Efficacy of Constraint-Induced Movement Therapy for Children With Cerebral Palsy With Asymmetric Motor Impairment Edward Taub,

Sharon Landesman Ramey, Stephanie DeLuca and Karen Echols Pediatrics 2004;113;305 DOI: 10.1542/peds.113.2.305

The online version of this article, along with updated information and services, is located on the World Wide Web at:
http://pediatrics.aappublications.org/content/113/2/305.full.html

PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly publication, it has been published continuously since 1948. PEDIATRICS is owned, published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois, 60007. Copyright 2004 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.

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Efficacy of Constraint-Induced Movement Therapy for Children With Cerebral Palsy With Asymmetric Motor Impairment
Edward Taub, PhD*; Sharon Landesman Ramey, PhD; Stephanie DeLuca, PhD*; and Karen Echols, PhD
ABSTRACT. Objective. Constraint-Induced Movement (CI) therapy has been found to be a promising treatment for substantially increasing the use of extremities affected by such neurologic injuries as stroke and traumatic brain injury in adults. The purpose of this study was to determine the applicability of this intervention to young children with cerebral palsy. Methods. A randomized, controlled clinical trial of pediatric CI therapy in which 18 children with diagnosed hemiparesis associated with cerebral palsy (796 months old) were randomly assigned to receive either pediatric CI therapy or conventional treatment. Pediatric CI therapy involved promoting increased use of the more-affected arm and hand by intensive training (using shaping) of the more-impaired upper extremity for 6 hours/ day for 21 consecutive days coupled with bivalved casting of the childs less-affected upper extremity for that period. Childrens functional upper-extremity skills were assessed in the laboratory (blinded scoring) and at home (parent ratings) just prior, after, and 3 weeks posttreatment. Treated children were followed for 6 months. Results. Children receiving pediatric CI therapy compared with controls acquired significantly more new classes of motoric skills (9.3 vs 2.2); demonstrated significant gains in the mean amount (2.1 vs 0.1) and quality (1.7 vs 0.3) of more-affected arm use at home; and in a laboratory motor function test displayed substantial improvement including increases in unprompted use of the more-affected upper extremity (52.1% vs 2.1% of items). Benefits were maintained over 6 months, with supplemental evidence of quality-of-life changes for many children. Conclusion. Pediatric CI therapy produced major and sustained improvement in motoric function in the young children with hemiparesis in the study. Pediatrics 2004; 113:305312; hemiparesis, cerebral palsy, pediatric rehabilitation, pediatric CI therapy, learned nonuse, upper extremity, CI therapy.
ABBREVIATIONS. CP, cerebral palsy; PT, physical therapy; OT, occupational therapy; CI, Constraint-Induced Movement; EIP, early-intervention program; EBS, Emerging Behaviors Scale; PMAL, Pediatric Motor Activity Log; TAUT, Toddler Arm Use Test; DASI-II, Developmental Activities Screening Inventory; ANCOVA, analysis of covariance; df, degrees of freedom; SD, standard deviation.
From the *Department of Psychology and Civitan International Research Center, University of Alabama, Birmingham, Alabama; and Center for Health and Education, Georgetown University, Washington, District of Columbia. Received for publication Aug 29, 2002; accepted May 28, 2003. Address correspondence to Edward Taub, PhD, Department of Psychology, University of Alabama, CPM 712, 1530 3rd Ave South, Birmingham, AL 35294-0018. E-mail: etaub@uab.edu PEDIATRICS (ISSN 0031 4005). Copyright 2004 by the American Academy of Pediatrics.

erebral palsy (CP), defined broadly as a nonprogressive motor impairment syndrome caused by a problem in the developing brain,1 affects at least 2 in 1000 children in the United States and 1 million children under the age of 21 in the industrialized world.25 Children with hemiparesis or substantially greater deficit in 1 upper extremity than the other comprise a significantly large group of those with CP.5 There is some question as to the efficacy of current physical therapy (PT) and occupational therapy (OT) treatment approaches to CP,1,6 8 especially with regard to transfer-of-treatment effect to the life situation. Until recently, few major theoretical or therapeutic advances were available to provide a basis for the development of new interventions for children with CP. In 1995, however, it was suggested that a promising new therapy for adults with hemiparesis consequent to stroke, known as Constraint-Induced Movement (CI) therapy,9 14 offered a potentially efficacious approach to the treatment of juvenile hemiparesis.15 The CI therapy protocol stems directly from basic research with monkeys (summarized in refs. 9 and 16). The studies reporting positive treatment effects for this intervention in humans include 2 with placebo control groups10,14 and replications in other laboratories.1719 The purpose of the present experiment was to carry out a randomized trial with a conventional physical rehabilitation control group to determine whether the full application of both components of the CI therapy protocol (extended, intensive training of the more-affected arm for many hours a day over a period of consecutive weeks and restraint of the less-affected arm for that period) would produce an improvement in motor function in young children with CP. A single case study has demonstrated positive changes in motor function of the more-involved arm.20
METHODS Subjects
Eighteen children were recruited from local-area early-intervention programs (EIPs), health care practitioners, or self-referrals. Eligibility criteria were a diagnosis of CP resulting in hemiparesis or substantially greater deficit in movement of 1 upper extremity in comparison to the other (2 subjects in each group; see Table 1), good health, 8 years old, and for children 18 months an etiology of stroke confirmed by magnetic resonance imaging findings. The University of Alabama Institutional Review Board approved the study protocol, and parents signed informed-consent statements. Children were assigned randomly to receive either pediatric CI therapy or conventional treatment. Randomness was achieved by assigning patients according to the group desig-

