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Abstract

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information

Aim  To determine if constraint-induced movement therapy (CIMT) is more effective than bimanual training (BIM) in improving upper limb activity outcomes for children with congenital hemiplegia in a matched-pairs randomized trial.

Method  Sixty-three children (mean age 10.2, SD 2.7, range 5–16y; 33 males, 30 females), 16 in Manual Ability Classification System level I, 46 level II, and 1 level III and 16 in Gross Motor Function Classification level I, 47 level II) were randomly allocated to either CIMT or BIM group day camps (60 hours over 10 days). The Melbourne Assessment of Unilateral Upper Limb Function assessed unimanual capacity of the impaired limb and Assisting Hand Assessment evaluated bimanual coordination at baseline, 3 and 26 weeks, scored by blinded raters.

Results  After concealed random allocation, there was no baseline difference between groups. CIMT had superior outcomes compared with BIM for unimanual capacity at 26 weeks (estimated mean difference [EMD] 4.4, 95% confidence interval [CI] 2.2–6.7; p<0.001). There was no other significant difference between groups post-intervention. Both groups demonstrated significant improvements in bimanual performance at 3 weeks, with gains maintained by BIM at 26 weeks (EMD 2.3; 95% CI 0.6–4.0; p=0.008).

Interpretation  Overall, there were only small differences between the two training approaches. CIMT yielded greater changes in unimanual capacity of the impaired upper limb compared with BIM. Results generally reflect specificity of practice, with CIMT improving unimanual capacity and BIM improving bimanual performance. Considerable inter-individual variation in response to either intervention was evident. Future research should consider serial sequencing unimanual then BIM approaches to optimize upper limb outcomes for children with congenital hemiplegia.


Abbreviations
AHA

Assisting Hand Assessment

BIM

Bimanual training

CIMT

Constraint-induced movement therapy

CIMT-BIM

Combined CIMT and bimanual training

EMD

Estimated mean difference

GEE

Generalized Estimating Equations

HABIT

Hand Arm Bimanual Intensive Training

JTTHF

Jebsen Taylor Test of Hand Function

MAS

Modified Ashworth Scale

MUUL

Melbourne Assessment of Unilateral Upper Limb Function

SDD

Smallest detectible difference

What this paper adds

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information
  •  This is the largest randomized trial to compare directly equal amounts of two intensive upper limb training approaches using activity-based day camps.
  •  Gains in upper limb function reflect the mode of training, with improved unimanual capacity following CIMT and gains in bimanual performance following BIM training.
  •  Considerable inter-individual variation in response to either intervention was evident.

Congenital hemiplegia is the most common form of cerebral palsy (CP), with a prevalence of one in 1300 live births.1 Periventricular white matter, cortical and subcortical lesions,2 size and location of the cerebral insult vary significantly, as does the resultant impact on unimanual upper limb capacity and bimanual performance.3 Together, these deficits in upper limb function affect the performance of daily activities.

Constraint-induced movement therapy (CIMT), involves constraint applied to the unimpaired upper limb coupled with intensive training of unimanual skills in the hemiplegic limb.4–6 Our systematic review of all upper limb non-surgical interventions7 identified a small number of randomized controlled trials of CIMT5,6 that demonstrated immediate gains in the amount of use and improved movement efficiency of the impaired upper limb; however, sample sizes were small with significant baseline inequality between groups. Outcome measures differed across trials, the majority of which had no reported validity or reliability for the population.6 A larger controlled clinical trial comparing CIMT with conventional care demonstrated improved bimanual performance following CIMT.4 The amount of intervention has varied between studies ranging from 60 to 120 hours, delivered either intensively for up to 6 hours a day for 10 to 21 days,5,6 or using a model of 2 to 3 hours a day over a 2-month period.4

In contrast to CIMT, Hand Arm Bimanual Intensive Training (HABIT) provides intensive training of bimanual coordination.5,8 This approach was developed in response to identified limitations in CIMT, that is, the inability of CIMT to enable practice of bimanual skills, particularly functional activities that are inherently bimanual. Evidence from one trial demonstrated a modest treatment effect for bimanual performance on the Assisting Hand Assessment (AHA), but no gains in unimanual capacity on the Jebsen Taylor Test of Hand Function (JTTHF) compared with a delayed-treatment control group.8 CIMT and HABIT have mainly been compared with control groups receiving regular therapy,4–6 however two recent studies have directly compared interventions receiving the same intensity of training. A combined CIMT and bimanual training approach (CIMT-BIM) was compared with an equal amount of standard care and found to be superior in improving movement efficiency and manual skills of the impaired upper limb and bimanual performance.9 In contrast, a small quasi-randomized trial directly comparing CIMT with HABIT found no difference between the two training approaches to improve movement efficiency and bimanual performance.10 Gordon10 concluded a larger randomized trial was required with longer-term follow-up to determine the retention of treatment effects.

