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Case Reports |
SC DeLuca, PhD, is a Civitan Post-Doctoral Fellow at the University of Alabama at Birmingham. This work was performed to fulfill the requirements for her Master of Arts degree and was supported by the Civitan International Research Center and the University of Alabama at Birmingham.
K Echols, PT, PhD, PCS, is Adjunct Associate Professor of Physical Therapy and Director, Pediatric Neuromotor Research Clinic, Civitan International Research Center
SL Ramey, PhD, is Susan H Mayer Professor of Child and Family Studies, Georgetown University, and Director, CHERITH, Georgetown Center on Health and Education, Washington, DC
E Taub, PhD, is University Professor and Professor of Psychology, University of Alabama at Birmingham, and is the originator of constraint-induced movement therapy
Address all correspondence to Dr DeLuca at Civitan International Research Center, University of Alabama at Birmingham, 1530 3rd Ave S, Birmingham, AL 35294 (USA) (sdeluca{at}uab.edu)
Submitted February 7, 2003;
Accepted May 26, 2003
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Key Words: Cerebral palsy • Constraint-induced movement therapy • Early intervention • Hemiparesis • Physical therapy
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Constraint-induced movement therapy for adults was developed over the past 16 years, based on extensive research with nonhuman primates.8,9 This animal research led to an important part of the conceptual framework guiding constraint-induced movement therapy known as "learned nonuse" (LNU). Learned nonuse was hypothesized to explain the nonuse of an extremity that primates exhibited after somatosensory deafferentation of the dorsal root of the spinal nerve innervating the involved extremity. Research demonstrated that, after deafferentation, a primate does not use the involved limb in any life situation for the entire life span unless 1 of 2 interventions is applied to reinitiate use of the deafferented limb. The 2 interventions found to be successful in producing permanent functional use of the limb were restraint of the intact limb for a period longer than 7 days and shaping of the deafferented limb for many consecutive days.8,9
Learned nonuse was hypothesized to also occur in humans after neurological injury. Specifically, it is hypothesized that LNU occurs as a result of a patient's conditioned suppression of movement, secondary to unsuccessful efforts at voluntary movement during the acute phase of a neurological injury.2–9 That is, following injury, a patient tries to use the affected extremity, but cannot due to physiological reactions in the nervous system during the acute phase of an injury; the repeated failure sets up the conditions leading to LNU. The nonuse, in turn, leads to a contraction of the cortical representation of the affected extremity and prevents the use of the affected extremity in the chronic phase of a patient's recovery.10–15 Constraint-induced movement therapy was developed to help patients overcome this nonuse and was derived specifically from the models shown to be effective with primates.
The clinical application of constraint-induced movement therapy was originally carried out with patients who were at least 1 year poststroke and had not shown progress after reaching the chronic phase. In addition, 2 studies16,17 supported the principles of constraint-induced movement therapy in patients 1 year following head injury. Several researchers2–7 reported improvements in functional use of the impaired UE following constraint-induced movement therapy. Studies10–14 with adults showed that 2 weeks of constraint-induced movement therapy resulted in cortical reorganization in which new areas of the brain were recruited to promote arm movement, which corresponded to substantial improvement in functional use of the affected UE. In adults, constraint-induced movement therapy is believed to affect 2 mechanisms: use-dependent cortical reorganization and overcoming LNU. After an adult has a stroke, the cortical representation of the affected UE reduces to about one half the size of the motor representation of the nonaffected UE.15,18 Two weeks of constraint-induced movement therapy has resulted in doubling the size of the cortical representation of the affected limb.15,18
Theoretically, a child with hemiplegic cerebral palsy may have neural tissue that is underutilized, although the mechanisms for this underutilization may differ from the mechanisms in adults. Rather than LNU, for example, a child may not develop neural pathways involved in movement because of the lack of ability to experience age-appropriate sensorimotor stimuli that lead to the development of UE skills. Taub and Crago15 first hypothesized in 1995 that constraint-induced movement therapy might be especially well suited for use with children because of the great capacity for plasticity in the developing nervous system.19–21
A few studies have begun to address the use of the principles of constraint-induced movement therapy in children. Charles et al22 reported success with 3 school-age children with hemiparesis by applying a modified version of constraint-induced movement therapy, which combines intensive training of the more affected UE with restraint of the less affected UE. Constraint-induced movement therapy for the children involved wearing a cotton sling on the less affected UE while an investigator engaged them in various functional and play activities for 6 hours each day for 14 consecutive days. Positive changes in functions such as manual dexterity, sensory discrimination, and limb coordination were reported. Earlier, Crocker et al23 reported the successful use of constraint-induced movement therapy in a single child, and more recently 2 additional articles24,25 have shown promising results, indicating increased UE use in children who receive constraint-induced movement therapy techniques. The techniques applied in the studies with children, however, have applied only a portion of the entire adult constraint-induced movement therapy protocol, primarily because the number of hours of restraint or treatment was reduced over what has been reported for adults who received constraint-induced movement therapy techniques.
