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Methods of constraint-induced movement therapy for children with hemiplegic cerebral palsy: Development of a child-friendly intervention for improving upper-extremity function

      Abstract

      Gordon AM, Charles J, Wolf SL. Methods of constraint-induced movement therapy for children with hemiplegic cerebral palsy: development of a child-friendly intervention for improving upper-extremity function. Arch Phys Med Rehabil 2005;86:837-44.
      We delineate the methodology for constraint-induced movement therapy (CIMT) modified for children with hemiplegic cerebral palsy (CP) and describe important considerations that need to be made when testing this intervention in children. The resulting intervention evolved from piloting and testing it with 38 children with hemiplegic CP who were between the ages of 4 and 14 years. Thirty-seven successfully completed the treatment protocol. The intervention retains the 2 major elements of the adult CIMT (repetitive practice, shaping) and was constructed to be as child-friendly as possible. It involves restraining the noninvolved extremity with a sling and having the child engage in unimanual activities with the involved extremity 6 hours a day for 10 days (60h). Specific activities are selected by considering joint movements with pronounced deficits and improvement of which interventionists believe have greatest potential. The activities are chosen to elicit repetitive practice and shaping. The intervention is conducted in groups of 2 to 3 children to provide social interaction, modeling, and encouragement. Each child is assigned to an interventionist to maintain at least a 1:1 ratio. CIMT can be modified to be child-friendly while maintaining all practice elements of the adult CIMT. The modified therapy is tolerated by most children. Further modifications will likely be required to hone in on the specific components of the intervention that are most effective before applying them to children who are most likely to benefit.