The INCITE study is a single-blind, matched-pairs randomized clinical trial comparing the efficacy of CIMT with an equal intensity of BIM training, reporting outcomes across all domains of the World Health Organization’s International Classification of Functioning, Disability and Health.8,11 This paper reports the activity outcomes of the study. The primary hypothesis is that CIMT will reduce upper limb activity limitations to a greater extent than BIM training, as CIMT studies have demonstrated improved unimanual capacity of the impaired upper limb with possible gains in bimanual performance, and HABIT has shown modest gains in bimanual performance, but limited changes in unimanual capacity. Also, based on motor learning theory and the concept of specificity of practice, it is hypothesized that CIMT will lead to greater gains in unimanual capacity compared with BIM training, however BIM training will lead to greater improvements in bimanual performance compared with CIMT.

Method

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information

Participants

Children were recruited through public and private medical specialists in Queensland and Victoria, Australia. They were eligible for inclusion if they had: (1) congenital hemiplegia and were aged 5 to 16 years; (2) the ability to follow instructions (determined during screening assessment and in consultation with caregivers); (3) predominant spasticity with modified Ashworth Scale (MAS) grades of between 1 and 3 for wrist flexors, forearm pronators and/or thumb adductors interfering with upper limb function. Children were ineligible if they had: (1) predominant dystonia and/or muscle contracture (MAS grades >3); (2) previous upper limb orthopaedic surgery; (3) serial casting or botulinum toxin (BoNT-A) injections in the upper limb within the 6 months before commencement of the intervention.

Design and procedure

Following screening assessments, children were matched in pairs according to age (12mo age bands), sex, side of hemiplegia and function based on MUUL scores (within 10%). Once matched, children were randomized to pairs using a computer generated list of random numbers and concealed envelopes opened by non-study personnel. A matched-pairs design minimized the likelihood of group differences at baseline, an identified limitation in upper limb rehabilitation studies.5,8

Outcome measures were conducted at baseline, 3 and 26 weeks following intervention by four experienced occupational therapists and physiotherapists who were aware of group allocation. The primary outcome measures were videotaped and scored in random order by trained occupational therapists masked to group allocation. Classification of hand function was achieved using the Manual Ability Classification System (MACS),12 Zancolli Scale,13 and the House et al.14 functional grading system.

Sample size

Sample size was calculated to detect change in the functional effects of CIMT and BIM training at 26 weeks. Based on data from a previous study,15 we proposed a mean difference of 7 units (10% of the anticipated control group mean at baseline on the MUUL)16 as the minimum difference required to have substantial clinical relevance. Our pilot data in a study of intramuscular BoNT-A injections and upper limb training yielded standard deviation (SD) changes of 7.6 and 9.6 units in the two groups. Based on a t-test comparison of changes using an SD of 9 units for both groups, a significance (alpha) level of 0.05, and 80% power, we required a minimum of 26 children in each group (total sample of 52).

Interventions

Each intervention was delivered in groups of 9 to 13 children for 6 hours daily for 10 days using an intensive day camp model (total 60 hours of intervention). This level of intervention was chosen as it was clinically more feasible than more intensive models of CIMT (126 hours over 21 days)6 and has been used successfully in previous CIMT studies.5 In total 6 day camps were run at community sporting facilities in Melbourne and Brisbane, Australia using a novel circus theme to optimize children’s motivation for engagement and participation. Each pair of camps (CIMT and BIM training) were grouped by age to ensure activities were developmentally tailored. Interventions used a goal-directed, activity-based framework, employing principles of motor learning, including specific task practice, fostering problem solving (individual and within the group framework), and modifying task and environmental constraints to support goal attainment. Specifically, they involved fine motor activities, functional goals that children/caregivers had identified before the camp, 2-hour circus training, mealtimes, gross upper limb games, and debriefing. All activities were analysed and selected to target specific movement components required for goal achievement, were novel, fun, motivating and fostered self-generated, voluntary repetition of desired movements.17 For the BIM training group, we adopted the strategy used by HABIT, providing explicit instructions on how each hand should be used before each activity. For both CIMT and BIM training, the focus was on completion of whole activities.

For pragmatic reasons the camps were run consecutively. The BIM training group was conducted first, and then tasks were modified for the CIMT group to accommodate the unimanual nature of the intervention. Each group received the same amount and overall content of intervention, delivered in the same environment. A core team of four experienced occupational therapists and physiotherapists conducted the camps, coordinating and training therapist volunteers, therapy students, circus trainers, and community youth workers with a ratio of one therapist for two children.