The purpose of this case report is to describe the use of a constraint-induced movement therapy protocol, which we termed "Pediatric Constraint-Induced Therapy (Pediatric CI Therapy)," that: (1) was given more intensively than others have reported and more intensively than recommended for adults with chronic stroke and (2) involved the use of a full-arm, bivalved cast on the less affected UE throughout the 3-week intervention. The report also describes a child who received more than one episode of the Pediatric CI Therapy protocol.
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Prior to intervention, Lilly's passive range of motion was not measured, but appeared to be within normal limits in all joints. She held her right UE in a pattern typical of hemiplegia—shoulder elevation, with protraction, medial (internal) rotation, and adduction, while maintaining the elbow, wrist, and fingers in a flexed position, the forearm in pronation, and the thumb tightly adducted across the palm. She was unable to maintain a sitting position by herself and had a head lag when pulled to a sitting position, but she had good head control in assisted sitting. Protective extension of the left UE was normal, but it was absent on the right side. Lilly was able to independently roll from a supine position to the right side and maintain a propped position when placed on her elbows. She was unable to perform any other independent gross motor skills on the floor (eg, roll from a supine position to a prone position, assume a prone position on her elbows, crawl, creep, assume or maintain a sitting position, assume a kneeling position, pulling up to stand). She performed all reaching tasks using the left UE. She did not use the right UE for any unilateral or bilateral activity. Lilly would tolerate hand-over-hand facilitation to reach for an object, but she was unable to extend the fingers of her right hand to grasp and could only momentarily maintain finger flexion around a small, lightweight object when it was placed in her hand.
Episodes of Care and Outcomes
Intervention 1: protocol.
Lilly's parents signed an informed consent statement that had been approved by the Institutional Review Board of the University of Alabama at Birmingham. The intervention began when Lilly was 15 months of age, and a lightweight, fiberglass cast was applied to the less affected UE from the shoulder to the fingertips. The cast was applied with the UE in 90 degrees of elbow flexion and with the wrist and fingers in a neutral position. The cast was bivalved so that it could be removed once a week to wash the arm, permit active range of motion, and check skin integrity. Intervention started the following day.
The daily 6-hour intervention was provided by the primary therapist (SCD), a graduate student in developmental psychology, who had worked in an adult constraint-induced movement therapy laboratory for over 11 years and who had been involved with the development of the adult constraint-induced movement therapy protocol. Lilly also had a minimum of 4 hours weekly with a board-certified pediatric physical therapist (KE) who performed play-based and functional activities to encourage use of the affected UE.
The Pediatric CI Therapy took place in the child's home for 15 weekdays. Activities were play-based and included use of a variety of toys and objects (eg, bubbles, pop-up toys, balls, musical keyboard, washable markers and paper). The intervention began with sensorimotor activities (eg, placing the hand in pudding, touching objects with unusual and distinctive surfaces) and also involved physical and verbal encouragement of gross motor movements (eg, being put in a quadruped position and facilitated in coming to a sitting position). Behavioral techniques included the use of rewards (predominantly verbal praise, smiles, hugs, cheers, and clapping) and were used throughout all activities. All activities were broken down into step-by-step tasks that could be worked on individually and then chained together in progressive steps toward the targeted goal. In the beginning, for example, the child was praised for any reaching motion; as her ability improved, she was required to make progressively more accurate (ie, targeted) reaching attempts to receive a reward. After she could easily reach, the task was made more difficult by requiring grasp and manipulation of objects in addition to accurate reaching.