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TABLE 1.

Individual Subject Characteristics Subject* Age (mo) Gender More-Impaired Deficit LessImpaired at Deficit Mild None None Mild None Mild None None None None None None Moderate None None Moderate None None Type of CP Treatment Seizure DASI-II Score Services at Enrollment (hour/wk) 2 PT-OT 2 PT-OT 2 EIP 5 EIP 10 EIP 2 PT-OT 1 OT 2 PT-OT 1 PT-OT 2 1 2 2 2 2 2 PT-OT PT PT-OT PT-OT EIP PT-OT PT-OT

Pediatric CI Therapy Group CHOP ORMA SEIE KSAN INEY ELDY NSER AMUS DEEL Mean Conventional Services Group VEON AHYA ERLE KIER RSNA ADAN TOON IAIE WNEY Mean

7 10 18 22 32 50 53 74 85 39.0 14 16 22 33 36 43 45 86 96 43.4

Male Female Female Male Male Male Male Male Male Male Female Male Male Female Male Male Female Male

Severe Severe Mild Moderate Moderate Moderate Severe Severe Moderate-severe Moderate Moderate-severe Moderate Severe Mild Moderate Severe Severe Moderate

Flaccid Spastic Spastic Spastic Spastic Spastic Flaccid Spastic Spastic Spastic Spastic Spastic Spastic Spastic Spastic Spastic Spastic Spastic

Yes Yes

71 78 124 117 81 107 76 142 110 88 112 88 41

Yes Yes Yes Yes

1 PT-OT

* Initials of children represent a transformation of identifications and do not identify the childrens actual names. Categories of mild, moderate, and severe are based on review of clinical records and rating by clinicians (detailed data about skill level were collected independently). Uncooperative/refusal to participate. Too old for test. nation indicated on a folded piece of paper, taped closed, and drawn from a jar set up before the beginning of subject enrollment.

Assessments
Children were pretested after randomization into 1 of the above groups then posttested after treatment or an equivalent time period for controls, with follow-up testing 3 weeks later. In addition, the pediatric CI therapy group received 3- and 6-month follow-up testing. Measures included the Emerging Behaviors Scale (EBS), the Pediatric Motor Activity Log (PMAL), and blinded ratings of videotaped sessions of the Toddler Arm Use Test (TAUT). The EBS is a tool developed here to provide quantification for the observation in pilot work that children receiving pediatric CI therapy rapidly acquired entirely new, age-appropriate behaviors. The EBS provides systematic (rather than anecdotal) information about young childrens neuromotor repertoire, particularly regarding the onset of newly acquired behaviors of importance in young childrens lives that had not been observed before treatment by the principal caregiver, the therapist, or other sources (eg, childs previous provider of physical rehabilitation services). The presence or absence of 31 motor patterns and functional activities are recorded, such as reaching, prehension of different types, carrying objects, releasing objects, individual finger use, weight bearing of different types, position transfer (eg, lie to sit, sit to crawl, and crawl to stand), mobility involving arm use, and hand gestures. Determination of the occurrence of the new classes of movement had to come from at least 2 of the following sources: a project therapist as recorded in the daily notes, the principal caregiver, a videotape of a treatment session, or occurrence and record of activity at a posttreatment testing session. The EBS score is the number of new behaviors that emerged after the beginning of treatment or the equivalent time point in control subjects. The PMAL is a semistructured interview administered every other day to a childs principal caregiver, which in the case of all subjects in this experiment was the mother. It obtains systematic data about 22 distinct arm-hand functional activities typical of young children. It is an adaptation of the Motor Activity Log developed for adult patients with stroke.10 The adult Motor Activity Log is psychometrically robust, yielding scores that remain stable during a 2-week period when either a placebo treatment14 or no treatment17 was administered. It has high internal consistency (Cronbachs .88 to .95), interrater reliability (patient compared with primary caregiver, Intra Class Correlation type 3, 1 90), and high test-retest reliability (r .94; P .01).14,17 The TAUT is a standardized laboratory motor test in which a series of 22 tasks/play activities are presented before treatment

