, functioning, participation and physical health, emotional well-being, access to services, pain and impact of disability, family health). Two versions were used: the CPQOL-Child self-report for children aged 9 years and above, and the proxy version, which was completed by all parents. The CPQOL-Teen version for young people aged 14 to 18 years was not available at the start of the study, so the child version was used for the entire sample. The CPQOL-Child has a high internal consistency (0.74–0.92) and 2-week test–retest reliability (0.76–0.89).16 Items were recoded and transformed into scaled scores (range 0–100) for each domain in accordance with the test manual.15

The secondary outcome, the KIDSCREEN-52,17 is a generic measure of health-related QOL that evaluates 10 domains (physical well-being, psychological well-being, moods and emotions, self-perception, autonomy, parent relations and home life, social support and peers, school environment, social acceptance [bullying], financial resources). The child self-report version was used with children aged 8 years and above, and the proxy version was completed by all parents. Internal consistency is good to excellent (0.77–0.89) and it has adequate test–retest reliability (0.55–0.77). In accordance with the test manual, the item scores for each domain were summed and transformed into T-values (mean 50, SD 10).17

Both measures were posted to families before each assessment and were checked for completion at each visit; however, there was no occurrence of incomplete questionnaires. Children with significant intellectual impairment did not complete the self-report versions of the CPQOL-Child and KIDSCREEN-52.

Statistical analysis

Baseline data from each outcome measure for each treatment group are reported using descriptive statistics. Continuous data were compared between groups by fitting a regression model using generalized estimating equations to baseline, 3-, 26-, and 52-week measurements with an interaction term between the intervention group and a three-level factor indicating time of measurement. The generalized estimating equation models assumed a Gaussian family, identity link, and unstructured working correlation matrix for repeat measurements on participants.18 We used conventional variance estimates. Analyses were conducted according to the intention-to-treat principle. There was no adjustment for multiple comparisons.19 Analyses were conducted using Stata 10 (StataCorp, College Station, TX, USA, 2007).

Results

Sixty-four children (32 matched pairs) were randomly assigned to treatment, one did not proceed and had no baseline data, and one withdrew after the first day. Sixty-two children received their allocated intervention (Fig. 1). There were no major adverse events. All children in the CIMT group tolerated wearing the glove on their unimpaired hand. Forty children aged 9 years or older were eligible to complete the CPQOL. In total, 35 children completed the CPQOL-Child self-report (CIMT, n=18; BIM, n=17), and five were unable to because of intellectual impairment. Forty-eight children were aged 8 years or older. Of these, 41 completed the KIDSCREEN-52 (CIMT n=22; BIM n=19), and the remaining seven children were unable to complete the assessment owing to intellectual impairment. Proxy reports of both QOL measures were completed by 61 mothers (96%), one father (2%), and one grandmother (2%).

Figure 1.

 Trial profile. Estimated mean difference (EMD) derived from generalized estimating equations. CIMT, constraint-induced movement therapy; BIM, bimanual therapy.

Baseline comparisons

After matching and concealed random allocation within pairs, there were no significant differences between groups on demographic or baseline measures except for the CPQOL-Child self-reported domain of participation (Table I). The mean age was 10 years 2 months (range 5–16y) and children were predominantly classified as level II on both the Manual Abilities Classification System and Gross Motor Function Classification System.

Table I.   Participant demographics and baseline characteristics of constraint-induced movement therapy (CIMT) and bimanual training (BIM) groups
CharacteristicCIMT (n=32)BIM (n=31)
  1. *North American usage: mental retardation. aThere were no significant differences between groups on any baseline demographic or quality-of-life measures except for the CPQOL-Child self-report domain of participation (p=0.05). MACS, Manual Ability Classification System; GMFCS, Gross Motor Function Classification System; CPQOL-Child Cerebral Palsy Quality of Life Questionnaire for Children.