      Key words

      CEREBRAL PALSY (CP) is a neurodevelopmental disorder caused by nonprogressive lesion(s) in the immature brain. The early central nervous system (CNS) damage results in chronic physical disabilities and often includes sensory impairments. The prevalence of congenital CP is approximately 2 per 1000 births, with hemiplegia accounting for approximately 25% of all new cases worldwide.
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      The cost of health care associated with CP in the United States is approximately $800,000 (2000 dollars) per person over their lifespan.
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      For people with hemiplegia, characterized by unilateral upper- and lower-extremity involvement, impaired manual dexterity is often among the most disabling motor symptoms. Treatment options include physical therapy (PT), occupational therapy (OT), conductive education, neurodevelopmental therapy, peripheral splinting and casting, pharmacotherapy (eg, botulinum toxin type A), and surgery. There is no strong evidence of successful treatment with of any of these approaches.
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      Management of upper limb dysfunction in children with cerebral palsy a systematic review.
      Recent evidence suggests that children with CP may improve motor performance if provided with sufficient opportunities to practice.
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      One treatment approach that provides those opportunities and that is becoming increasingly popular is forced use or constraint-induced movement therapy (CIMT). Forced use facilitates practice with the involved extremity by restraining the noninvolved extremity; it can be used alone or along with conventional PT and OT.
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      Forced use of hemiplegic upper extremities to reverse the effect of learned nonuse among chronic stroke and head-injured patients.
      In contrast, CIMT involves intensive targeted practice with the involved extremity along with the restraint.
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      • et al.
      Technique to improve chronic motor deficit after stroke.
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      The latter is an active, task-driven treatment administered by a trained practitioner that combines principles from the fields of behavioral psychology and motor learning.
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      Methods for a multisite randomized trial to investigate the effect of constraint-induced movement therapy in improving upper extremity function among adults recovering from a cerebrovascular stroke.
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      The intervention originated from the observation that unilaterally deafferented monkeys normally do not make use of the deafferented limb,
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      but can be induced to do so by restricting movement of the intact limb,
      and they will continue to use the limb even when the movement restriction ceases.
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      Deafferentation in monkeys pointing at a target without visual feedback.
      The procedure was first used in human patients with hemiparetic stroke, and much of the published research has focused on that population.
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      Repetitive task practice a critical review of constraint-induced movement therapy in stroke.
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      Constraint-induced movement therapy bridging from the primate laboratory to the stroke rehabilitation laboratory.
      The results of these studies have been promising, and currently there is a national randomized control led trial (RCT) (Extremity Constraint-Induced Therapy Evaluation [EXCITE]) under way to determine the efficacy of CIMT in patients with subacute or chronic stroke.
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      • Miller J.P.
      • Blanton S.
      • et al.
      Methods for a multisite randomized trial to investigate the effect of constraint-induced movement therapy in improving upper extremity function among adults recovering from a cerebrovascular stroke.
      Recently, both parent/caregivers and clinicians have been eager to incorporate this CIMT into the treatment regimen of children with CP, despite a lack of data adequate to support its effectiveness in this population. The increased plasticity in the developing nervous system may result in an even better outcome for children with hemiplegia. However, to date, forced use and CIMT have only been used in case studies or small-scale studies.
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      Upper extremity casting adjunct treatment for a child with cerebral palsy hemiplegia.
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      The effects of constraint induced therapy on hand function in children with hemiplegic cerebral palsy.
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      Clinical experience of constraint induced movement therapy in adolescents with hemiplegic cerebral palsy—a day camp model.
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      Pediatric constraint-induced movement therapy for a young child with cerebral palsy two episodes of care.
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      Efficacy of constraint-induced movement therapy for children with cerebral palsy with asymmetric motor impairment.
      Despite its promise, there appears to be considerable confusion, as reflected in both the literature and the field, because the term “constraint-induced movement therapy” is often incorrectly used synonymously with “forced use,” and there is no agreed-on protocol.
      CIMT as developed for adults, however, is an intensive intervention that, in its original form, is not child-friendly and is potentially invasive. Specifically, it requires restraining the noninvolved hand of a patient 90% of his/her waking hours for 14 consecutive days. Furthermore, for 10 of the 14 days, patients engage in 6 hours of intensive programming. During this time, they use their involved upper extremity in numerous activities that are characterized by 2 types of practice: repetitive task practice and shaping.
      Repetitive task practice is the performance of functional tasks continuously for 15 to 20 minutes. It is targeted movement that is embedded in a functional activity. This approach allows practice of a movement in the context of the preceding and following movement and a functional task. The environment can be manipulated to vary the task requirements, elicit specific movements, or grade the difficulty; it is always monitored so that frequent successes can be achieved. Shaping involves a motor task goal being approached in small steps by successive approximation, and/or grading of the task difficulty based on the patient’s capabilities.
      • Taub E.
      • Skinner B.F.
      • Morgan W.G.
      The shaping game a teaching technique.
      • Panyan M.V.
      Shaping is similar to adaptive or part-practice that is documented in the motor learning literature.
      • Mane A.M.
      • Adams J.A.
      • Donchin E.
      Adaptive and part-whole training in the acquisition of a complex perceptual-motor skill.
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      • Lee T.D.
      Both repetitive task practice and shaping involve adult-oriented, monotonous tasks (eg, screwing and unscrewing bolts, adult-appropriate functional tasks) that would not likely hold a child’s interests for the long period of engagement required. Thus, the types of practice used in adult CIMT need to be modified to be suitable for children.
      Another key distinction in considering a synonymous intervention for children is that the underlying focus of the adult CIMT is on overcoming learned nonuse, in which stroke patients have had repeated failures using their involved extremity, thus reinforcing a learned behavior not to use that extremity. Children may have “developmental disuse,” whereby they may have not used the involved extremity for different tasks. As a result of participation, they may be asked to use their limbs unimanually for the first time in their lives. Thus, they are required to focus on their impairments, and the likely high rate of initial failures at performing these tasks may cause great frustration and potentially affect self-esteem. In addition, unlike stroke patients who often are highly motivated to regain lost function, young children have never used their involved extremity in a typical manner and are likely participating because of their parents’ motivation. Finally, during the restraint wear for the remainder of the 90% of waking hours each day (ie, forced use), children would be required to continue to use their involved extremity exclusively. This demand may cause additional frustration, but, more important, result in increased family burden and safety concerns. For these and other reasons, the procedures associated with CIMT in adults may not be totally appropriate for children.
      For the last several years, our laboratory has been developing and testing a child-friendly version of CIMT. The effort resulted from our prior work in examining hand motor control in children with CP in which we observed marked improvement in performance of the involved hand within 1-hour testing sessions.
      • Wolf S.L.
      • Lecraw D.E.
      • Barton L.A.
      • Jann B.B.
      Forced use of hemiplegic upper extremities to reverse the effect of learned nonuse among chronic stroke and head-injured patients.
      This improvement suggests that at least part of the disability may be due to developmental disuse of the more affected extremity, and that it may be amenable to intervention. We began a series of case studies of CIMT
      • Charles J.
      • Lavinder G.
      • Gordon A.M.
      The effects of constraint induced therapy on hand function in children with hemiplegic cerebral palsy.
      and the intervention development was subsequently supported by funding from the National Institutes of Health.
      Given the inherent confusion found in both the literature and rehabilitation field about the meaning of the term CIMT, defining it in a manner more extensive than what is typically allowed in a research article is becoming increasingly necessary. The purpose of this article is to clarify the essential elements of CIMT and to present the methodology of the intervention as modified for children with hemiplegia. Furthermore, we describe considerations that are important when testing this intervention with children. The modified intervention may provide the basis for future RCTs.

