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ORIGINAL ARTICLE
Efficacy of Forced-Use Therapy in Hemiplegic Cerebral Palsy In-Young Sung, MD, PhD, Ju-Seok Ryu, MD, Sung-Bom Pyun, MD, PhD, Seung-Don Yoo, MD, Woo-Hyun Song, MD, Mi-Jeong Park, OT ABSTRACT. Sung I-Y, Ryu J-S, Pyun S-B, Yoo S-D, Song W-H, Park M-J. Efficacy of forced-use therapy in hemiplegic cerebral palsy Arch Phys Med Rehabil 2005;86:2195-8. Objective: To determine the efficacy of forced-use therapy (FUT) on the improvement of upper-extremity function in children with hemiplegic cerebral palsy (CP). Design: Prospective case series. Setting: Outpatient ambulatory clinic in South Korea. Participants: Thirty-one patients with hemiplegic CP were assigned to the FUT group (n⫽18) or to the control group (n⫽13). The mean age of the patients in the FUT group was 33.2 months and in the control group it was 43.2 months. Interventions: The FUT group wore a short-arm Scotchcast on the unaffected arm for 6 weeks and also participated in a conventional rehabilitation program that included stretching exercises and functional occupational therapy for the upper extremity. The control group underwent the conventional rehabilitation program only. Main Outcome Measure: Hand function tests, including the box and block test (BBT), Erhardt Developmental Prehension Assessment (EDPA), and WeeFIM instrument taken before and after 6 weeks of treatment. Results: Before treatment, there was no significant difference between groups in the BBT, EDPA, and WeeFIM scores. After 6 weeks of treatment, however, the FUT group showed significant improvement in the affected arm in the BBT and EDPA scores, compared with the control group (P⬍.05). The self-care score on the WeeFIM was also significantly improved in the FUT group (P⬍.05). Conclusions: FUT combined with a conventional rehabilitation program appears to be more effective than a rehabilitation program alone in improving affected hand function in children with hemiplegic CP. Key Words: Cerebral palsy; Hemiplegia; Rehabilitation. © 2005 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation ENERALLY, CHILDREN WITH the hemiplegic type of cerebral palsy (CP) have a better prognosis for walking G than do children with other types of CP, and most of the former can walk independently before 3 years of age.1 However, their
From the Department of Physical Medicine and Rehabilitation Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea. Presented in part to the American Academy of Physical Medicine and Rehabilitation, November 2002, Orlando, FL. Supported by the Asan Institute for Life Science for the Promotion of Science for Young Scientists (grant no. 2003-181). No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the author(s) or upon any organization with which the author(s) is/are associated. Reprint requests to In-Young Sung, MD, PhD, Dept of Physical Medicine and Rehabilitation, Asan Medical Center, University of Ulsan College of Medicine, 388-1 Pungnap2dong, Songpa-gu, Seoul, 138-736, South Korea, e-mail:
[email protected]. 0003-9993/05/8611-9831$30.00/0 doi:10.1016/j.apmr.2005.05.007
upper-extremity functioning lags behind their lower extremity functioning. That is, children with CP can walk fairly well using their affected lower extremities, but they seldom use their affected hands because of more affected motor function and sensory deficits than in the leg. The term constraint-induced movement therapy (CIMT) denotes several treatment modalities in which the primary objective is to discourage the use of the unaffected or less affected arm by having the patient wear a splint, cast, or sling as a reminder not to use that arm, thus forcing the use of the paretic arm.2,3 This therapy was developed to help patients overcome the learned nonuse of their paretic arm.2 Learned nonuse is thought to develop after a central nervous system (CNS) lesion (diaschisis or cortical shock in stroke).4 As a result, the nonuse of the affected limb is negatively reinforced by the results of these attempts, such as a fall or the failure to accomplish the intended goal. Although the ability to use the affected limb is recovered after weeks or months, the learned nonuse continues, and the patient does not use that limb to the full extent of its true potential.4,5 There is much controversy concerning the recovery mechanism of the injured CNS, as well as the mechanism and therapeutic effects of rehabilitation treatment. In monkeys with injuries in the brain region that is responsible for motor control of the arm, intensive treatment of the affected hand both improved hand function and induced structural changes in the brain. This suggests that intensive rehabilitation treatment could facilitate motor function and brain reorganization.6 Although many studies applied CIMT to stroke victims, there have been fewer studies of either CIMT or forced-use therapy (FUT) in children with CP.3,7,8 We therefore investigated whether upper-extremity casting combined with a conventional rehabilitation program is more effective than a conventional rehabilitation program alone in children with hemiplegic CP. METHODS Thirty-one patients with hemiplegic CP who had been receiving rehabilitation treatment at the outpatient service of Asan Medical Center were recruited and randomly assigned to the FUT group (n⫽18) or the control group (n⫽13). Fifteen patients in the FUT group and 6 in the control group had right hemiplegia; 3 in the FUT group and 7 in the control group had left hemiplegia. Inclusion criteria included a diagnosis of hemiplegic CP, good health, 8 years old or younger, and able to walk independently. The FUT group included 10 boys and 8 girls (mean age, 33.2⫾8.1mo) and the control group included 5 boys and 8 girls (mean age, 43.2⫾27.9mo). There were no significant differences in age and sex distribution between the 2 groups (table 1). Children with severe paralysis of the upper extremity, cognitive dysfunction that rendered them unable to cooperate during testing, and insecure ambulators were excluded from this study. Informed consent was obtained from the parents after they were given a detailed explanation about the study’s purpose and its possible complications during the study. Study procedures were approved by the Institutional Review Board of the Asan Medical Center. Arch Phys Med Rehabil Vol 86, November 2005