Demographic
 Age, mean (SD)10 years 1 month (2y 5mo)10 years 2 months (2y 5mo)
 Sex, Males/Females, n17/1516/15
 Side of hemiplegia, left, n (%)16 (50)11 (36)
 Epilepsy, n (%)5 (16)8 (26)
 Learning disability*, n (%)10 (31)9 (29)
Classification
 MACS, n (%)
  Level I8 (25)8 (26)
  Level II23 (72)23 (74)
  Level III1 (3)0
 GMFCS, n (%)
  Level I8 (25)8 (26)
  Level II24 (75)23 (74)
School
 Mainstream school, n (%)29 (91)29 (94)
 Special school, n (%)3 (10)2 (7)
CPQOL-Child self-report (n)1817
 Social well-being, mean (SD)80.9 (12.3)80.5 (9.5)
 Feelings about functioning, mean (SD)77.4 (13.7)74.6 (13.9)
 Participation, mean (SD)78.1 (18.6)69.1 (15.7)a
 Emotional well-being, mean (SD)85.4 (11.0)82.7 (13.9)
 Pain, mean (SD)26.2 (23.4)35.9 (18.2)
CPQOL-Child parent proxy report
 Social well-being, mean (SD)78.9 (12.6)79.4 (12.9)
 Feelings about functioning, mean (SD)70.6 (12.1)67.5 (13.1)
 Participation, mean (SD)69.8 (15.2)65.4 (18.1)
 Emotional well-being, mean (SD)79.0 (11.7)76.9 (14.4)
 Pain, mean (SD)24.7 (14.7)28.4 (17.0)
 Access to services, mean (SD)65.7 (15.8)62.5 (20.2)
 Family health, mean (SD)70.3 (15.9)66.4 (16.9)
KIDSCREEN-52 self-report (n)2219
 Physical well-being, mean (SD)46.5 (6.4)45.8 (6.8)
 Psychological well-being, mean (SD)48.5 (7.7)49.7 (7.0)
 Moods and emotions, mean (SD)50.0 (8.6)48.3 (8.0)
 Self-perception, mean (SD)51.1 (11.8)51.0 (11.9)
 Autonomy, mean (SD)49.5 (11.0)48.5 (10.1)
 Parent relations and home life, mean (SD)51.8 (11.8)50.8 (6.9)
 Financial resources, mean (SD)49.6 (11.5)44.9 (9.7)
 Social support and peers, mean (SD)53.5 (12.2)50.1 (8.2)
 School environment, mean (SD)54.3 (11.5)54.8 (8.4)
 Social acceptance, mean (SD)48.0 (10.3)46.9 (10.2)
KIDSCREEN-52 parent proxy report
 Physical well-being, mean (SD)42.0 (8.7)43.2 (7.7)
 Psychological well-being, mean (SD)45.5 (10)48.7 (9.2)
 Moods and emotions, mean (SD)47.5 (7.8)47.3 (9.5)
 Self-perception, mean (SD)47.1 (8.7)47.8 (8.9)
 Autonomy, mean (SD)47.2 (9.0)46.1 (8.7)
 Parent relations and home life, mean (SD)48.0 (10)48.7 (9.2)
 Financial resources, mean (SD)51.0 (7.7)50.2 (11.6)
 Social support and peers, mean (SD)50.0 (10)45.5 (10.2)
 School environment, mean (SD)50.8 (9.9)51.54 (11.0)
 Social acceptance, mean (SD)45.7 (11.1)42.1 (11.8)

Primary outcomes

CPQOL-Child self-report

There were significant differences between groups regarding feelings about functioning and emotional well-being favouring CIMT; however, these were not maintained beyond 3 weeks. There were no significant differences between the two training groups in the other QOL domains at each follow-up period (Table II). Children from neither group reported changes in social well-being. Both groups demonstrated significant changes in feelings about functioning immediately after intervention that were maintained at 52 weeks post intervention. Significant changes in perception of participation and physical health were reported by both groups immediately after intervention and were maintained at 52 weeks. Children in the BIM group also reported immediate improvements in feelings about pain and impact of disability, but these were not maintained beyond 3 weeks. Children in the CIMT group reported immediate changes in emotional well-being that were not maintained in the longer term.