Treatment
The pediatric CI therapy involved 2 components. First, the childs less-impaired upper extremity was casted from upper arm to fingertips by using a lightweight fiberglass cast. The cast was bivalved to enable easy weekly removal to check skin integrity and allow range of motion. Second, the day after casting, the pediatric CI therapy training procedures (by a process known as shaping) began.11,16,21 Occupational therapists, physical therapists, or a PT assistant provided 6 hours of therapy per day for the next 21 days. Shaping involved presenting interesting and useful activities to the child in ways that provided immediate, frequent, and repetitive rewards (primarily verbal praise, smiles, and supportive gestures, with some food and toys) for the childs efforts and increasingly functional use of the more-impaired extremity. Tasks such as reaching, grasping, holding, manipulating an object, bearing weight on the arm, and making hand gestures were divided into their small component skills, which were worked on individually and later chained together to comprise a target activity. When the child demonstrated a new movement skill, the therapist proceeded to shape this by increasing the demands for more precision, strength, fluency, automaticity, and/or functional versatility. The pediatric CI therapist also incorporated everyday tasks (eg, dressing/undressing, eating, bathing, and grooming) in the therapy sessions. Shaping tasks were selected by considering 1) the family and childs goals, 2) the intrinsic motivating properties of an activity, 3) promotion of independence by acquisition of age-appropriate self-help skills, and 4) the movements that therapists believed had the greatest potential for improvement.11 Parents were encouraged to join in therapy-related activities and encourage their child to use newly acquired skills when the therapist was not present. When a child showed signs of fatigue, frustration, or reduced interest, the therapist adapted the activities but did not cease the therapy. Rest intervals were given as needed. In adults, intensive training is by far the more important component of the treatment procedure.14 One of us who had been involved in the development from its inception of the use of the shaping technique for neurologically impaired adults participated in the training and twice weekly in supervision of the therapists. The children in the control group continued their participation in previously established EIPs, which included receiving conventional PT and/or OT for a mean of 2.2 hours per week.

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and after the less-impaired arm cast is removed after treatment. The examiner tries to elicit the childs best effort to secure or activate a toy or perform a given task. If the child does not use the more-impaired arm or hand at first, then the child is asked to try the task with that arm/hand, restraining the less-impaired upper extremity if necessary. Videotapes of these sessions were scored independently by 2 experienced pediatric occupational therapists (interrater reliability .98) who were blind to the treatment group and pre- or posttreatment status of the children. A list of test activities, the scoring format, and the scoring scales for both the PMAL and TAUT are contained in Appendices 1 and 2, respectively. The Developmental Activities Screening Inventory (DASI-II) is an educational screening tool designed to help identify children at risk for educational delay at an early age.22

TABLE 2. EBS Means and SD for CI and Conventional Therapy Groups at Pre- and Posttreatment Time of Assessment Classes of Behavior Exhibited (No.) Pediatric CI Therapy Mean (SD) 12.2 (5.64) 21.5 (4.45) 9.3 Control (Conventional Therapy) Mean (SD) 12.7 (6.51) 15 (5.66) 2.3

Pretreatment Posttreatment Change*

* Increase in number of motor patterns and classes of behavior exhibited from pre- to posttreatment is designated the emerging behaviors score. TABLE 3. PMAL Means, SD, and Change Scores for CI and Conventional Therapy Groups at Pre- and Posttreatment and 3-Week Follow-Up Time of Assessment Pediatric CI Therapy Mean (SD) Amount of use (05) Pretreatment Posttreatment 3-wk follow-up 3-mo follow-up 6-mo follow-up Quality of use (05) Pretreatment Posttreatment 3-wk follow-up 3-mo follow-up 6-mo follow-up 0.8 (0.44) 2.8 (1.14) 2.6 (1.29) 2.1 (1.33) 2.4 (1.34) 0.9 (0.62) 2.7 (0.97) 2.6 (1.25) 2.6 (1.36) 2.7 (1.01) Change Control (Conventional Therapy) Mean (SD) 1.1 (0.75) 1.2 (0.82) 1.2 (0.67) Change

RESULTS

The children in the pediatric CI therapy and control groups did not differ significantly in terms of age, gender, severity of hemiparesis, or prior treatment history. The data are presented in Table 1. Childrens ages ranged from 7 months to 8 years, with a mean of 41.5 months.
Adjustment to Long Arm Cast