Intervention procedures were used to promote floor mobility skills and gross motor arm skills (eg, reaching when Lilly wanted to be picked up), as well as fine motor arm and hand skills. These intervention procedures were provided primarily by the primary therapist but also by the pediatric physical therapist during the time she was available (ie, 4 hours per week). Floor mobility activities focused on weight bearing on the more affected UE, and that extremity was used when facilitated to assume a sitting position. For parental attention, the child was required to reach for the parent(s) with the affected UE prior to being picked up. Fine motor behaviors focused primarily on eating and reaching for her pacifier. Eating began with finger foods (eg, graham crackers, teething biscuits, french fries) and progressed to use of a padded spoon and fork and an infant drinking cup with 2 handles.
Intervention 1: outcomes.
Table 1 and Figure 1 highlight new behaviors and responses to the intervention protocol. As early as intervention day (ID) 1, new functional behaviors appeared, and, by ID3, Lilly voluntarily used her affected UE to reach for and pop a blown bubble. By ID4, the first purposeful grasp appeared, involving grasp of a bubble wand. Lilly was cooperative and seemed eager to engage in the intervention. The cast was worn for 24 hours a day every day for the 3-week intervention period. It was removed once a week for 10 to 15 minutes to allow active range of motion and to check skin integrity. When the cast was removed, Lilly continued to use her right UE for reaching and object manipulation.
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The parents' major concerns at the beginning of intervention 1 concerned the child's comfort and her ability to feed herself while the less affected UE was in the cast, especially because she had difficulty with weight gain. These concerns dissipated during the first week of the first intervention when the parents observed their child's ability to use her more affected UE. On ID2, the mother recorded in her daily log that her child "definitely notices her right hand and is looking at it a lot." Over the first weekend, the mother commented, "I feel so encouraged and really am noticing her being aware of the arm." On several occasions during the second week of the intervention, the mother reported, "She just seems more aware of her entire right side," and she reported that her daughter was using the affected UE "even when we don't remind her." Later, the mother's concerns focused on being able to motivate her daughter to use her more affected UE when the primary therapist was not present. At the end of the first intervention period, the parents reported being extremely pleased and said they were eager to participate in another round of intervention if it were offered at a later date. Over the next several months, the mother reported numerous improvements in the child's recognition and use of the more affected side of her body.
We used several tests to assess Lilly's performance before and after each intervention period. These measures included: (1) the fine motor scale of the Peabody Developmental Motor Scales26 (PDMS), (2) the Denver Developmental Screening Tool27 (DDST), (3) the Pediatric Motor Activity Log (PMAL), and (4) the Toddler Arm Use Test (TAUT). The PMAL and TAUT were developed specifically for this protocol based on the measures used for adults.
The PDMS is a standardized assessment often used for clinical evaluation and in research with children who have neuromotor dysfunction. The PDMS fine motor scale is used to examine grasp, eye-hand coordination, hand use, and manual dexterity, which are skills that we specifically targeted for improvement. Each item is scored as 0 (the child makes no attempt), as 1 (the child makes some attempt), or as 2 (the child completes the item in the specified, age-appropriate manner). The manual for the PDMS fine motor scale reports excellent test-retest reliability (r=.94) and interexaminer reliability (r=.98).26 We followed the standardized administration and scoring procedures, and we did not estimate the reliability of our measurements.
Lilly's performance on the PDMS fine motor scale, which was administered by the pediatric physical therapist, improved from a baseline total score of 43 to 62 (Fig. 2). Specifically, the skills that improved were bringing her hands together in midline, forearm pronation and supination, ulnar/palmar grasping with the hands, transferring a cube between hands, using a raking grasp, and poking her index finger into a hole.
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Lilly's mean PMAL score before the intervention on the "How Much" scale was 0.1, indicating no attempted use of the involved UE according to parental report; following intervention, it was 1.2, indicating occasional self-initiated attempts use of the involved UE according to parental report. Testing 4 weeks after intervention indicated that Lilly's score remained 1.2. The "How Well" scale ratings increased from a preintervention mean of 0.1, indicating none to very poor quality of use with the involved UE according to parental report, to a postintervention mean of 2.8, indicating moderate quality of use with the involved UE according to parental report; 4 weeks following intervention, her score decreased to 1.8 (Fig. 4), indicating poor quality of use with the involved UE according to parental report.
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Before intervention, Lilly did not use her more affected UE on any of the free-choice trials. Following intervention, she used her more affected UE spontaneously in the free-choice condition in 50% of the tasks (Fig. 5). Her mean Amount of Use scale score was 0.2 before intervention and 1.0 following intervention. The mean Functional Ability scale score was 0.2 before intervention and 1.4 after intervention. The global rating remained stable at 1.0 from preintervention to postintervention (Fig. 6).