Participants in the CIMT group wore a tailor-made glove on their unimpaired limb while attending the day camp, which was removed for toileting, aerial circus activities, and the low ropes course because of safety. When the glove was removed for circus activities, the fingers of the unimpaired hand were taped together with elastoplast to simulate the glove. The glove was constructed from breathable fabric with a volar plastic insert to prevent grasp. This design was chosen so that children could use their hand as a support, was less intrusive than a full arm cast or sling and potentially safer as children could use their hand for safety; it has been used successfully in other CIMT trials.4

Outcome measures

Primary outcomes

The MUUL16 was selected to measure quality of movement of the impaired upper limb, and the AHA18 to measure bimanual performance. Our recent systematic review of upper limb activity measures found the MUUL to be the best measure of unimanual capacity and the AHA the only measure of bimanual performance with good evidence of validity and reliability.19 The MUUL was reported as a percentage with higher scores reflecting greater quality of upper limb movement. As studies of intrarater and interrater reliability of the MUUL have varied in terms of age and type of CP, we sought to establish intrarater reliability for our independent rater. Twenty videoed MUUL assessments were randomly selected and re-scored 3 months after initial scoring. Intraclass correlation coefficients (ICC, 3.1) were calculated with 95% confidence intervals (95% CI) from MUUL percentage scores.20 The ICC for the total percentage score of MUUL was 0.93 (95% CI 0.84–0.97). The standard error of measurement (SEM) was 2.7% and smallest detectible difference (SDD) was 7.4%. The mean difference of total MUUL percentage scores was 1.1% (95% limits of agreement −5.0 to 7.1).

The AHA was reported in log odds probability units (logits) using Winsteps, version 3.65.0 (copyright 2006, John M Linacre, Winsteps, Chicago, IL, USA) with item calibrations anchored according to their level of difficulty.21 The logit scale was transformed by the test developers to a more easily interpretable 1 to 100 logit scale with higher scores reflecting greater ability to use the impaired upper limb in bimanual activities. A change of four raw score points is considered to represent a clinically significant effect.21

Secondary outcomes

The following impairment measures were included: grip strength using a hand-held dynamometer (Smedley; Takei Scientific Instruments Co Ltd, Niigata, Japan); and sensory measures with a moving two-point discrimination test on the pulp of the index finger using the Disk-criminator (Mackinnon Dellon Partnership, Baltimore, MD, USA). Stereognosis was assessed by matching three familiar objects and six similar matched objects.17 Test–retest and interrater reliability were high.22

Movement efficiency of the impaired upper limb was measured using the JTTHF.23 The total time to complete all six tasks was recorded in seconds with smaller numbers reflecting faster performance. The JTTHF was selected as a secondary measure. While there may be demonstrated change in previous upper limb studies,5 as yet there is no evidence of validity or reliability for children with congenital hemiplegia.19 Grip strength and JTTHF were measured on both the impaired and unimpaired hand to determine whether there were deleterious effects of wearing a constraint on the unimpaired limb.

Statistical analysis

Analyses were on an intention-to-treat basis according to CONSORT24 guidelines using STATA 10 (StataCorp, College Station, TX, USA). Baseline data from each outcome measure were summarized for each treatment group and descriptive statistics calculated. A significance level of 0.05 (two-tailed) was used. Continuous data were compared between groups by fitting a regression model using generalized estimating equations (GEE) to baseline, 3- and 26-week measurements with an interaction term between intervention group and three-level factor indicating time of measurement. Matching characteristics of age, sex, and side of hemiplegia were entered as covariates. An exchangeable working correlation matrix for the repeat measurements on participants was used.25

The study was approved by the Ethics in Human Research Committees at The Royal Children’s Hospital, Melbourne; La Trobe University; The Royal Children’s Hospital and Health Services District, Brisbane; and The University of Queensland. Written informed consent was obtained from parents and adolescents aged 12 years or older and verbal assent from younger participants.

Results

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information

Sixty-four children (32 matched pairs) were randomly assigned to treatment and 62 children received their allocated intervention. There was one dropout on day two of the camp from the BIM training group, because of pre-existing emotional and behavioural difficulties. Compliance was 100% for CIMT, and 97% with BIM training (Fig. 1).

image

Figure 1.  Trial profile following CONSORT guidelines. UL, upper limb; CIMT, constraint-induced movement therapy; BIM, bimanual training.