During the casting session, children showed some temporary upset, particularly when the cast saw was used to bivalve the just-applied cast into 2 parts. Within 1 to 2 days, however, all children had adjusted very well to the presence of the cast. During the weekly removal of the cast, 4 of the 9 children had some mild skin redness, rash, or pinching, which was treated effectively with Neosporin and/or BandAids and the insertion of additional cotton wrap when reapplying the cast. No children resisted cast reapplication, and several specifically asked to have it put back on their arm.
Upper-Extremity Movement

2.0 1.8 1.3 1.6 1.8 1.7 1.7 1.8

0.1 0.1

1.6 (1.20) 1.9 (1.13) 1.8 (1.01)

0.3 0.2

Before analysis of posttreatment data, 1-way analyses of variance were conducted on pretreatment scores. These tests indicated that there were no preexisting differences between treatment groups on any of the measures. On the EBS, the children in the pediatric CI therapy group exhibited a mean of 9.3 new motor patterns and classes of functional activity that they had never been observed to perform previously. This was greater by a factor of 4.3 than the 2.2 new motor behaviors the children in the control group acquired during the treatment period (Table 2). A repeatedmeasures analysis of covariance (ANCOVA) with pretreatment score as the covariate revealed a significant effect of treatment (F 84.59; degrees of freedom [df] 1; P .0001). The distribution of individual child scores between groups was nonoverlapping, with a range of 7 to 12 newly acquired skills for CI therapy children and 0 to 6 for conventional treatment controls. In the pediatric CI therapy group, 5 of 9 children gained 10 new upper-extremity skills; 5 of 9 control children gained 2. The PMAL scores (Table 3) for pretreatment and 3 weeks later (follow-up) were analyzed using a repeated measures, multivariate ANCOVA for the 2 scales used (Amount of Use and Quality of Use) with initial level of deficit (ie, pretreatment score) serving

as the covariate. The overall multivariate ANCOVA showed a significant effect for the treatment time interaction (F 12.54; df 4; P .0001). Univariate ANCOVAs confirmed that there were significant gains on both scales for children receiving pediatric CI therapy compared with the control children: mean gains of 2.1 and 1.7 from pre- to posttreatment for Amount of Use and Quality of Use, respectively, compared with 0.1 (F 43.02; df 2; P .0001) and 0.3 (F 34.54; df 2; P .0001) for controls. Children given pediatric CI therapy maintained these gains over the next 3 weeks. There were no differences in outcome based on gender or on the presence of spasticity, which was never more than moderate in this hemiparetic sample. There was a suggestion that children with low DASI-II scores did not improve as much as a result of pediatric CI therapy as children who scored above the age norm. DASI-II scores could not be obtained from 3 subjects in the CI therapy group, 2 because they exceeded the 60-months age limit for which this test is valid and 1 because of lack of cooperation. Of the 6 remaining subjects in the CI therapy group, 3 scored 1 standard deviation (SD) below the age norm, whereas 3 scored above the age norm. For the 6 PMAL scores (3 Amount of Use scores and 3 Quality of Use scores) in each subgroup, there was 1 tie score, and otherwise the above-norm subjects exhibited more improvement than the below-norm subjects, with no other overlap between them in scores. The nonparametric Mann-Whitney U test revealed
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that this difference was of borderline significance for the Amount of Use Scale (P .05) and had a trend toward significance for the Quality of Use Scale (P .075). However, the children with low DASI-II scores still showed a significant and clinically meaningful benefit from the intervention (paired t test: P .05 on both scales). Childrens performance on the TAUT revealed large and statistically significant between-group differences after treatment in percent of items for which children used their more-impaired upper extremity without prompts, percent of items scored as successfully accomplished through use of the more-impaired arm, and overall quality of use of that extremity. Within each group, children receiving pediatric CI therapy increased first-time use of their moreimpaired upper extremity from pre- to posttreatment by a mean of 53.9% (SD 35.64), whereas controls increased by a mean of 18.00% (SD 31.12). In addition, blinded ratings of overall independent functional use of the more-impaired arm showed a mean gain for the pediatric CI therapy group of 16.8% (SD 21.53) compared with 5.0% (SD 15.4) for the control group.
Follow-Up

Children in the pediatric CI therapy group maintained their treatment gains at 3 and 6 months follow-up. The childrens PMAL Amount of Use gain scores had means (SDs) of 1.3 (1.11) at 3 months and 1.6 (1.23) at 6 months, indicating a small but not statistically significant decline from 3-week posttreatment performance change (1.8). Quality of Movement means (SDs) were 1.7 (.83) and 1.8 (.61) at 3 and 6 months, respectively, essentially the same as at 3-week posttreatment (1.7 [.97]).
DISCUSSION