      Methods

      Overview

      Our initial effort to adapt CIMT for children began in 1997, and the method described below is based on piloting and testing with 38 children between the ages of 4 and 14 years. The criteria for participation were: (1) congenital hemiplegia with a difference of at least 50% between the 2 limbs on timed motor tasks of the Jebsen-Taylor Test of Hand Function,
      • Jebsen R.H.
      • Taylor N.
      • Trieschmann R.B.
      Objective and standardized test of hand function.
      and (2) the ability to extend the wrist at least 20° and fingers 10° from full flexion.
      • Wolf S.L.
      • Binder-MacLeod S.A.
      Electromyographic biofeedback applications to the hemiplegic patient. Changes in upper extremity neuromuscular and functional status.
      Of the 38 children, 37 successfully completed the treatment protocol, while 1 child with high levels of frustration discontinued the program at the staff’s request.
      The essential considerations in modifying CIMT for use with children were that the intervention: (1) maintain, in addition to the restraint, the 2 major elements of the adult CIMT (repetitive practice, shaping), (2) be aligned as closely as possible with the methodology employed in the EXCITE trial,
      • Winstein C.J.
      • Miller J.P.
      • Blanton S.
      • et al.
      Methods for a multisite randomized trial to investigate the effect of constraint-induced movement therapy in improving upper extremity function among adults recovering from a cerebrovascular stroke.
      and (3) be as child-friendly as possible. We strived to maintain these central tenets and to provide a treatment intensity that was synonymous with that in the adult studies without using hand-over-hand assistance (except for eating, as required) or facilitation techniques such as tapping. The modified treatment also requires time-on-task that is similar to that for adults. This stipulation precluded using the modified treatment approach with children under the age of 4 years, which was also consistent with our philosophy of using extreme caution in discouraging noninvolved hand use during potential critical periods of development of hand skills.

      General procedures

      The intervention involves restraint of the noninvolved extremity using a sling and engaging the child in unimanual activities with the involved extremity 6 hours a day for 10 days (60h). The sling is strapped to the child’s trunk and the distal end sewn shut to prevent using the noninvolved hand as an assist (fig 1). The sling is worn continuously throughout this time period except when certain agreed-on activities are performed or when a break is requested (total time not to exceed 30min per 6h session).
      Figure thumbnail gr1
      Fig 1A child with the sling. Note that the sling is sewn shut and secured snugly to the trunk with a waist strap to prevent use as an assist.
      The intervention is designed to be conducted with groups of 2 to 3 children to facilitate social interaction, modeling, and encouragement. Each child is assigned to a trained interventionist to maintain at least a 1:1 ratio. In our experience, a ratio of 2 interventionists per child is optimal so that accurate record keeping and documentation do not interrupt treatment. The interventionists can be physical or occupational therapists, and we have found preprofessional therapists and undergraduates who have demonstrated an ability to work with children to be suitable interventionists as well. All interventionists are required to read our procedure manual and to attend a standardized training session before the intervention to ensure they all provide treatment in the same way. We have also found it helpful to have a supervisor on site to provide consultation and guidance to ensure a smooth flow throughout the day and to provide general program organization. At the end of each day, the interventionists and supervisors meet to discuss each child, review progress, solve problems, and plan for the next day.
      The intervention takes place in a room(s) (located at our university) arranged so that spaces can be used for children to work individually with their interventionist(s) or with 1 or 2 other children. There is a flow from 1 workspace to another and from areas designated for fine motor activities to areas designated for gross motor activities. Toys and supplies are openly displayed so that children are free to make choices of activities and interventionists have easy access to them. A selection of choices are agreed on by the children and interventionists based on the target movements (eg, tasks requiring fine grasp, wrist supination), depending on the child’s impairments and interests. Parents can observe the intervention through a 2-way mirror.