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FORCED-USE THERAPY IN CEREBRAL PALSY, Sung Table 1: Subject Characteristics Characteristics
FUT Group
Control Group
Age (mo) Sex (male/female) Hemiplegia (right/left)
33.2⫾8.1 10/8 15/3
43.2⫾27.9 5/8 6/7
and buttons. When a patient grasped more than 3 types of objects, we used the average value of the developmental age for prehension.10 The WeeFIM, adapted for children from the adult version of FIM instrument,9 evaluates 6 ADLs (self-care, sphincter control, transfers, locomotion, communication, social cognition) with a total of 18 items, each rated on a scale of from 1 (complete dependence) to 7 (complete independence). We compared hand functions in both groups before and after the 6-week program. We calculated the changes in hand function in each group and used them to compare the FUT and control groups. We also used the BBT to examine each child’s unaffected arm to determine whether there was any decline in motor function and complications (eg, limitation of range of motion [ROM], skin problems) caused by long-term immobilization.
NOTE. Values are mean ⫾ standard deviation (SD) or n.
A short-arm Scotchcasta that extended from just below the elbow to the fingertips was applied to the unaffected upper extremity.3,7 The FUT group received occupational therapy (OT) twice a week for 6 weeks. In our preliminary study3 of 5 children with hemiplegic CP, FUT resulted in significant improvement of hand function without any complications after the unaffected arm was immobilized for 6 weeks with a shortarm Scotchcast and the degree of functional improvement persisted for 3 months after the cast removal.3,7,8 Though the optimal period for wearing the cast still needs to be investigated, these results led us to set the duration of cast immobilization at 6 weeks. Each treatment session, which lasted 30 minutes, consisted of individualized functional OT for the affected hand after the patient did stretching exercises for 5 to 10 minutes. Functional OT started with concrete therapeutic goal setting.9 Tasks such as reaching, grasping, holding, manipulating an object, bearing weight on the arm, and making hand gestures were divided into small component skills, which were worked on individually and later chained together to complete a target activity. The occupational therapist also incorporated activities of daily living (ADLs), including eating, grooming, dressing, and using the toilet, into the therapy sessions.8 We used the term forced-use therapy because the patients were not enrolled in a daily intensive training program at the hospital after the cast was applied. Rather, they received conventional OT and parents were instructed to encourage their children to use the affected hand in daily routine activities. The control group received conventional OT identical to that given the experimental group. Two hand function tests, the box and block test (BBT) and the Erhardt Developmental Prehension Assessment (EDPA), and the WeeFIM instrument were performed before the treatment protocol was started and again after the 6-week program was completed. The BBT was developed to assess hand function by measuring the number of blocks shifted from 1 side to the other.10 During the EDPA, which measures the functional age of the hand, the examiner observes the patterns of the prehensile behavior during the grasping of cubes, cylinders,
Statistical Analysis We used the SPSS, statistical package, version 10.1,b for statistical analysis. The group means were compared between the FUT and control groups using nonparametric paired and unpaired t tests. The significance level was set at P less than .05. RESULTS Comparison Between the FUT and Control Groups Before treatment, there were no significant differences between the FUT and control groups, as measured by the BBT, EDPA, and WeeFIM score (table 2). After 6 weeks, the FUT group showed greater improvement, as measured by the BBT, than the control group (2.33⫾1.75 vs 1.08⫾1.93, P⬍.01; table 3). EDPA assessment also found that the FUT group showed greater improvement (.78⫾.69mo vs .47⫾.11mo, P⬍.01; see table 3). As measured by the WeeFIM, the FUT group also showed greater improvement after treatment than the control group in FIM motor scores (P⬍.05, see table 3). This was especially true for the self-care scores, which were significantly more improved in the FUT group than in the control group (1.22⫾.94 vs .23⫾.44, P⬍.01; see table 3). In the cognitive domain, the WeeFIM gain after treatment was higher in the FUT group than in the control group, but this difference was not significant (P⫽.11, see table 3). Complications We found no decline in hand function of the immobilized unaffected arm after 6 weeks in the FUT group, or any cases of joint stiffness or skin problems.