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The goal of the second episode of Pediatric CI Therapy was to improve prehension, self-help skills, and independent initiation in use of the right UE. During the second intervention period, training was carried out for 21 consecutive days with 6 hours of intervention a day. Weekends were included in an attempt to maximize the effectiveness of the intervention by increasing the number of intervention hours to give Lilly's entire family (eg, her father was involved with work activities for much of the first intervention) an opportunity to be involved in the intervention activities.
Lilly was cooperative during the application of the cast for the second intervention. The primary therapist used intervention procedures similar to those used during the first intervention, starting with more difficult activities, when appropriate. Fine motor behaviors focused primarily on refining hand and finger movements to improve task performance during a variety of play and functional activities. Lilly's emerging prehension skills were applied to increasingly difficult tasks, including grasp, release, isolated finger movements, and forearm supination. Activities started with physical guidance using hand-over-hand assistance, which was "faded" over several days as Lilly became more capable. Other age-appropriate functional skills were systematically facilitated, including pulling up to stand at low surfaces, crawling up and down stairs, and self-feeding using utensils.
Intervention 2: outcomes.
Table 2 and Figure 7 show the child's abilities during the second intervention. By ID2, Lilly appeared to have more controlled and sustained fine motor behaviors, including greater ability to eat finger foods and use a typical spoon. By ID5, she was able to pick up a small object (eg, a small cookie) from a flat surface. Unlike during the first intervention, Lilly showed no unwillingness to carry out new skills when her parents were present. Prior to intervention 2, Lilly was not an active participant in interactions with her 2 siblings. She often simply watched their interactions as they played together. On one intervention day, Lilly increased her interaction with her siblings during play and even pushed one of her siblings away from a toy. This was important because it was a demonstration of Lilly's increased motor competency in everyday social interactions. On ID7, the primary therapist removed the self-adhesive strap from Lilly's walker that helped her maintain grasp by the affected hand. At the end of the day, Lilly was able to maintain independent grasp of her walker with the affected UE take 10 to 20 steps. By ID14, she was beginning to oppose her thumb and finger. Throughout the final week of the intervention, she showed increasingly complex sequences of new UE skills, including the ability to match her grasp to objects of varying sizes, release objects with greater targeting accuracy, and manipulate objects for play during grasp (eg, using a spoon to stir in a pot).
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Intervention 2 assessments involved only the PMAL and the TAUT. Immediately prior to intervention 2, the mean score on the "How Much" scale of the PMAL was 1.6. Four weeks later, following completion of intervention 2, the mean "How Much" scale score was 2.1. In a follow-up assessment 4 weeks later, the mean score was 1.9. The mean "How Well" scale score was 2.2 before intervention 2 and remained stable at 2.2 after intervention 2, but it increased 4 weeks following intervention 2 to 2.5.
The "How Well" measure demonstrated an increase in abilities during intervention 1, which appeared to be maintained during intervention 2. The parent ratings of how often the child used her UE also remained fairly stable. Lilly's parents indicated she had improved during both intervention episodes.
During the videotaped testing session prior to intervention 2, Lilly did not use the more affected UE on any of the free-choice trials. This was a decrease from the testing following intervention 1, when Lilly used the more affected UE during 50% of the free-choice trials. After intervention 2, however, she used her more affected UE for 100% of the free choice-trials. The mean score on the Amount of Use scale of the TAUT and the mean score on the Functional Ability scale of the TAUT changed little following the second intervention. The mean Functional Ability and Amount of Use scale scores did vary between the end of the first intervention and the beginning of the second intervention. At the end of the first intervention, the Functional Ability scale score was 1.4, and by the beginning of the second intervention, the score had risen to 2.5, which indicated a small increase in the functional ability between the first and second interventions. At the end of the second intervention, the score was 2.6. The Amount of Use scale score had also risen slightly from a mean of 1.0 following intervention 1 to a mean of 1.75 before intervention 2 and a mean of 1.86 after intervention 2. The global rating scale score increased from 1.0 prior to intervention 2 to 3.0 following intervention. The raters indicated that Lilly's UE performance had improved to a degree that it would have the potential carryover into her everyday life activities.