      Task selection

      To engage the child in active intervention and to sustain their attention, we established a list of fine motor and manipulative gross motor activities that elicit the general movement behaviors of interest and include a range of functional and play activities in which children might typically participate on a given day. The activities are appropriate for the age of the child and all can be performed unimanually. Specific activities are selected by considering: (1) joint movements with pronounced deficits; (2) joint movements that interventionists believe have the greatest potential for improvement; and (3) child preference for activities that have similar potential for improving identified movements. The task is made progressively more difficult as the child improves in performance by requiring greater speed or accuracy, increased movement repetition, or performance-sensitive adaptations. We adapt the task constraints to allow success and to remove them as one’s skill improves. Task performance is recorded, and task- and age-specific structured feedback is provided for encouragement in a consistent manner. Only positive reinforcement is used. This rule is initiated when the child dons the restraint on the morning of the first intervention day; at that time, to build confidence, the interventionist always begins with a quick task that results in successful completion.
      Table 1 shows the types of activities we use, with examples of targeted movements and how the constraints are graded to vary the difficulty. Overall, we have established a list of 61 tasks across 7 categories. These tasks include board games (eg, Candyland, Monopoly), card games (eg, Old Maid, Uno), manipulative games (eg, Don’t Break the Ice, Battleship), puzzles, arts and crafts (eg, drawing, painting), functional tasks (eg, eating, cleaning table), and gross motor activities (bowling, scatch). We view the choice of specific activities as less important than the movements they elicit. For example, board games can be used to encourage wrist supination and extension, precision grasp, and grasp maintenance. By engaging the child in these activities for the entire 6-hour period per day (≈50%–60% typically spent in time-on-task), we elicit the 2 distinct types of massed practice that are central to CIMT: repetitive practice and shaping.
      Table 1CIMT Activities
      Activity CategoryNo. of ActivitiesShapingRepetitive Task PracticeTargeted MovementsGraded Constraints
      Board games14914Supination, wrist extension, precision grasp, maintaining grasp through changes in spatial orientationActive wrist extension: position deck of cards to elicit wrist extension and grade difficulty by changing position of deck.
      Card games666Supination, precision graspPrecision grasp: less difficult when cards are beveled on deck for easier grasp. Increase difficulty by not beveling the cards.
      Functional tasks1148Wrist extension, supination, and pronationSupination and pronation: for turning key in lock, vary starting position of key to grade from using only supination to using both supination and pronation.
      Gross motor747Shoulder flexion, shoulder abduction, shoulder external rotation, wrist extensionShoulder flexion: elicit shoulder flexion by moving child from easier position stabilized against a wall, to free-standing position that requires more control.
      Manipulative games211421Finger individuation, precision grasp, wrist extension, modifying grasp to accommodate various objectsPrecision grasp: to increase difficulty provide child with increasingly smaller or more complex objects to manipulate.
      Puzzles202In-hand manipulation, precision grasp, release accuracyRelease accuracy: once competency in releasing puzzle pieces is attained, increase difficulty by introducing a puzzle with smaller pieces.
      Arts and crafts202Supination, precision grasp, maintaining grasp through changes in spatial orientationMaintaining grasp: begin child at an easier level with a built-up brush, and increase difficulty by removing assist. Smaller brushes can be introduced.

      Repetitive practice

      We embed repetitive practice in play and functional activities. The activities are performed continuously for at least 15 to 20 minutes. Most task activities could be used for repetitive practice (table 1). An example of repetitive practice embedded in a play activity is the game Connect Four (a vertical 4-in-a-row game with checker disks). The objective is to be the first player to get 4 disks in a row—either horizontally, vertically, or diagonally—by dropping them in slots located at the top of the vertically oriented playing grid. The motor components of play involve grasping the disk, appropriately orienting the disk for placement into a slot, reaching to the top of the grid, and releasing the disk to drop it into the appropriate slot. Depending on the child’s motor capabilities and designated target movements, play can be structured differently to grade the difficulty of a specific movement. In the context of Connect Four, disks can be placed to make picking them up more difficult throughout the game. For example, picking up a disk from a group that is in an enclosed space such as a shallow bowl, versus picking up a disk that is flat on a surface such as a table, is relatively easy. Likewise, placing a disk on a nonskid surface such as Dycem makes it easier to grasp than when it is placed on the table surface. As a child improves, the difficulty in grasping and picking up a disk can be manipulated to elicit a better grasp pattern.

      Shaping

      Shaping involves approaching a behavioral objective (task) in small steps by successive approximation. As the child improves, the task is made more challenging, taking into consideration the his/her abilities (shaping should not exceed a child’s abilities). Interventionists alter constraints to grade tasks according to target movements they want the child to achieve. The strategies include varying temporal, spatial, and accuracy constraints. Temporal strategies include asking the child to complete a task within a particular time limit (30s for all shaping tasks). Spatial strategies include altering the location in which the task is presented to the child. Accuracy strategies include challenging the child’s accuracy in completing the task. Slightly more than half of the tasks can be used to elicit shaping (table 1).
      One example of shaping, using the game Connect Four, involves asking the child to put the game away by picking up disks from the table and placing them in the game box within defined time intervals. The interventionist records the number of disks the child can pick up and place in the box in 30 seconds. Spatial constraints are added by moving the box to a different position in relation to the disks (eg, further away to encourage reach, to the contralateral side to encourage reaching across midline). Accuracy is manipulated by decreasing the opening in the box in which the disks are placed.