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Baseline comparisons

After matching and concealed random allocation within pairs, there were no differences between groups on demographic or baseline measures (Table I). The mean age was 10 years 2 months (range 5–16y) and children were predominantly classified in Level II for both the Manual Ability Classification System (MACS) and the Gross Motor Function Classification System (GMFCS). Four children were matched on all variables except side of hemiplegia, resulting in fewer children in the BIM training group with right-sided hemiplegia. Half the children were not receiving any therapy before the study and 17% were receiving regular weekly or fortnightly physiotherapy and/or occupational therapy.

Table I.   Participant demographics and baseline characteristics of CIMT and BIM groups
CharacteristicsCIMT (n=32)BIM (n=31)
  1. aNorth American usage: mental retardation.Spast, spasticity; MACS, Manual Ability Classification System; GMFCS, Gross Motor Function Classification System; CIMT, constraint-induced movement therapy; BIM, bimanual therapy; CI, confidence interval; IQR, interquartile range; OT, occupational therapy; M2PD, moving two-point discrimination; MUUL, Melbourne Assessment of Unilateral Upper Limb Function; AHA, Assisting Hand Assessment; JTTHF, Jebsen Taylor Test of Hand Function.

Demographic
 Age, mean (95%CI), y:mo10:1 (9:1–11:0)10:2 (9:2–11:1)
 Sex, male n (%)17 (53)16 (52)
 Side of hemiplegia, left, n (%)16 (50)11 (36)
 Motor type, dystonia+spasticity, n (%)1 (3)3 (10)
 Epilepsy, n (%)5 (16)8 (26)
 Learning disabilitya, 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)
 Zancolli scale, n (%)
  Level 118 (56)15 (48)
  Level 2a9 (28)11 (36)
  Level 2b5 (16)5 (16)
 House scale, n (%)
  Spontaneous use3 (9)4 (13)
  Active assist24 (75)25 (81)
  Passive assist5 (16)2 (6)
School
 Typical school, n (%)29 (91)29 (94)
 Special school, n (%)3 (9)2 (6)
Concurrent occupational therapy, n (%)
 Weekly or fortnightly2 (6)4 (13)
 Monthly5 (16)3 (10)
 Other9 (28)8 (26)
Concurrent physiotherapy, n (%)
 Weekly or fortnightly6 (19)3 (10)
 Monthly4 (12)1 (3)
 Other5 (16)9 (29)
Body structure, function
 Grip impaired (kg), median (IQR)5.2 (2.7–6.6)4.8 (3.6–7.1)
 Grip unimpaired (kg), median (IQR)16 (11.9–18.6)14.7 (12.3–17.4)
Sensation
 Stereognosis (/9), median (IQR)5 (4–7)6 (3–8)
 M2PD (mm), median (IQR)5 (3–7.8)5 (3–7)
Activity
 MUUL, mean (95% CI)67.1 (62.5–71.6)70.8 (66.7–74.9)
 AHA, mean logits (95% CI)61.7 (57–66.4)63.0 (58.4–67.6)
 JTTHF (impaired), mean (95% CI)365.7 (294.1–437.3)323 (261.2–384.8)
 JTTHF (unimpaired), median (IQR)41.0 (41–60.5)44.5 (40–52)

Amount of intervention

Overall, 58% (n=376) received the allocated 60 hours of intervention in the study. A further 39% (n=12) in BIM training and 41% (n=13) in CIMT received 54 hours of therapy owing to public holidays or illness. One child in BIM training received 42 hours because of illness.

Concurrent therapy

At 3 and 26 weeks, there were no changes to pre-existing therapy for all children with the exception of one child (CIMT group) who received upper limb BoNT-A injections between the 3- and 26-week follow-ups.

Outcome measures

Primary outcomes

All outcomes (means or medians and 95% CIs) are reported for each group at each follow-up period (Table S1, published online). Results of regression modelling using GEEs (Table II) found a significant group by time interaction on the MUUL at 26 weeks favouring CIMT (EMD 4.4, 95% CI 2.2–6.7; p<0.001). There was no other between group differences at either 3 or 26 weeks. The CIMT group made immediate significant gains on all measures, with improved unimanual capacity on the MUUL (EMD 2.8, 95% CI 1.2–4.3; p<0.001) and bimanual performance on the AHA (EMD 3.1, 95% CI 1.4–4.7; p<0.001; Table III and Fig. 2). At 26 weeks, the CIMT group demonstrated continued improvement on the MUUL (EMD 4.5, 95% CI 2.9–6.1; p<0.001), however they did not retain initial significant gains on the AHA. The BIM training group demonstrated initial gains on the AHA (EMD 1.9, 95% CI 0.2–3.6; p=0.03) that were maintained at 26 weeks (EMD 2.3, 95% CI 0.6–4.0; p=0.008).