In this controlled treatment trial, upper-extremity CI therapy, which had been used successfully with adult patients with stroke,9 19 was adapted for use with young children with hemiparesis associated with CP; modifications were made to render the treatment developmentally appropriate. The intervention produced a large improvement in the use of the more-affected extremity, at least as great as that obtained with adults. It may well have been greater, because the children exhibited a mean of 9.3 new motor behaviors and patterns of functional behavior that had not been observed before the relatively brief, 3-week therapy period. Further, gains on the PMAL were sustained over a 6-month follow-up period, with parental reports of important developmental and social-emotional benefits for the children. Control subjects showed no improvement on the PMAL from pre- to posttreatment; they did exhibit a modest improvement on the EBS, but it was not significant. Verbal descriptions by parents of major changes in quality of life and general development confirmed the more formal quantification of parental reports obtained on the PMAL. Several children began to crawl soon after treatment ended. One child who previously showed no awareness of his more-im308

paired extremity made large gains in all areas of development. He began to use all the muscles of his more-impaired side in coordinated fashion, started commando crawling, pivoted in prone, and pushed up on extended arms. Two children began to sit independently. A 412-year-boy who had never used his more-affected arm or hand began to play ball and, in 6 months, realized his desires of being able to fish with his father and join a Little League Baseball team, batting with both hands and using a specially adapted glove on his more-impaired hand to keep it in place. An unanticipated change many parents reported was an increase in their childrens social and communication skills, often accompanied by signs of increased confidence and enjoyment in daily activities. Two of the pediatric CI therapy subjects were 7 and 10 months of age, respectively. There can be some question concerning the accuracy of the diagnosis of CP at those ages. However, both children had experienced severe prenatal strokes, as revealed by marked clinical symptoms and confirmed by magnetic resonance imaging findings. Three randomized, controlled trials testing the efficacy of widely used, professionally endorsed forms of physical rehabilitation failed to report significant benefits.23,24 Two other randomized trials showed possible and/or small-magnitude benefits,25,26 but methodological limitations render interpretation of their results problematic. In our study, treated children showed large and significant gains in both quality and amount of use of the more-impaired extremity. Our results also differ markedly from those of 4 studies in which restraint of the less-affected upper extremity alone was used for varying periods in children with hemiparesis without the intensive training component of pediatric CI therapy.2730 Restraint alone was found to result in a modest to weak treatment effect that is only a portion of what was obtained in this study when both the restraint plus the training components of pediatric CI therapy were used. Although restraint alone is becoming increasingly popular, it would seem to be of limited value when not coupled with active training, as by a shaping technique. This is similar to the situation with adult patients with stroke in which restraint alone produces just 20% of the treatment effect obtained with the full protocol.14 The main therapeutic factor underlying the clinical efficacy of CI therapy is the concentrated extended nature of the training conducted for many hours daily over a period of consecutive weeks.12,14 A literature search did not reveal any report of a conventional pediatric (or adult) PT training procedure conducted at this level of intensity. Because research has shown that this is the critical parameter, it is likely that some conventional interventions would produce an enhanced effect with this schedule of delivery of services. This has been shown to be the case with adults for whom conventional PT procedures (combined neurotherapeutic facilitation, task practice, and aquatic therapy) given 5 hours/day for 2 weeks produced a very good clinical effect. However, this intense and atypical schedule of delivery of services

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would result in a therapy possessing the most important defining characteristic of CI therapy (ie, concentrated practice for an extended period).12,14 It is, of course, still possible that other new methodological characteristics introduced in CI therapy might confer an advantage on it in terms of treatment outcome. Similar considerations arise with respect to the issue of whether the treatment effect observed here is merely the result of time spent in treatment and not the treatment itself. This issue was addressed in a study with adult patients with stroke from this institution in which it was found that a general fitness placebo control group shown by pretreatment questionnaire to be credible and involving the same amount of subject/therapist interaction as in a CI therapy group failed to show a meaningful treatment effect.12,14 There is little reason to believe that the situation would be different for children. It would be of value to examine both the question of nonspecific effects from pediatric CI therapy and the dose-response effect of the intensity of treatment in future research. In adults, the efficacy of CI therapy is predicated on 2 linked but independent mechanisms: use-dependent cortical reorganization and overcoming learned nonuse. After stroke the cortical-representation zone of the more-affected upper extremity in the primary motor cortex contracts to approximately one-half the size of the motor-representation area in the less-affected hemisphere.31,32 CI therapy results in a doubling in the size of the cortical representation zone of the more-affected limb, which has the effect of equalizing the size of the motor output areas on both sides of the brain,31 as well as recruiting ipsilateral motor cortex into playing a major role in the innervation of the movements of the more-affected arm.33 As noted, the common factor in all forms of CI therapy, including its use with young children in this study, seems to be repeatedly practicing use of the more-affected upper extremity in concentrated fashion for many hours a day for a period of consecutive weeks. This factor is presumed likely to produce the large use-dependent cortical reorganization that has been observed and the long-term persistence of the CI therapy effect. It should be noted that this usedependent brain plasticity effect provides support for the belief that the intensity of treatment, which increases the frequency of use of the more-affected arm, is a significant factor in producing the therapeutic result observed here. It also provides indirect support that the results observed here were not due to a placebo effect. The learned nonuse mechanism, the experimental support of which is described in detail elsewhere,9,14,16 comes into operation as a result of the fact that substantial neurologic injury usually leads to depression in motor function that is considerably greater than will be the case after spontaneous recovery of function has taken place. The depressed responsivity of motoneurons leads to a reduced ability to move the more-affected limb in the early postinjury period. During the period of depressed neural function, the individual is either unable to move the