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Many factors may have contributed to Lilly's UE gains. She had an excellent relationship with her early intervention providers, who actively sought to maintain and extend her newly acquired skills after intervention 1. Similarly, Lilly's parents and grandparents actively promoted the maintenance and everyday use of UE skills. When examining the gains made during each intervention, the child's maturation also must be considered as a potential contributing factor to the child's skill acquisition. Children naturally go through periods during development when multiple skills are acquired, and it is possible that the intervention coincided with periods in development when this process was ongoing. Even so, it seems likely that the changes during the 2 interventions were primarily the result of the high intensity of the Pediatric CI Therapy (6 hours a day).
One indication that changes in UE skills were more likely the result of the interventions than maturation was the fact that Lilly's spontaneous use of the more affected UE declined during the period between the end of intervention 1 and the beginning of intervention 2, as measured by the free-choice trials of the TAUT. The parental reports given for the PMAL, however, indicated a continued increase in how much Lilly used her UE during this period. The PMAL indicated gains in both how well and how much she used the involved UE during intervention 1, which was followed by a continued increase in how much Lilly used the UE during all other periods. This continued increase makes it difficult to estimate the effects of maturation alone, but the PMAL results must be considered with caution because of the potential bias in the reports of the parents, who were aware of the interventions. When balanced against the results from the free-choice trials of the TAUT, however, for which the raters were not aware of the intervention period, Lilly appears to have lost at least some spontaneous use of the more affected UE between the end of intervention 1 and the beginning of intervention 2. If maturation alone had been responsible for the increased use during intervention 1, then the spontaneity of use in the more affected UE probably would have continued to increase during, between, and after interventions. Instead, the pattern changed during each 3-week intervention period, with an abrupt decline between the 2 interventions. This pattern seems to represent a response to the intervention episodes.
We also observed other specific aspects of Pediatric CI Therapy that may have led to the positive outcomes. First, the constraint of the less impaired UE appeared to facilitate Lilly's focus on using the more affected UE. Second, the positive relationship with the graduate student who provided both episodes of Pediatric CI Therapy and the administration of therapy in the toddler's home may have been additional influences on the positive outcomes.
The Pediatric CI Therapy protocol is now being subjected to a more rigorous evaluation using a randomized, controlled trial involving one intervention episode for children from infancy through school age with hemiparesis associated with cerebral palsy. This effort will help health care professionals to understand several components of this intervention. Although this report describes a unique case because of the 2 intervention episodes, the entire protocol of this intervention must be examined with a larger number of children. To date, only portions of the constraint-induced movement therapy protocol3–10 developed for adults has been applied to a small number of children23–25; however, none of the interventions were as intense as the Pediatric CI Therapy described in this report. Professionals and parents often focus primarily on the constraint in constraint-induced therapy for both adults and children. Although the constraint was an important part of our intervention, both episodes of the interventions also were more intensive than most other interventions described in the pediatric rehabilitation literature, including the interventions reported involving children and constraint-induced movement therapy.22–25 High intensity may be identified in future research as an important contributing factor, but it also might prove to be unneeded.
Future efforts also need to examine the effectiveness of multiple intervention episodes of Pediatric CI Therapy with a larger sample of children with cerebral palsy, the applicability of Pediatric CI Therapy to children with diagnoses other than cerebral palsy, the contribution of all of the primary components of Pediatric CI Therapy to its overall effectiveness, and whether functional changes relate to cortical reorganization and, if so, whether changes vary as a function of age. The cost-benefit ratio of Pediatric CI Therapy also is an important issue to study. Until it becomes accepted as a standard practice of rehabilitative care, third-party reimbursers may be reluctant to pay for therapy with the frequency and dosage levels required in Pediatric CI Therapy. Physical therapy and occupational therapy are costly given that many children with neuromotor disabilities often receive therapy for years. It is vital to understand whether the benefits of short bursts of intensive therapy as given with Pediatric CI Therapy would be more cost beneficial and produce greater intervention effects than when given at traditional dosage levels. Short- and long-term cost-benefit analyses must be a part of future research efforts and will help determine the practicality of transferring Pediatric CI Therapy to clinical settings. Although the costs are likely to be high, the potential long-term benefits may outweigh the costs, especially if children are treated early enough and with sufficient intensity to effect substantial functional changes, which could, in turn, reduce costs in other areas such as health care utilization and special education.
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This work was approved by the Institutional Review Board of the University of Alabama at Birmingham.
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