Table II.   Estimated mean difference (EMD) over time in quality of life domains between groups receiving constraint-induced movement therapy (CIMT) or bimanual training (BIM) according to CPQOL-Child, parent proxy and self-reports
 EMD, parent proxy reportEMD, child self-report
Baseline to 3 weeks (95% CI); pBaseline to 26 weeks (95% CI); pBaseline to 52 weeks (95% CI); pBaseline to 3 weeks (95% CI); pBaseline to 26 weeks (95% CI); pBaseline to 52 weeks (95% CI); p
  1. aChild self-report, n=18; parent proxy report, n=32. bChild self-report, n=17; parent proxy report, n=31. cDomains not assessed in child self-report. EMD generated from generalized estimating equations. CPQOL-Child, Cerebral Palsy Quality of Life Questionnaire for Children; CI, confidence interval.

Social well-being
 CIMTa6.1 (3.9 to 8.2); <0.0015.7 (1.9 to 9.5); 0.0035.6 (1.6 to 9.6); 0.0064.6 (−0.4 to 9.6); 0.074.3 (−2.3 to 10.9); 0.25.4 (−1.3 to 12.2); 0.1
 BIMb2.4 (0.3 to 4.5); 0.023.4 (−0.5 to 7.2); 0.092.3 (−1.8 to 6.4); 0.3−0.2 (−5.4 to 5.1); 0.9−1.8 (−8.7 to 5.2); 0.63.4 (−3.7 to 10.5); 0.4
 Difference between groups3.6 (0.6 to 6.6); 0.022.3 (−3.1 to 7.8); 0.43.3 (−2.5 to 9.0); 0.34.8 (−2.5 to 12.0); 0.26.1 (−3.5 to 15.6); 0.22.0 (−7.8 to 11.8); 0.7
Feelings about functioning
 CIMT4.9 (1.7 to 8.0); 0.0026.4 (2.7 to 11.1); <0.0013.5 (−0.1 to 7.1); 0.068.4 (5.5 to 11.3); <0.0019.3 (3.7 to 15.0); 0.0019.4 (3.0 to 15.9); 0.004
 BIM7.8 (4.7 to 10.8); <0.0017.4 (3.7 to 11.1); <0.0018.1 (4.4 to 11.7); <0.0013.8 (0.8 to 6.8); 0.012.4 (−3.6 to 8.4); 0.48.6 (1.8 to 15.4); 0.01
 Difference between groups−2.9 (−7.3 to 1.4); 0.2−0.9 (−6.2 to 4.3); 0.7−4.6 (−9.7 to 0.5); 0.084.6 (0.4 to 8.7); 0.036.9 (−1.4 to 15.1); 0.10.8 (−8.5 to 10.2); 0.9
Participation and physical health
 CIMT5.2 (3.3 to 7.1); <0.0016.2 (1.7 to 10.8); 0.0085.1 (0.6 to 9.6); 0.034.8 (1.3 to 8.4); 0.00710.0 (3.5 to 16.5); 0.0039.1 (1.5 to 16.7); 0.02
 BIM7.8 (5.9 to 9.7); <0.0019.6 (5.0 to 14.3); <0.0019.2 (4.5 to 13.9); <0.0013.7 (0.1 to 7.3); 0.056.1 (−0.8 to 13.0); 0.0811.8 (3.7 to 19.8); 0.004
 Difference between groups−2.6 (−5.3 to 0.1); 0.06−3.4 (−9.9 to 3.1); 0.3−4.1 (−10.6 to 2.4); 0.21.2 (−3.9 to 6.2); 0.73.9 (−5.6 to 13.3); 0.4−2.6 (−13.7 to 8.4); 0.6
Emotional well-being
 CIMT3.4 (0.5 to 6.3); 0.023.4 (−1.0 to 7.8); 0.12.3 (−1.8 to 6.3); 0.33.8 (0.2 to 7.4); 0.043.7 (−2.6 to 10.0); 0.21.8 (−4.7 to 8.2); 0.6
 BIM5.4 (2.6 to 8.2); <0.0014.2 (−0.3 to 8.6); 0.074.0 (−0.2 to 8.2); 0.06−1.8 (−5.6 to 1.9); 0.31.1 (−5.6 to 7.7); 0.85.7 (−1.1 to 12.5); 0.1
 Difference between groups−2.0 (−6.0 to 2.0); 0.3−0.8 (−7.1 to 5.5); 0.8−1.7 (−7.6 to 4.1); 0.65.7 (0.5 to 10.9); 0.032.6 (−6.5 to 11.7); 0.6−3.9 (−13.2 to 5.5); 0.4
Pain
 CIMT2.8 (−1.7 to 7.3); 0.2−2.4 (−8.8 to 4.1); 0.5−0.6 (−6.7 to 5.6); 0.9−4.7 (−12.4 to 3.0); 0.2−6.9 (−17.1 to 3.2); 0.2−7.8 (−18.3 to 2.8); 0.1
 BIM−2.2 (−6.7 to 2.3); 0.3−2.3 (−8.8 to 4.2); 0.5−4.0 (−10.3 to 2.4); 0.2−10.6 (−18.5 to −2.6); 0.009−6.4 (−17.2 to 4.4); 0.2−12.1 (−23.0 to −0.9); 0.03
 Difference between groups5.0 (−1.4 to 11.3); 0.1−0.1 (−9.2 to 9.1); 0.93.4 (−5.4 to 12.2); 0.85.9 (−5.2 to 16.9); 0.3−0.5 (−15.3 to 14.3); 0.94.3 (−11.0 to 19.7); 0.6
Accessc
 CIMT2.4 (−1.0 to 10.9); 0.14.9 (−1.0 to 11.0); 0.12.7 (−3.2 to 8.7); 0.4
 BIM3.2 (−2.2 to 8.6); 0.52.3 (−3.7 to 8.3); 0.53.7 (−2.3 to 9.7); 0.2
 Difference between groups−0.7 (−8.4 to 7.0); 0.92.7 (−5.8 to 11.2); 0.5−0.9 (−9.3 to 7.5); 0.8
Familyc
 CIMT3.6 (0.5 to 6.7); 0.023.3 (−1.3 to 7.9); 0.22.5 (−2.6 to 7.7); 0.2
 BIM4.4 (1.4 to 7.5); 0.0048.6 (3.9 to 13.3); <0.00110.5 (5.3 to 15.7); <0.001
 Difference between groups−0.8 (−5.1 to 3.5); 0.7−5.4 (−11.9 to 1.2); 0.1−7.9 (−15.3 to 0.6); <0.001