more-affected limb or make more than clumsy, inefficient movements. The resulting motor failure results in suppression of future attempts to use the more-affected limb. At the same time, the individual learns to compensate by using only the uninvolved limb for most purposes. These efforts are at least partially effective, and the individual is consequently rewarded by this success. The result is that the individual learns not to try to use the more-affected limb, the use of which is then held in powerful inhibition. When appropriate techniques are applied, however, learned nonuse can be overcome. Two such techniques involve intensive training of the moreaffected limb and restriction of the less-affected limb. Both procedures require use of the more-affected limb for normal activities, or the individual is either rendered virtually helpless or unable to carry out the required behavioral task. In children who sustain central nervous system injury in the prenatal, perinatal, or early-postnatal periods, the situation differs in some ways. For these children, different classes of behavior fail to develop entirely. Thus, most of these children do not have the experience of performing motor activities that are later lost after a central nervous system injury, as in the case of adults after stroke. One might, therefore, more appropriately refer to developmental disregard in these children. However, the same mechanisms of punishment of ineffective, clumsy attempts to use the more-affected extremity and reward for at least partially successful use of the less-affected extremity alone are presumed to operate in young children as they do in individuals with a mature motor repertoire. Developmental disregard might then be viewed as a special case of learned nonuse. In almost all the research using CI therapy with adults, an exclusion factor has been substantial cognitive deficit as indicated by a score of 24 on the Mini-Mental State Examination. Relatively few adult patients with stroke had to be excluded on this basis, and, within the limited range studied, no correlation was observed between score on the Mini-Mental State Examination and CI therapy outcome. In this study, children scoring 1 SD below the age norm on the DASI-II exhibited a significant treatment effect, but there was a trend for it to be less large than that for the above-norm subjects. However, because of the very small sample size (3 in each of the subgroups), one can consider this difference suggestive only, but a possible guide for future work. CP in infants and children not only involves delays or permanent deficits in neuromotor function but also has a profound effect on other functional domains, including sensory awareness and responsiveness, and type and level of engagement with the physical and social environment. The consequences of major motor disabilities are profound for all aspects of a childs quality of life.1,8 It would be of value to ascertain more systematically than we did the extent to which pediatric CI therapy also contributes to changes in other developmental domains, presumably by introducing new motor capabilities into the childs behavioral repertoire and allowing the child to experience rapid gains. The parental
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reports and videotaped sessions indicate that many children receiving pediatric CI therapy increased their self-confidence, interacted more with their environment, and demonstrated new sensory awareness of the hemiparetic extremity. It remains for future research to evaluate these reports objectively. An additional objective of future research would be to determine whether the results reported here can be replicated in other laboratories. It would also be important to determine whether casting the lessaffected arm for 3 weeks results in any short-term or long-term loss of function in that extremity and, additionally, what the dose-response relationship is between treatment outcome and amount of both lessaffected limb restraint and more-affected limb training. A major concern in the practitioner community is that there are strong pressures to recommend only the amount (intensity) of rehabilitation therapy for which health care payers are currently willing to reimburse. However, virtually all interventions for young children in which treatment is given on the present attenuated basis remained untested and unproven.34 In contrast, the data presented from this randomized, controlled trial of CI therapy indicate that a more-intensive schedule of delivery of pediatric rehabilitation services than is given conventionally can produce large, sustained gains. As in clinical research with adults, CI therapy in young children with hemiparesis leads to rapid and large changes in motoric function.
APPENDIX I: PMAL

How-Often Scale
0.0Never used weaker arm when the task was attempted. 0.5 1.0Seldom used weaker arm when the task was attempted. 1.5 2.0Used weaker arm 25% of times when the task was attempted. 2.5 3.0Used weaker arm on half of the occasions the task was attempted. 3.5 4.0Used weaker arm on most of the occasions the task was attempted. 4.5 5.0Used weaker arm on almost every occasion that the task was attempted.