      Home practice

      In addition to the 6 hours of structured treatment, the adult CIMT studies typically require forced use for most (90%) of the day. We chose not to have the children wear the sling at home because of the additional invasiveness and the safety concerns associated with potentially having an unsupervised child with a sling (ie, risk of falls). To compensate for this lack of restraint wear at home, we ask parents to have their children engage in home practice activities, either unilateral or bilateral, that require use of the involved extremity. We view this intervention as a window of opportunity. Thus, the practice also begins a regular routine of involved-hand use in which parent and caregivers can problem solve with staff members, with the hope that this interface with the child will continue beyond the 2-week intervention. We encourage parents and caregivers to continue the practice for 2 hours daily after the intervention. Parent/caregivers must keep daily logs in which the specific activities are documented.

      Documentation

      We keep data logs of all activities, including the time spent in shaping and task practice and the number of efforts and successes. This information, along with the parent and caregiver logs that show how long each task was performed, is used to compare any changes in outcome with the intervention dosage.

      Discussion

      Suitability of CIMT for children with hemiplegia

      Our methodology represents our best judgment, prior clinical and experimental experience working with children with hemiplegic CP, knowledge acquired from the adult CIMT literature, and the testing of the 38 children. The high intervention completion rate suggests that the approach is child-friendly and, in our experience, the children enjoy participating. Despite the successful application and promise of the intervention and our attempts to make it child-friendly, it is still taxing. It requires commitment and participation of children, their families, and interventionists. CIMT is potentially physically and psychologically invasive. It must be administered with appropriate sensitivity and a complete understanding of the frustrations a child is likely to experience.
      To date, there have been few RCTs on treatment approaches for upper-extremity function in children with CP.
      • Boyd R.N.
      • Morris M.E.
      • Graham H.K.
      Management of upper limb dysfunction in children with cerebral palsy a systematic review.
      Through careful administration and monitoring of the therapy, we have been able to minimize its risks, which include injury due to absence or delay of corrective postural responses if a child loses balance and a loss of self-confidence as a result of requiring children to focus on their impairments. Beyond the risks associated with administering CIMT, its efficacy is yet to be determined. In the absence of substantial evidence, the risk-to-benefit ratio, as well as the costs associated with such intensive therapy, require careful consideration. Thus, the approach should not be attempted indiscriminately. Beyond general efficacy, there are several other important questions requiring answers before this treatment approach can be advocated. Issues that need to be further explored include the optimal duration of training, what training methods are most effective, the appropriate ages for participation, how the intervention affects long-term potential, who might best benefit, and whether the protocol should adhere strictly to the adult CIMT protocol. Close adherence to that protocol has resulted in an intervention that requires children to spend considerable periods of time-on-task. In our experience, this generally precludes participation of children below the age of 4 years. Further modification would be required to administer this intervention to younger children; it would involve accommodating nap schedules, devising innovative methods of sustaining a young child’s attention, and the probable omission of traditional forms of repetitive practice and shaping.
      Increased CNS plasticity in younger children
      • Chen R.
      • Cohen L.G.
      • Hallett M.
      Nervous system reorganization following injury.
      has been widely assumed to mean earlier is better. For example, blind children taught Braille early during development show involvement of the visual cortex,
      • Sadato N.
      • Pascual-Leone A.
      • Grafman J.
      • et al.
      Activation of the primary visual cortex by Braille reading in blind subjects.
      • Cohen L.G.
      • Celnik P.
      • Pascual-Leone A.
      • et al.
      Functional relevance of cross-modal plasticity in blind humans.
      whereas those taught later do not.
      • Chen R.
      • Cohen L.G.
      • Hallett M.
      Nervous system reorganization following injury.
      Nevertheless, knowledge about the effect of lesion timing and outcome is limited, and recent findings suggest poorer outcome in language development for children who had a stroke under the age of 1 year than children who had a stroke at a later age.
      • Chapman S.B.
      • Max J.E.
      • Gamino J.F.
      • McGlothlin J.H.
      • Cliff S.N.
      Discourse plasticity in children after stroke age at injury and lesion effects.
      Therefore, much more research on the relation between age and plasticity is needed. Our experience with the intervention suggests that the relation between age and outcome is not as simple as it might appear. In fact, it may be inversely related during early development, given the reduced treatment intensity younger children would likely receive if attention given them is inadequate, thereby resulting in less time-on-task. The efficacy of the various treatment approaches may well be dependent on age at the time of treatment, with early intervention aimed at enlarging primary neural networks and later ones aimed at increasing practice.
      • Hadders-Algra M.
      Early brain damage and the development of motor behavior in children clues for therapeutic intervention?.
      Younger children could certainly participate in a forced use paradigm.
      • Willis J.K.
      • Morello A.
      • Davie A.
      • Rice J.C.
      • Bennett J.T.
      Forced use treatment of childhood hemiparesis.
      Although seemingly less invasive, there may be a substantial drop-out rate associated with casting due to discomfort, and the lack of continuous supervision could result in undue psychologic risks or risk of injury. Nevertheless, a study on the cumulative effects of early forced use followed by more active CIMT may be warranted.