CPQOL-Child parent proxy report

There were no significant differences between groups on any domain except for family health at 52 weeks, favouring BIM, and social well-being at 3 weeks, favouring CIMT (Table II). Neither group reported changes in pain and impact of disability or access to services. Parents of children from both groups reported an immediate change in perceived social well-being, which was maintained to 52 weeks post intervention for the CIMT group. Parents from both groups reported changes in feelings about functioning, which were maintained to 52 weeks by the BIM group only, and in participation and physical health, which was maintained by both groups. Both groups demonstrated initial changes in emotional well-being which were not maintained at 26 or 52 weeks. The BIM group reported changes in family health which were maintained to 52 weeks post intervention. Of the four questions in this domain, two, which ask about the caregiver’s work and financial situation, would not be impacted by either intervention. The other two questions are about the parent’s health and happiness. Perusal of raw data revealed equivalent changes across the four questions; thus, the significant difference in this domain is thought not to be only related to the intervention.

Secondary outcomes

KIDSCREEN-52 self-report

Significant differences between groups favouring CIMT, were found only for the domains physical well-being, psychological well-being, and self-perception at 3 weeks and were not maintained (Table III). Neither group reported changes in the areas of autonomy, parents and home life, financial resources, social support and peers, school environment, or social acceptance. The CIMT group reported immediate changes in physical well-being and psychological well-being that were maintained to 52 weeks, as well as in the domain of moods and emotions (maintained to 26 weeks) and self-perception (not maintained beyond 3 weeks).