How-Well Scale
0.0Weaker arm was not used at all for that activity (no use). 0.5 1.0Weaker arm was moved during that activity but was not helpful (very poor). 1.5 2.0Weaker arm was of some use during that activity but needed some help from the stronger arm or moved very slowly or with difficulty (poor). 2.5 3.0Weaker arm was used for that activity, but movements were slow or were made with only some effort (fair). 3.5 4.0Movements made by weaker arm for that activity were almost typical for age but not quite as fast or accurate (almost normal). 4.6 5.0Ability to use the weaker arm for that activity was typical for age (normal).

The following is a list of activities and the scoring format for use of the more-affected arm in different activities in the life situation. (Note that a revised version of this test is currently in use.)

APPENDIX II: CHILD ARM USE TEST Laboratory Motor Test

How Often 1. Hold a bottle/cup 2. Pick up and hold a small item while sitting in a chair 3. Pick up and hold a large item while sitting in a chair 4. Eat finger foods 5. Pick up an object out of arms reach 6. Push a button 7. Open a door or cabinet 8. Use arm to move across floor 9. Take off shoes or socks 10. Pull a toy with a string 11. Turn a knob 12. Pick up a cylindrical object (eg, crayon, marker, or drumstick) 13. Throw a ball or similar object 14. Hold a handle on a riding, pulling, or push toy 15. Push up front of body with weaker arm while on stomach 16. Hold an item while in standing position 17. Carry an item from place to place 18. Stop or roll a ball 19. Pop bubbles 20. Push into sitting position 21. Reach to be picked up by parent 22. Push arm through sleeve of clothing

How Well

Test objects are placed in front of the subject (tasks 110) or affixed with Velcro to a vertical board facing the subject (tasks 1121). Test performance was videotaped and rated independently by 2 blinded, experienced clinicians having a high interrater reliability ( .95).
Scoring Instructions

(Rated from videotape; raters were blinded to group and pre- or posttreatment status.)
Arm Selection

On unforced first attempt of each activity (both arms available for use), which arm was chosen? R Right arm L Left Arm Use the following scales to rate the childs best effort with the more-involved arm.
Amount of Participation

0 1 2

Child does not attempt to use the more-involved arm. Child moves more-involved arm during task, but it does not contribute to task completion. Child uses more-involved arm to carry out the task regardless of whether the task was performed in an age-typical manner.

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Hand Used (Right/Left) 1. 2. 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13. 14. 15. 16. 17. 18. 19. 20. 21. Strike a toy with hammer Use markers for scribbling Pick up snack Pull lever Push button Turn knob Push top Remove puzzle pieces with large knob Remove puzzle pieces with small knob Squeeze horn Remove ball Place or throw ball Remove phone (vertical orientation) Remove phone (horizontal orientation) Unclip clothes pin (weak spring) from the edge of vertical surface Unclip clothes pin (moderate spring) from edge of vertical surface Unclip clothes pin (strong spring) from edge of vertical surface Unclip clothes pin (weak spring) from edge of horizontal surface Unclip clothes pin (moderate spring) from edge of horizontal surface Unclip clothes pin (strong spring) from edge of horizontal surface Release clothes pin

Amount of Participation

How Well

Willingness

Global Rating

How-Well Scale

REFERENCES
1. Pellegrino L. Cerebral palsy. In: Batshaw ML, Ed. When Your Child Has a Disability: The Complete Sourcebook of Daily and Medical Care. Baltimore, MD: Paul H. Brookes Publishing Co; 2000:275287 2. Batshaw ML, Periet YM. Children with Handicaps: A Medical Primer. Baltimore, MD: Paul H. Brookes Publishing Co; 1996 3. Campbell SK. Introduction to the special issue. Pediatr Phys Ther. 1990; 2:123125 4. Capute A, Accardo P. Developmental Disabilities in Infancy and Childhood. Baltimore, MD: Paul H. Brookes Publishing Co; 1990 5. Hagberg B, Hagberg G, Beckung E, Uvebrandt P. Changing panorama of cerebral palsy in Sweden. VII. Prevalence and origin in the birth period 199194. Acta Paediatr. 2001;90:271277 6. Bower E, McClellan DL. Evaluating therapy in cerebral palsy. Child Care Health Dev. 1994;20:409 419 7. Guralnick M, Ed. The Effectiveness of Early Intervention: Second Generation Research. Baltimore, MD: Paul H. Brookes Publishing Co; 1997 8. Kurtz, LA. Understanding rehabilitation therapies. In: Batshaw ML, Ed. When Your Child Has a Disability: The Complete Sourcebook of Daily and Medical Care. Baltimore, MD: Paul H. Brookes Publishing Co; 2000: 109 122 9. Taub E. Somatosensory deafferentation research with monkeys: implications for rehabilitation medicine. In: Ince LP, Ed. Behavioral Psychology in Rehabilitation Medicine: Clinical Applications. New York, NY: Williams & Wilkins; 1980:371 401 10. Taub E, Miller NE, Novack TA, et al. Technique to improve chronic motor deficit after stroke. Arch Phys Med Rehabil. 1993;74:347354 11. Taub E, Crago J, Burgio L, et al. An operant approach to overcoming learned nonuse after CNS damage in monkeys and man: the role of shaping. J Exp Anal Behav. 1994;61:281293 12. Taub E, Pidikiti RD, DeLuca SC, Crago JE. Effects of motor restriction of an unimpaired upper extremity and training on improving functional tasks and altering brain/behaviors. In: Toole J, Ed. Imaging and Neurologic Rehabilitation. New York, NY: Demos; 1996:133154 13. Morris DM, Crago JE, DeLuca SC, Pidikiti RD, Taub E. ConstraintInduced (CI) Movement therapy for motor recovery after stroke. Neurorehabilitation. 1997;9:29 43 14. Taub E, Uswatte G, Pidikiti R. Constraint-Induced Movement therapy: a new family of techniques with broad application to physical rehabilitationa clinical review. J Rehabil Res Dev. 1999;36:237251 15. Taub E, Crago J. Behavioral plasticity following central nervous system damage in monkeys and man. In: Julesz B, Kovacs I, Eds. Maturational Windows in Adult Cortical Plasticity. SFI Studies in the Sciences of Complexity. Vol. 23. Redwood City, CA: Addison-Wesley; 1995:201215 16. Taub E. Movement in nonhuman primates deprived of somatosensory