      Considerations

      Inclusion and exclusion criteria

      Given the long duration and intensity of this intervention, careful selection of children who will tolerate and benefit from the intervention is necessary. To date, the type of patient most likely to benefit is not known. But successful completion of the protocol requires that children be cognitively competent and able to understand and follow instructions. They cannot have visual problems that would prevent them from performing the intervention. Children who cannot easily move their arm or use their hand (eg, grasp objects) would not receive appropriate intensity, and their frustration level would be too high. Of course, children who already use their involved extremity extensively or whose performance is too good would not likely benefit. Floor and ceiling effects need to be established. A key question is how to quantify hand function that is too mildly or too severely affected. We have adopted criteria from the adult stroke studies in which subjects must extend the wrist at least 20° and fingers at least 10° from full flexion.
      • Wolf S.L.
      • Binder-MacLeod S.A.
      Electromyographic biofeedback applications to the hemiplegic patient. Changes in upper extremity neuromuscular and functional status.
      But analogous studies have not been performed in children. Severe spasticity may also limit grasp and release and limit arm mobility; therefore, that must be considered. Finally, children cannot have balance problems that would put them at risk when wearing a restraint. Thus, this intervention may only be appropriate for a limited group of children with hemiplegia. All 38 children to whom we have administered CIMT have met these inclusion criteria.

      Personnel

      CIMT is a specific training protocol that is unlike other techniques usually employed in PT or OT intervention sessions. Although therapists are qualified to carry out the intervention protocol, they are sometimes required to forego other therapeutic techniques even when they might deem them potentially helpful, particularly if they are not motor-learning based. We have employed both therapists and nontherapists as interveners, and have found that the quality of the intervention has less to do with therapeutic knowledge than it does with the ability to work with children and adhere to the CIMT principles. All interveners must therefore be trained. Sensitivity to the needs of the children and an understanding of the larger psychologic implications of intensive focus on the involved extremity motor impairments are particularly important. To date, our interventions have been supervised by a certified social worker and physical therapist who has more than 30 years experience working with children.

      Costs

      Despite being conducted in a group setting, CIMT as currently defined is a costly intervention that is unlikely to be reimbursed by third-party payers. Each child is assigned 1 to 2 trained interventionists for 60 hours, and they are supervised by a trained professional. The intervention requires suitable space and activities appropriate for the child’s age. Substantial time is also involved in training and supervising interventionists. In addition to efficacy studies, research on the cost of the intervention will eventually be required.

      Restraint selection

      The type of restraint used for the noninvolved hand in pediatric populations is extremely important because it affects comfort, compliance, and, potentially, treatment intensity. Several methods have been used with adults, including mitts, half-gloves, and slings. The choice for adults is likely not as crucial because they are generally more compliant and likely to tolerate discomfort. Nevertheless the choice of a mitt for adult use addresses concerns about loss of balance and falls in the home environment. To date, pediatric studies have used casts,
      • Yasukawa A.
      Upper extremity casting adjunct treatment for a child with cerebral palsy hemiplegia.
      • Willis J.K.
      • Morello A.
      • Davie A.
      • Rice J.C.
      • Bennett J.T.
      Forced use treatment of childhood hemiparesis.
      bivalve casts,
      • Deluca S.C.
      • Echols K.
      • Ramey S.L.
      • Taub E.
      Pediatric constraint-induced movement therapy for a young child with cerebral palsy two episodes of care.
      splints,
      • Crocker M.D.
      • MacKay-Lyons M.
      • McDonnell E.
      Forced use of the upper extremity in cerebral palsy a single-case design.
      mitts,
      • Eliasson A.C.
      • Bonnier B.
      • Krumlinde-Sundholm L.
      Clinical experience of constraint induced movement therapy in adolescents with hemiplegic cerebral palsy—a day camp model.
      and slings.
      • Charles J.
      • Lavinder G.
      • Gordon A.M.
      The effects of constraint induced therapy on hand function in children with hemiplegic cerebral palsy.
      We chose a sling, which is less restrictive than some restraints, but more so than others. This approach is reasonably child-friendly. The sling can be taken off for tasks requiring balance and function, such as hand-washing and toileting. Fastening the sling to the trunk prevents bimanual use or cheating that might occur if the sling was free or if a cast or mitt was worn.
      We also did not want to have the child wear the restraint in the home environment. This approach helped alleviate any frustration from wearing the sling during the 6 hours and lessen the burden of parental supervision to ensure the child’s safety.