Table II.   Results of comparison of activity outcomes for the CIMT and BIM training groups at 3 and 26 weeks
FactorsModel regression coefficient (95% CI)p value
  1. CIMT, constraint-induced movement therapy; BIM training, bimanual training; MUUL, Melbourne Assessment of Unilateral Upper Limb Function; AHA, Assisting Hand Assessment; JTTHF, Jebsen Taylor Test of Hand Function.

Model 1: MUUL
 Constant68.8 
 Treatment−3.2 (−8.7 to 2.4)0.3
 Timing 1 (3wks)0.9 (−0.6 to 2.5)0.2
 Timing 2 (26wks)0.0 (−1.5 to 1.6)0.9
 Treatment by time 11.8 (−0.4 to 4.0)0.1
 Treatment by time 24.5 (2.2 to 6.7)<0.001
 Age0.2 (−0.8 to 1.2)0.7
 Sex3.1 (−2.3 to 8.4)0.3
 Side of hemiplegia−3.4 (−9 to 2.1)0.2
Model 2: AHA
 Constant69.0 
 Treatment−0.4 (−6.4 to 5.7)0.9
 Timing 1 (3wks)1.9 (0.2 to 3.6)0.03
 Timing 2 (26wks)2.3 (0.6 to 4.0)0.008
 Treatment by time 11.2 (−1.2 to 3.5)0.3
 Treatment by time 2−0.7 (−3.1 to 1.8)0.6
 Age−0.3 (−1.4 to 0.9)0.6
 Sex2.3 (−3.5 to 8.2)0.4
 Side of hemiplegia−5.0 (−11.1 to 1.0)0.1
Model 3: JTTHF
 Constant505.5 
 Treatment30.7 (−58.4 to 119.9)0.7
 Timing 1 (3wks)−14.9 (−36.5 to 6.8)0.2
 Timing 2 (26wks)−34.3 (−56.2 to −12.5)0.002
 Treatment by time 1−11.1 (−41.7 to 19.4)0.5
 Treatment by time 2−25.6 (−57 to 5.7)0.1
 Age−17.3 (−34.2 to 0.4)0.4
 Sex−36.8 (−123.8 to 50.1)0.4
 Side of hemiplegia35.2 (−54.4 to 124.8)0.5
Table III.   Difference and changes over time for activity outcomes between CIMT and BIM training groups
Baseline toDifference between groupsChange in CIMTChange in BIM
3wksa26wksb3wksc26wksc3wksc26wksc
  1. aA positive value in this column indicates a greater increase from baseline to 3 weeks for the CIMT group for the MUUL and AHA, and a negative value indicates a greater decrease (time) for the CIMT group on the JTTHF (i.e. faster performance). bA positive value in this column indicates a greater increase from baseline to 26 weeks for the CIMT group for the MUUL and AHA, and a negative value indicates a greater decrease (time) for the CIMT group on the JTTHF (i.e. faster performance). cA positive value in these columns for MUUL and AHA indicates an increase from baseline to the follow-up period, and a negative value for the JTTHF indicates improved performance from baseline. dEstimated mean difference using Generalized Estimating Equations. MUUL, Melbourne Assessment of Unilateral Upper Limb Function; AHA, Assisting Hand Assessment; JTTHF, Jebsen Taylor Test of Hand Function; CIMT, constraint-induced movement therapy; BIM, bimanual training.

Estimated mean difference (95% CI)d
 MUUL1.8 (−0.3 to 4.0) 0.14.4 (2.2 to 6.7) <0.0012.8 (1.2 to 4.3) <0.0014.5 (2.9 to 6.1) <0.0010.9 (−0.6 to 2.5) 0.30.0 (−1.5 to 1.6) 0.9
 AHA1.2 (−1.2 to 3.5) 0.3−0.7 (−3.1 to 1.7) 0.63.1 (1.4 to 4.7) <0.0011.6 (−0.1 to 3.4) 0.061.9 (0.2 to 3.6) 0.032.3 (0.6 to 4.0) 0.008
 JTTHF−11.1 (−41.7 to 19.4) 0.5−25.7 (−57.0 to 5.7) 0.1−26 (−47.6 to −4.4) 0.02−60 (−82.5 to −37.5) <0.001−14.9 (−36.3 to 6.5) 0.2−34.3 (−56.2 to −12.5) 0.002
image

Figure 2.  Results of Melbourne Assessment of Unilateral Upper Limb Function (Melbourne) and the Assisting Hand Assessment (AHA) for constraint induced movement therapy (CIMT) and bimanual training BIM at 3 and 26 weeks. Squares and diamonds indicate group means for BIM and CIMT respectively; error bars represent 95% confidence intervals.