Table III.   Estimated mean difference (EMD) over time in quality of life domains between groups receiving constraint-induced movement therapy (CIMT) or bimanual training (BIM) according to KIDSCREEN-52, parent proxy and self-reports
 EMD, parent proxy reportEMD, child self-report
Baseline to 3 weeks (95% CI); pBaseline to 26 weeks (95% CI); pBaseline to 52 weeks (95% CI); pBaseline to 3 weeks (95% CI); pBaseline to 26 weeks (95% CI); pBaseline to 52 weeks (95% CI); p
  1. aChild self-report, n=22; parent proxy report, n=32. bChild self-report, n=19; parent proxy report, n=31. EMD generated from generalized estimating equations. CI, confidence interval.

Physical well-being
 CIMTa2.3 (−1.6 to 6.1); 0.23.1 (−0.5 to 6.6); 0.13.3 (−0.3 to 6.9); 0.075.6 (1.9 to 9.3); 0.0035.2 (0.8 to 9.5); 0.024.6 (0.6 to 8.6); 0.02
 BIMb0.5 (−3.4 to 4.4); 0.81.8 (−3.7 to 7.2); 0.50.0 (−3.8 to 3.8); 0.90.3 (−3.7 to 4.2); 0.91.5 (−3.1 to 6.1); 0.51.2 (−3.1 to 5.5); 0.6
 Difference between groups1.8 (−3.7 to 7.2); 0.52.2 (−2.9 to 7.3); 0.43.3 (−1.9 to 8.5); 0.25.4 (−0.0 to 10.8); 0.053.7 (−2.6 to 10.0); 0.33.4 (−2.4 to 9.3); 0.2
Psychological well-being
 CIMT2.5 (−0.6 to 5.6); 0.12.6 (−0.0 to 5.2); 0.051.8 (−1.3 to 5.3); 0.34.6 (1.6 to 7.7); 0.0034.6 (0.5 to 8.7); 0.034.1 (0.1 to 8.0); 0.04
 BIM1.0 (−2.1 to 4.2); 0.50.1 (−2.9 to 5.8); 0.5−0.6 (−4.4 to 3.2); 0.8−2.1 (−6.2 to 3.3); 0.60.5 (−3.9 to 4.8); 0.80.6 (−3.6 to 4.8); 0.8
 Difference between groups1.4 (−3.4 to 5.4); 0.72.5 (−1.3 to 6.2); 0.22.4 (−2.8 to 7.5); 0.46.1 (1.6 to 10.7); 0.0084.8 (−1.2 to 10.9); 0.14.4 (−1.5 to 10.2); 0.1
Moods and emotions
 CIMT1.1 (−0.9 to 3.8); 0.20.7 (−1.5 to 2.9); 0.51.4 (−1.9 to 4.9); 0.47.6 (4.2 to 10.9); <0.0014.8 (0.8 to 8.8); 0.022.1 (−0.8 to 5.0); 0.1
 BIM−1.1 (−3.4 to 1.2); 0.4−0.2 (−2.4 to 2.0); 0.90.7 (−2.9 to 4.2); 0.73.3 (−0.2 to 6.8); 0.060.9 (−3.2 to 5.1); 0.73.7 (−0.7 to 8.1); 0.1
 Difference between groups2.5 (−0.8 to 5.8); 0.10.9 (−2.2 to 4.0); 0.80.7 (−4.1 to 5.6); 0.84.3 (−0.6 to 9.1); 0.083.9 (−1.9 to 9.6); 0.2−0.4 (−6.5 to 5.6); 0.9
Self-perception
 CIMT2.4 (−0.7 to 5.6); 0.12.9 (−0.5 to 6.2); 0.094.8 (1.4 to 8.3); 0.0064.4 (1.7 to 7.1); 0.0024.0 (−0.8 to 8.8); 0.11.4 (−3.6 to 6.4); 0.6
 BIM0.8 (−2.3 to 4.0); 0.6−1.6 (−2.9 to 6.1); 0.41.0 (−2.7 to 4.6); 0.6−2.8 (−5.9 to 0.3); 0.08−2.0 (−7.2 to 3.1); 0.4−0.1 (−5.4 to 5.1); 0.9
 Difference between groups1.6 (−2.9 to 6.1); 0.54.5 (−0.3 to 9.3); 0.073.9 (−1.1 to 8.9); 0.17.2 (2.9 to 11.5); 0.0036.0 (−1.1 to 13.1); 0.11.5 (−5.7 to 8.7); 0.7
Autonomy
 CIMT−0.5 (−3.4 to 2.5); 0.82.0 (−1.0 to 5.0); 0.22.0 (−0.9 to 4.8); 0.23.6 (0.7 to 6.5); 0.022.1 (−2.3 to 6.5); 0.33.7 (−0.2 to 7.5); 0.06