      Duration

      The duration of intervention is also important. The adult CIMT studies typically involve wearing the sling for 10 weekdays, 90% of waking hours, with 6 hours of active therapy.
      • Wolf S.L.
      • Blanton S.
      • Baer H.
      • Breshears J.
      • Butler A.J.
      Repetitive task practice a critical review of constraint-induced movement therapy in stroke.
      We believe this duration is too demanding for young children. There are risks associated with keeping children in a restraint when unsupervised, which may happen in the home environment. However, reducing the intervention duration drastically may have repercussions for overall treatment intensity. So we chose to restrain the arm for 6 hours a day for 10 days (60h) of active therapy but to remove the restraint during all other waking hours. However, we ask parents to engage their children in an hour of unimanual practice (without the restraint), and to keep a log of activities so that we could relate compliance with outcome. The 2-week period was chosen to minimize disruption of parents’ and caregivers’ schedules and to permit participation during the summer or school breaks.

      Location

      We have provided the intervention in the home environment.
      • Charles J.
      • Lavinder G.
      • Gordon A.M.
      The effects of constraint induced therapy on hand function in children with hemiplegic cerebral palsy.
      We believed that although the ability to utilize toys, games, and tasks in this environment was advantageous because it reduced the number of new variables introduced, conducting the intervention in the home was disruptive of family life. In addition, the home environment lacked appropriate social context. Consistency in the children’s environment would also be reduced, a circumstance that is not feasible in certain urban households. We chose to conduct CIMT at our university because we could better modify the space to meet the children’s needs, to control the tasks, to minimize distractions, and to allow children to interact with each other, thus increasing social support.

      Recruitment and retention

      The procedures outlined above require an extensive commitment by both the children and the families who participate. Although less intrusive than conducting the intervention in the home environment, parents and caregivers must arrange transportation to the intervention site for 2 consecutive weeks. They must be willing to commit to the hour-long home practice and to complete daily exercise logs. To ensure that parents and caregivers understand and agree to the requirements for participation and understand the safety issues involved, we ask them to sign a behavioral contract, which is a formal written agreement between the parent/caregiver and interventionist that outlines their responsibilities. It serves to ensure that the parent and caregiver understand the intervention, and maximizes compliance and safety.
      At this time, the patients who will most likely benefit from the intervention are unidentified. Our inclusion criteria (particularly wrist and finger extension) were largely adapted from the inclusion criteria of the EXCITE trial,
      • Winstein C.J.
      • Miller J.P.
      • Blanton S.
      • et al.
      Methods for a multisite randomized trial to investigate the effect of constraint-induced movement therapy in improving upper extremity function among adults recovering from a cerebrovascular stroke.
      though we cannot determine whether they are sufficient. Based on our experience in recruiting the 38 children tested to date, approximately 30% of children with hemiplegic CP may not meet the criteria. The percentage of children who actually benefit from CIMT may be higher or lower than what we experienced.