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Secondary outcomes

There was no difference between groups on any secondary outcome measure; however both groups improved movement efficiency of the impaired upper limb by 26 weeks. The CIMT group demonstrated immediate improvements in movement efficiency (JTTHF; EMD-26, 95% CI, −47.6 to −4.4; p<0.001) that further improved by 26 weeks (Table III). The BIM training group demonstrated a significant change from baseline to 26 weeks on the JTTHF (EMD −34.3, 95% CI, −56.2 to −12.5; p=0.002). There were no changes in sensation (stereognosis, moving two-point discrimination) or grip strength on the impaired upper limb, and no deleterious effect on the unimpaired upper limb for the CIMT group at any follow-up (Table S1).

Adverse events

There was no major adverse event. All children in the CIMT group tolerated wearing the glove on their unimpaired hand.

Discussion

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information

This study investigated whether unimanual training was superior to bimanual training in improving quality and efficiency of movement of the impaired upper limb and its use as an assisting hand in bimanual activities. Matched pairing according to age, sex, side of hemiplegia and upper limb function strengthens the study design by minimizing baseline differences in this generally heterogenous population. Overall, there were only small differences between the two training approaches. Results reflect the mode of upper limb training with the CIMT group demonstrating statistically significant gains in unimanual capacity (MUUL) and the BIM training group improvements in bimanual performance (AHA).

Gains in unimanual capacity demonstrated by the CIMT group were evident for both quality of upper limb movement (MUUL) and movement efficiency (JTTHF). Significant immediate changes on both measures for the CIMT group were followed by continued improvement to 26 weeks. However, change on the MUUL demonstrated by the CIMT group, whilst statistically significant, failed to exceed the 7.4% determined to be of clinical importance.

The MUUL has previously been used as an outcome comparing CIMT-BIM to an equal amount of usual care,9 with similar changes in MUUL percentage scores to the current study. In a further prospective pre-post feasibility study of CIMT, the MUUL demonstrated improvement for one child post-intervention that also failed to reach clinical significance.26 This might suggest that the MUUL is not sensitive to capturing change in upper limb movement quality following intensive training. This may relate to the focus of the measurement tool itself. Fifty-four per cent of items on the MUUL measure impairment (e.g. range of motion) rather than activity performance.19 Both interventions in the present study were activity based and may have less impact on impairments (e.g. spasticity and alignment) compared with other interventions such as intramuscular BoNT-A injections aimed at reducing spasticity and improving upper limb function.

Overall group estimated mean change in bimanual performance (AHA) was modest following either training. The effect size (ES 0.22) was greater than HABIT (ES 0.16),8 however smaller than CIMT-BIM (ES 0.43)9 and CIMT (ES 1.12).4,27 Differences in study populations could account for variable responses to intervention. First, studies by Eliasson et al.4 and Aarts et al.9 targeted younger children (18mo–4y and 2y 6mo–8y respectively), while HABIT8 and the current study had a broader age range to 16 years. This might suggest that younger children may achieve greater changes in bimanual performance than older children following intensive upper limb training. Second, severity of upper limb impairment differed between studies. The current study comprised 97% of children classified in either MACS level I or II. In contrast, CIMT-BIM9 comprised 24% of children in MACS level III, indicating a greater severity of impairment. Similarly, a study of CIMT4 included children with more severe hand involvement (mean AHA logit score of −2.86 and −2.22 for the intervention and control groups respectively). In contrast, the current study included children with less severe bimanual performance difficulties (mean AHA logit of 1.5). This suggests that children with poorer hand function at baseline could achieve greater gains in bimanual performance following intensive upper limb intervention. Secondary analysis of characteristics of best responders in the INCITE trial found overall, 47% of children achieved a clinically significant response on the AHA, however, age and severity were not identified as significant predictors of favourable bimanual performance outcomes (unpublished material, Sakzewski L, Ziviani J, Boyd RN.). These results highlight the heterogenous nature of the population of children with congenital hemiplegia who exhibit variable responses to intervention and as yet, definitive predictors of clinically significant outcomes are not clearly understood.

There was no change on any of the impairment measures of sensation (moving two-point discrimination and stereognosis), or grip strength on either the impaired or unimpaired limb between baseline and post-intervention. These results confirm there are no deleterious effects of CIMT on the constrained unimpaired limb and gains in upper limb function can occur in the absence of any change in impairment. Results provide further evidence to challenge the principles underpinning neurodevelopmental therapy models that are still commonly used in clinical practice today. Results support an activity-based motor learning approach that focuses on increasing activity rather than addressing impairments with the desire to improve activity limitations.