 BIM1.1 (−1.9 to 4.0); 0.60.2 (−2.8 to 3.3); 0.90.4 (−2.6 to 3.5); 0.82.4 (−0.9 to 5.7); 0.20.5 (−4.0 to 5.0); 0.83.0 (−1.2 to 7.2); 0.2
 Difference between groups−1.5 (−5.7 to 2.6); 0.51.8 (−2.5 to 6.0); 0.41.5 (−2.6 to 5.7); 0.51.2 (−3.0 to 5.4); 0.61.6 (−4.7 to 7.9); 0.60.7 (−4.9 to 6.3); 0.8
Parent relations/home life
 CIMT3.6 (1.1 to 6.2); 0.0052.2 (−0.5 to 4.9); 0.13.4 (0.4 to 6.3); 0.032.4 (−0.8 to 5.6); 0.11.9 (−1.8 to 5.6); 0.31.5 (−2.0 to 5.0); 0.4
 BIM−0.2 (−2.7 to 2.4); 0.90.1 (−2.6 to 2.8); 0.90.5 (−2.7 to 3.7); 0.80.6 (−2.8 to 4.0); 0.71.8 (−2.9 to 6.5); 0.53.1 (−0.6 to 6.9); 0.1
 Difference between groups3.8 (0.2 to 7.4); 0.042.1 (−1.7 to 6.0); 0.32.9 (−1.5 to 7.2); 0.21.8 (−2.9 to 6.5); 0.50.1 (−5.3 to 5.5); 0.9−1.6 (−6.7 to 3.5); 0.5
Financial resources
 CIMT1.8 (−0.3 to 4.0); 0.14.1 (0.4 to 7.8); 0.033.9 (0.7 to 7.0); 0.021.5 (−1.5 to 4.5); 0.34.0 (−1.3 to 9.3); 0.13.2 (−0.9 to 7.2); 0.1
 BIM−2.1 (−4.2 to −0.1); 0.042.2 (−1.4 to 5.7); 0.20.4 (−2.9 to 3.6); 0.81.8 (−1.4 to 5.1); 0.42.6 (−2.8 to 8.0); 0.33.6 (−0.7 to 8.0); 0.1
 Difference between groups4.0 (1.6 to 9.0); 0.0091.9 (−3.2 to 7.1); 0.53.5 (−1.0 to 8.0); 0.1−0.4 (−4.7 to 4.0); 0.91.4 (−6.1 to 9.0); 0.7−0.5 (−6.5 to 5.5); 0.9
Social support and peers
 CIMT−1.9 (−5.5 to 1.6); 0.3−0.8 (−4.7 to 3.2); 0.70.4 (−3.4 to 4.1); 0.81.1 (−3.9 to 6.2); 0.74.7 (−1.5 to 10.8); 0.13.7 (−0.8 to 8.1); 0.1
 BIM2.2 (−1.3 to 5.6); 0.21.0 (−3.0 to 4.9); 0.62.2 (−1.7 to 6.0); 0.31.7 (−3.6 to 7.0); 0.5−0.0 (−6.3 to 6.2); 0.91.7 (−3.0 to 6.4); 0.5
 Difference between groups−4.0 (−9.0 to 0.8); 0.1−1.7 (−7.3 to 3.9); 0.6−1.8 (−7.1 to 3.5); 0.5−0.5 (−7.9 to 6.8); 0.94.7 (−4.1 to 13.5); 0.31.9 (−4.5 to 8.4); 0.6
School environment
 CIMT1.2 (−1.3 to 3.7); 0.30.1 (−2.7 to 2.9); 0.91.0 (−1.6 to 3.7); 0.53.4 (−1.2 to 8.1); 0.12.7 (−2.0 to 7.4); 0.31.0 (−3.7 to 5.7); 0.7
 BIM0.6 (−1.9 to 3.1); 0.61.0 (−1.9 to 3.9); 0.50.4 (−2.4 to 3.3); 0.8−0.1 (−5.1 to 4.9); 0.9−0.6 (−5.6 to 4.5); 0.81.7 (−3.4 to 6.8); 0.5
 Difference between groups0.6 (−2.9 to 4.1); 0.7−0.9 (−4.9 to 3.2) 0.70.6 (−3.3 to 4.5); 0.83.6 (−3.2 to 10.4); 0.33.3 (−3.6 to 10.1); 0.3−0.7 (−7.7 to 6.2); 0.8
Social acceptance
 CIMT−0.2 (−3.2 to 2.8); 0.9−2.5 (−6.2 to 1.2); 0.21.7 (−2.7 to 6.1); 0.5−0.3 (−2.7 to 2.2); 0.8−1.7 (−6.7 to 3.4); 0.53.0 (−1.9 to 7.9); 0.2
 BIM3.1 (0.1 to 6.0); 0.051.8 (−2.0 to 5.5); 0.44.2 (−0.6 to 8.9); 0.09−0.3 (−3.0 to 2.3); 0.8−0.6 (−6.0 to 4.7); 0.82.1 (−3.1 to 7.4); 0.4
 Difference between groups−3.3 (−7.4 to 0.9); 0.1−4.3 (−9.6 to 1.0); 0.1−2.4 (−8.9 to 4.1); 0.50.1 (−3.6 to 3.7); 0.9−1.1 (−8.5 to 6.4); 0.80.8 (−6.4 to 8); 0.8