      Assessment

      A major challenge in determining outcome of any upper-extremity intervention in children with hemiplegia is to identify what constitutes improved hand function and to choose assessments to quantify any potential change. This challenge is difficult because most existing tests of pediatric hand function are not designed for children with unilateral impairments and all have limitations. We propose that assessment should take place on 3 levels: (1) changes in unimanual hand performance, (2) changes in bimanual hand performance, and (3) changes in functional outcome.
      Because CIMT typically requires intensive practice with the involved extremity, the most obvious measurements focus on unimanual performance. Standardized tests (for typically developing children) that quantify the time to complete a battery of unimanual tasks, such as the Jebsen-Taylor Test of Hand Function
      • Jebsen R.H.
      • Taylor N.
      • Trieschmann R.B.
      Objective and standardized test of hand function.
      and unimanual items of the Bruininks-Oseretsky Test of Motor Proficiency,
      • Bruininks R.
      would serve this purpose. Because speed is only 1 aspect of hand performance, these tests should be augmented with tests that focus on the quality of the involved upper-extremity movement, such as the Melbourne Assessment of Unilateral Upper Limb Function
      • Randall M.
      • Johnson L.
      • Reddidough D.
      or the Quality of Upper Extremity Skills Test.
      • DeMatteo C.
      • Law M.
      • Russell D.
      • Pollock N.
      • Rosenbaum P.
      • Walter S.
      Although CIMT focuses on unimanual practice, we do not expect that children with hemiplegia will continue to perform the unimanual activities practiced during the intervention once the restraint is removed. Increases in amount and quality of bimanual performance during everyday activities of daily living (ADLs) are therefore equally, if not more, important. Unfortunately, most tests focus exclusively on unimanual performance. Items from the Bruininks-Oseretsky Test
      • Bruininks R.
      that address bimanual performance potentially could be used. The Assisting Hand Assessment
      • Krumlinde-Sundholm L.
      • Eliasson A.C.
      Development of the Assisting Hand Assessment a Rasch-built measure intended for children with unilateral upper limb impairments.
      is a promising new test of effectiveness in which a child with unilateral disability makes use of his/her affected (assisting) hand during bimanual activity performance. The test is standardized for children between ages 18 months and 5 years, but standardization for older children is still in progress.
      Finally, increased frequency and improved quality of movement during performance of ADLs (eg, showering or dressing) constitute real-life improvement and/or greater independence. One method with which to assess this emerging independence is by using caregiver reports that measure the amount and quality of motor function in the daily environment using motor activity logs.
      • Charles J.R.
      • Lavender G.
      • Taub E.
      • Gordon A.M.
      Motor Activity Log.
      However, these logs have not been tested for validity and reliability in children with CP. Other tests, such as the WeeFIM
      Guide for the Functional Independence Measure for Children (WeeFIM) of the Uniform.
      and Pediatric Evaluation of Disability Inventory
      • Haley S.M.
      • Coster W.J.
      • Ludlow L.H.
      • Haltiwanger J.T.
      • Andrellos P.J.
      have been standardized and could supplement use of the logs.
      Additional considerations and limitations for determining efficacy of CIMT include intertrial and intersession variability, the appropriateness of the test for the age of the child, impairment level and testing environment, whether developmental changes occur over the course of study, the cognitive status of the child, the extent to which the test has been standardized and validated, as well as qualitative changes that may not be captured by the above assessments. To obtain a more complete picture of potential changes, clinical tests may need to be augmented with qualitative studies, kinematic assessments, kinetic assessments, or all 3 for different tasks to quantify changes in movement patterns. Finally, many tests take substantial time to perform, a concern for the child, parent, and clinician. To minimize the burden, only a limited number of tests can be performed and no 1 study can likely determine outcome across all of these domains.

      Conclusions

      The outlined methodology is the first effort to describe the application of CIMT with children in more detail than is possible in data-based articles. It attempts to carefully define CIMT, to clarify that it is not simply forced use or traditional PT and OT with a restraint. Overall, we have demonstrated that CIMT can be successfully modified for 4- to 14-year-old children with hemiplegic CP. The modified intervention maintains all practice elements of the adult CIMT (repetitive practice, shaping) and is closely aligned with the methodology employed in the EXCITE trial.
      • Winstein C.J.
      • Miller J.P.
      • Blanton S.
      • et al.
      Methods for a multisite randomized trial to investigate the effect of constraint-induced movement therapy in improving upper extremity function among adults recovering from a cerebrovascular stroke.
      Furthermore, our approach is child-friendly and tolerable for most children. Intervention techniques are embedded into functional and play activities in which children typically participate and ample social interaction is allotted. The modified CIMT intervention may form the basis for testing the efficacy using RCTs. Despite the successful modification and application to children, considerable work is needed to determine the efficacy of CIMT in children with CP. Although CIMT was modified specifically for this population, the methodology should be applicable to other children with hemiplegia (ie, due to stroke, traumatic brain injury, hemispherectomy), though the efficacy for these populations may need to be determined separately. Further modification of CIMT will likely be required to hone in on the specific components of the intervention that are found to be most effective and to apply it with children who are most likely to benefit.

      Acknowledgment

      We thank the interventionists who have volunteered their time. We thank Jennifer Schneider for assistance with this manuscript, and Sarah Blanton and Lena Krumlinde-Sundholm for helpful comments.

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