Similar to other studies of CIMT, we encountered very few difficulties and no adverse events with children wearing the constraint glove. We had high retention with very few dropouts with either intervention. The novel circus theme, group experience, and appropriate level of challenge helped to counterbalance potential frustration.28 There was also strong engagement noted in the CIMT group once they had become accustomed to the glove. Motivation to maintain continual bimanual use was more challenging in the BIM training group.

A limitation of the current study is the lack of a true control group. Although considered, the heterogenous population of children with congenital hemiplegia and the complications of matching children into three groups pragmatically precluded this option. However, given the limited data regarding the natural history of upper limb development in children with congenital hemiplegia, a control group may have shed additional light on the potential for change following intervention.

The presence of intellectual impairment did not preclude participation in the study, with five children attending special education successfully participating. As such, this study is more representative of the population of children with congenital hemiplegia than previous studies that limited inclusion based on intellectual functioning.5 In the present study, large intra-individual variability was evidenced with a broad continuum of mild to severe upper limb involvement at baseline and in response to intervention. The next important step is to determine specific child characteristics that lead to significant clinical improvements and retention of treatment effects to 12 months post-intervention.

Conclusion

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information

In a large single-blind, matched-pairs randomized trial, findings suggest that there are small differences between the two training approaches. CIMT was superior to BIM training to achieve gains in unimanual capacity and BIM training improved bimanual performance. Considerable inter-individual variation in response to upper limb training was evident and requires further investigation to determine predictors of clinically important change. Based on our outcomes, we propose future research should determine if a sequential combination of unimanual and bimanual training approaches further enhances upper limb outcomes for children with congenital hemiplegia compared with standard care.

Acknowledgements

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information

We thank all the children and families who participated in this study. We thank Kym Corn and Tracey Carson, the occupational therapists for independently scoring MUUL and AHA assessments. We also thank Rose Gilmore, the occupational therapist for the coordination of the Melbourne camps and Kerry Provan, the occupational therapist for coordinating the Brisbane camps. We thank and acknowledge the support of the YMCA of Victoria and all the volunteers who helped during the camps, visiting volunteer therapists (Katrijn Klingels, Professor Hilda Feys, Dr Anna Mackay, Madonna Jeffries), the therapy staff from the Department of Rehabilitation, Cerebral Palsy Health at the Royal Children’s Hospital, Brisbane, Flipside Circus, Brisbane, and Circus Oz, Melbourne. We thank Lena Krumlinde-Sundholm for transforming our raw AHA data to logits. We extend our thanks to Associate Professor Leonid Churilov, Division of Statistics and Decision Support, National Stroke Research Institute, Australia and Dr Rob Ware, School of Medicine, University of Queensland, Australia for their statistical advice.

Funding support was received from the National Health and Medical Research Council for the Dora Lush postgraduate scholarship (LS 384488), a Career Development Grant (RB NHMRC 473860), and a project grant for INCITE: A Randomised Trial of Novel Upper Limb Rehabilitation in children with Congenital Hemiplegia (NHMRC 368500).