(Same as for PMAL)

Willingness Scale

0. 1. 2. 3.

Never attempted activity with more-involved arm. Considerable resistance: pulled away or took an excessive amount of time to attempt with more-involved arm. Some resistance: required a few prompts (tapping or cueing the more-involved arm) or needed less-involved arm restrained. No resistance: attempted activity with moreinvolved arm with minimal prompting.

Global Rating

Rate your overall reaction to the childs ability to utilize the more-involved arm, with 0 being no ability and 10 being age-typical ability. ------------------------------------------------------------------0 1 2 3 4 5 6 7 8 9 10

ACKNOWLEDGMENTS
Grant support for this research was provided by the Alabama Health Services Foundation, the Civitan International Research Center, the National Institute of Child Health and Human Development of the National Institutes of Health, the Administration on Developmental Disabilities, and the Maternal and Child Health Bureau. We are grateful to Mary-Rebekah Trucks, Bonnie Rose, Anna Hart, Tina Mull, and Sonya Pearson, who provided the pediatric CI therapy to the children; Dr Laura Vogtle, Jan Rowe, and Pam Bruner, who conducted the expert testing and rating of children; Dr Charles Law for casting procedures; and Dr Alan Percy for careful reading of the manuscript. We are also appreciative of the assistance of Shobrena Shamburger and Angela Matthews with manuscript preparation, and we thank the Service Guild Early Intervention Program of Birmingham for providing testing and treatment space.

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ARTICLES

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GLUE BOYS

In Nairobi, orphaned boys wander the streets barefoot and dirty, many sniffing glue. Crusted white layers are stuck to their nostrils. They are known across the continent as glue boys, and some fall into committing petty crimes like stealing cell phones and wallets, aid workers report, mostly because they have no other way to survive.

Wax E. A generation orphaned by AIDS. Washington Post Weekly. October 27November 2, 2003

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EFFICACY OF CI THERAPY FOR CHILDREN WITH CP Downloaded from pediatrics.aappublications.org by guest on August 21, 2011

Efficacy of Constraint-Induced Movement Therapy for Children With Cerebral Palsy With Asymmetric Motor Impairment Edward Taub, Sharon Landesman Ramey, Stephanie DeLuca and Karen Echols Pediatrics 2004;113;305 DOI: 10.1542/peds.113.2.305
Updated Information & Services References including high resolution figures, can be found at: http://pediatrics.aappublications.org/content/113/2/305.full.ht ml This article cites 21 articles, 4 of which can be accessed free at: http://pediatrics.aappublications.org/content/113/2/305.full.ht ml#ref-list-1 This article has been cited by 26 HighWire-hosted articles: http://pediatrics.aappublications.org/content/113/2/305.full.ht ml#related-urls This article, along with others on similar topics, appears in the following collection(s): Neurology & Psychiatry http://pediatrics.aappublications.org/cgi/collection/neurology _and_psychiatry Information about reproducing this article in parts (figures, tables) or in its entirety can be found online at: http://pediatrics.aappublications.org/site/misc/Permissions.xht ml Information about ordering reprints can be found online: http://pediatrics.aappublications.org/site/misc/reprints.xhtml

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PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly publication, it has been published continuously since 1948. PEDIATRICS is owned, published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois, 60007. Copyright 2004 by the American Academy of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.

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