KIDSCREEN-52 parent proxy report

Immediately after intervention there were significant difference between groups in the area of financial resources, favouring BIM, and in the area of parent relations and home life, favouring CIMT, neither of which was maintained (Table III). There was no difference between groups in other domains. There were no within-group changes in the BIM group for any domain except financial resources (not maintained). In the CIMT group there were immediate changes in self-perception and parent relationships and home life, and the change in self-perception remained significantly different from baseline at 52 weeks.

Discussion

This study reports the QOL outcomes of a large randomized trial comparing equal doses of intensive upper limb training (CIMT and BIM). Similar to previously reported activity and participation outcomes of the study,11,12 there were minimal differences between the training approaches on QOL outcomes. This study uniquely demonstrated that either of the intensive upper limb training approaches delivered using a novel circus-themed group ‘day camp’ can lead to domain-specific changes measured on a condition-specific QOL outcome and that these changes are long lasting. A generic QOL measure appeared less able to determine long-term domain-specific changes in QOL after intervention.

In contrast to the results of this study, previous upper limb rehabilitation trials have failed to identify an impact of intervention on QOL.6–8 A recent Cochrane systematic review investigating the efficacy of intramuscular upper limb botulinum toxin A injections and occupational therapy compared with occupational therapy alone20 found no difference between groups on global measures of well-being using the Child Health Questionnaire6–8 or the Pediatric Quality of Life Inventory.7 An additional study by Redman et al.,21 which used the Pediatric Quality of Life Inventory, found no significant overall effect on QOL but reported some trends favouring botulinum toxin A and occupational therapy in some areas of well-being.20 The Child Health Questionnaire and Pediatric Quality of Life Inventory have been criticized as focusing on measurement of function (e.g. what a child can do or limitations in specific activities) rather than children’s overall well-being (e.g. how children feel about their life) as assessed on the CPQOL-Child.5 This conceptual difference in how QOL is measured may be reflected in these differing results. The unique group-based intervention in the current study, which used a novel and engaging circus theme, may also have contributed to the positive outcomes in QOL reported by both children and their caregivers. The group dynamics, sharing of fun and challenging experiences with children of similar abilities, and establishment of informal networks between families all occurred, but were not formally measured. Each of these aspects may have contributed to the enhanced feelings of well-being experienced by both participating children and their families.