References

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information
  • 1
    Stanley F, Blair E, Alberman E. Cerebral Palsies: Epidemiology and Causal Pathways. Clinics in Developmental Medicine No. 151. London: Mac Keith Press, 2000.
  • 2
    Cioni G, Sales B, Paolicelli P, Petacchi E, Scusa M, Canapicchi R. MRI and clinical characteristics of children with hemiplegic cerebral palsy. Neuropediatrics 1999; 30: 24955.
  • 3
    Sakzewski L, Ziviani J, Boyd R. The relationship between unimanual capacity and bimanual performance for children with congenital hemiplegia. Dev Med Child Neurol 2009; 52: 8116.
  • 4
    Eliasson AC, Krumlinde-Sundholm L, Shaw K, Wang C. Effects of constraint-induced movement therapy in young children with hemiplegic cerebral palsy: an adapted model. Dev Med Child Neurol 2005; 47: 26675.
  • 5
    Charles J, Wolf S, Schneider J, Gordon A. Efficacy of a child-friendly form of constraint-induced movement therapy in hemiplegic cerebral palsy: a randomized control trial. Dev Med Child Neurol 2006; 48: 63542.
  • 6
    Taub E, Ramey SL, DeLuca S, Echols K. Efficacy of constraint-induced movement therapy for children with cerebral palsy with asymmetric motor impairment. Pediatrics 2004; 113: 30512.
  • 7
    Sakzewski L, Ziviani J, Boyd R. Systematic review and meta-analysis of therapeutic management of upper-limb dysfunction in children with congenital hemiplegia. Pediatrics 2009; 123: 1111E22E.
  • 8
    Gordon AM, Schneider JA, Chinnan A, Charles JR. Efficacy of a hand-arm bimanual intensive therapy (HABIT) in children with hemiplegic cerebral palsy: a randomized control trial. Dev Med Child Neurol 2007; 49: 8308.
  • 9
    Aarts P, Jongerius P, Geerdink Y, van Limbeek J, Geurts A. Effectiveness of modified constraint-induced movement therapy in children with unilateral spastic cerebral palsy: a randomized controlled trial. Neurorehabil Neural Repair 2010; 24: 50918.
  • 10
    Gordon AM, Chinnan A, Gill S, Petra E, Hung YC, Charles J. Both constraint-induced movement therapy and bimanual training lead to improved performance of upper extremity function in children with hemiplegia. Dev Med Child Neurol 2008; 50: 9578.
  • 11
    World Health Organization. International Classification of Functioning, Disability and Health. Geneva: WHO, 2001.
  • 12
    Eliasson A, Krumlinde-Sundholm L, Rosblad B, et al. The Manual Ability Classification System (MACS) for children with cerebral palsy: scale development and evidence of validity and reliability. Dev Med Child Neurol 2006; 48: 54954.
  • 13
    Zancolli EA, Zancolli ER. Surgical management of the hemiplegic spastic hand in cerebral palsy. Surg Clin North Am 1981; 61: 395406.
  • 14
    House J, Gwathmey F, Fidler M. A dynamic approach to the thumb-in palm deformity in cerebral palsy. Evaluation and results in fifty-six patients. J Bone Joint Surg 1981; 63: 21625.
  • 15
    Boyd R. The Central and Peripheral Effects of Botulinum Toxin A in Children with Cerebral Palsy. Melbourne: La Trobe University, 2004.
  • 16
    Randall M, Johnson I, Reddihough D. The Melbourne Assessment of Unilateral Upper Limb Function: Test Administration Manual. Melbourne: Royal Children’s Hospital, 1999.
  • 17
    Boyd R, Sakzewski L, Ziviani J, et al. INCITE: a randomised trial comparing constraint induced movement therapy and bimanual training in children with congenital hemiplegia. BMC Neurol 2010; 10: 4, published online: DOI: 10.1186/1471-2377010-4.
  • 18
    Krumlinde-Sundholm L, Eliasson A. Development of the assisting hand assessment: a rasch-built measure intended for children with unilateral upper limb impairments. Scand J Occup Ther 2003; 10: 1626.
  • 19
    Gilmore R, Sakzewski L, Boyd R. A systematic review of upper limb activity measures for 5–16 year old children with congenital hemiplegia. Dev Med Child Neurol 2009; 52: 1421.
  • 20
    Shrout P, Fleiss J. Intraclass correlations: uses in assessing rater reliability. Psychol Bull 1979; 86: 4208.
  • 21
    Holmefur M, Aarts P, Hoare B, Krumlinde-Sundholm L. Test-retest and alternate forms reliability of the assisting hand assessment. J Rehabil Med 2009; 41: 88691.
  • 22
    Klingels K, De Cock P, Molenaers G, et al. Upper limb motor and sensory impairments in children with hemiplegic cerebral palsy. Can they be measured reliably? Disabil Rehabil 2009; 32: 40916.
  • 23
    Taylor N, Sand P, Jebsen R. Evaluation of hand function in children. Arch Phys Med Rehabil 1973; 54: 12935.
  • 24
    Schulz K, Altman D, Moher D. CONSORT 2010 Statement: updated guidelines for reporting parallel group randomised trials. BMC Med 2010; 8: 18.
  • 25
    Liang KY, Zeger SL. Longitudinal data analysis using generalised linear models. Biometrika 1986; 73: 1322.
  • 26
    Wallen M, Ziviani J, Herbert R, Evans R, Novak I. Modified constraint-induced therapy for children with hemiplegic cerebral palsy: a feasibility study. Dev Neurorehabil 2009; 11: 12433.
  • 27
    Hoare BJ, Wasiak J, Imms C, Carey L. Constraint-induced movement therapy in the treatment of the upper limb in children with hemiplegic cerebral palsy. Cochrane Database Syst Rev 2007; (2): CD004149.
  • 28
    Gilmore R, Ziviani J, Sakzewski L, Shields N, Boyd R. A balancing act: children’s experience of modified constraint-induced movement therapy. Dev Neurorehabil 2010; 13: 8894.

Supporting Information

  1. Top of page
  2. Abstract
  3. What this paper adds
  4. Method
  5. Results
  6. Discussion
  7. Conclusion
  8. Acknowledgements
  9. References
  10. Supporting Information
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