Condition-specific measures of QOL, such as the CPQOL-Child, are designed to be applicable to one group and are considered useful in detecting small changes. In contrast, a generic measure such as the KIDSCREEN-52 is applicable to many groups and can more readily compare outcomes across differing populations.3,22 The results of this study might suggest this to be true, in that the CPQOL-Child was better able to detect domain-specific changes in well-being for children with unilateral CP. Parents and children from both groups reported similar changes in specific domains of the CPQOL-Child. However, this was not the same for the KIDSCREEN-52. Children in the CIMT group reported significant changes between baseline and 3 weeks post intervention in the domains of physical well-being, psychological well-being, and moods and emotions that were maintained to 52 weeks. The parents of children in the CIMT group did not perceive changes in those domains, nor did parents or children in the BIM group. The diverse results on the CPQOL-Child and KIDSCREEN-52 may reflect differences in sensitivity between a condition-specific and generic measure of QOL.

The two domains on the CPQOL-Child demonstrating significant improvements after intervention (feelings about functioning and participation and physical health) appear to directly reflect both the focus of the intervention programmes and specific child/caregiver-identified goals. For example, questions in the domain about functioning include some that address how children feel about how they use their arms and hands and the ability to dress, eat, and drink independently.15 Both interventions aimed to improve arm and hand skills and, overall, 70% of goals identified by children/caregivers and addressed in the intervention related to self-care activities.12 Seventy-five per cent of self-care goals addressed aspects of dressing, eating, or drinking. In the domain of participation and physical health, questions included those that addressed participation in school and sporting activities and whether children were able to do the things they wanted.15 Overall, 21% of the goals identified by children/caregivers related to leisure, sport, and recreation. Significant gains were made in the achievement of individualized goals12 and these appear also to be reflected in improvement in well-being across these two related domains of QOL.

A limitation of the current study is the lack of a control group that received no intervention, which may have shed additional light on the longitudinal changes in QOL that might occur as a result of psychosocial and cognitive development and changing expectations at home, school, and with peer relationships. Additionally, not all children included in the study could independently self-report their QOL owing to immaturity or cognitive difficulties. Given the obvious differences between child and parent reports, the lack of perspective of younger children limits a full evaluation of the impact of an intensive upper limb functionally focused training programme on QOL. Adolescents included in this study completed the CPQOL-Child, as the CPQOL-Teen version was not available at the start of the trial. The CPQOL-Teen version has additional themes such as independence, relationships, acceptance of disability, and getting on well at school, which could not be assessed in the current study. The impact of these interventions on the domains of QOL may be different for younger children and for adolescents. Further subgroup analysis to determine differential effects as a result of age could not be performed owing to the relatively small numbers of adolescents in the study. The sample size for the overall INCITE trial of 63 (32 pairs) was adequately powered based on the primary outcome of the study. However, for the CPQOL-Child, a larger sample size of 72 was required and therefore this study may not have been adequately powered to detect QOL outcomes. Although both groups experienced statistically significant changes, particularly in the areas of feelings about functioning and participation and physical health, it remains unclear whether the extent of those changes is clinically meaningful. The maintenance of changes over a 52-week period may suggest clinical significance.

Importantly, the findings of this study highlight that an intensive block of 60 hours of upper limb training, not only impacts on activity,11 individualized, and participation outcomes,12 but also translates into domain-specific changes in QOL. This contradicts recommendations that intervention studies should only measure constructs within the same domain of the International Classification of Functioning, Disability and Health that the intervention targets.20 Our findings demonstrate the benefit of a broader examination of intervention outcomes, and importantly highlights that well-designed, goal-directed, and functionally focused interventions can impact more broadly on a child’s sense of well-being.

Conclusion

In this randomized comparison trial, findings suggest that intensive goal-directed upper limb training using either a unimanual or bimanual approach can impact more broadly on children’s perception of well-being in areas relating to feelings about functioning and participation and physical health. QOL assessment using a condition-specific outcome measure is recommended for future similarly focused interventions.

Acknowledgements

We would like to thank all the children and families who participated in this study. This study was supported by a National Health and Medical Research Council Dora Lush postgraduate scholarship (LS 384488), Career Development Grant (RB NHMRC 473860), and project grant for INCITE: A Randomized Trial of Novel Upper Limb Rehabilitation in Children with Congenital Hemiplegia (NHMRC 368500).

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