Table 2: Outcome Measures in the FUT and Control Groups Before Treatment Outcome Measures
BBT affected limb (n) BBT unaffected limb (n) EDPA (mo) WeeFIM Motor Self-care Communication Cognitive Total
After Treatment
FUT Group
Control Group
FUT Group
Control Group
8.17⫾5.03 15.30⫾9.39 6.86⫾1.40
8.46⫾7.29 19.92⫾15.78 7.02⫾1.43
10.50⫾5.73 18.12⫾10.06 7.64⫾1.65
9.54⫾7.14 23.15⫾17.12 7.06⫾1.42
60.89⫾10.58 24.22⫾6.10 8.56⫾2.04 21.56⫾4.93 82.44⫾13.97
56.38⫾18.94 20.92⫾8.85 9.46⫾4.27 23.54⫾11.02 79.92⫾29.59
62.78⫾9.96 25.44⫾5.82 8.83⫾1.92 22.17⫾4.57 84.94⫾13.37
57.38⫾18.91 21.15⫾8.73 9.54⫾4.25 23.69⫾10.93 81.08⫾29.58
NOTE. Values are mean ⫾ SD.
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FORCED-USE THERAPY IN CEREBRAL PALSY, Sung Table 3: Functional Changes After Treatment in the FUT and Control Groups Outcome Measures
⌬BBT ⌬EDPA (mo) ⌬WeeFIM Motor Self-care Cognitive Total
FUT Group
Control Group
†
2.33⫾1.75 0.78⫾0.69†
1.08⫾1.93 0.47⫾0.11
1.89⫾2.14* 1.22⫾0.94† 0.61⫾0.98 1.94⫾1.70
1.00⫾2.20 0.23⫾0.44 0.15⫾0.55 1.15⫾2.20
NOTE. Values are mean ⫾ SD. Abbreviation: ⌬, change between pre- and posttreatment. *P⬍.05; †P⬍.01.
DISCUSSION Learned nonuse is believed to occur after a patient experiences repeated unsuccessful attempts to use an affected limb for ADLs.11 The resulting conditioned suppression is reinforced by successful attempts of the same tasks with the unaffected limb, as well as by other sources. Although earlier studies have shown CIMT to be an effective treatment to increase use of the more affected upper extremity, most of those studies were conducted with adult stroke patients.12,13 There have been fewer studies of the effectiveness of CIMT for pediatric patients with hemiplegic CP,3,8,9 although children who received pediatric CIMT acquired significantly more new classes of motor skills, scored better on a laboratory motor function test, and showed more unprompted use of the more affected upper extremity than did controls.9 To our knowledge, this study is the largest to date on the efficacy of FUT for pediatric patients with hemiplegic CP.8,9 Similar to previous reports,3,8,9,13 we found that the FUT group was more successful than the control group in overcoming learned nonuse, suggesting that a nonintensive, repeated limb-use program can elicit skill reacquisition and reduce impairment.14,15 Interestingly, improvement in the FUT group was observed on all functional evaluation tests, including hand function tests and ADLs. In previous studies, ADL questionnaires may have lacked responsiveness to changes in arm function,16 and patients may have had more difficulty in performing personal care activities, as measured by the rehabilitation activities profile, because of their attempts to use their unaffected arms.17 We have shown here, however, that the FUT group displayed significant improvement on the self-care item in the WeeFIM, which suggests that the improvements resulting from the combination of FUT and a conventional rehabilitation program can be transferred to real-life settings.2 In a previous study,9 patients with CP received intensive OT, consisting of 6 hours a day for 21 consecutive days, a program difficult to apply in most clinical settings. It is noteworthy, therefore, that in our study, the FUT group showed greater improvement than the control group, although our routine OT program consists of 2 sessions a week of 30 minutes each. These results indicate that the duration of treatment is less important than participation in home programs and daily routine activities. Forced use of the paretic limb during normal activity is important in improving limb function in children with hemiplegic CP.2,9,18 Another important finding is that the cognitive domain in WeeFIM scores was much, but not significantly, improved in the FUT group. CP not only involves delays or permanent deficits in motor function, but may also involve profound deficits in other functional domains such as sensory awareness,
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responsiveness, and level of engagement with the physical and social environment.9 Parental reports and videotaped sessions indicated that many children receiving pediatric CIMT displayed increased self-confidence, interacted more with their environment, and demonstrated more sensory awareness of the hemiparetic extremities.9 A follow-up study is needed to verify the effect of FUT on cognitive function. Recently, several possible mechanisms of functional recovery and treatment in adult stroke patients have been suggested. The first proposed mechanism associated with brain plasticity is “reactivation of inactive area” by sprouting and formation of new synapses around the injured area, which compensates for the impaired function.10 Another is that a neurotransmitterrelated mechanism that peripheral stimuli such as repetitive ROM exercises or special therapeutic exercise may influence brain reorganization.10 Also, the early decline of upper-limb function in hemiplegia may cause secondary muscle atrophy and weakness, and these may negatively influence the cerebral cortex. To identify the relation between functional recovery after FUT and brain plasticity in pediatric patients, successive studies combined with brain mapping techniques using positron-emission tomography or functional magnetic resonance imaging are needed. Although the CIMT program is a recommended daily intensive rehabilitation program in a hospital setting, we adopted a FUT program that encouraged patients to use the affected hand in daily activities and under supervision of their parents after a cast was applied to the unaffected upper extremity. We used this protocol because of the poor cooperation of pediatric patients in the treatment program in a hospital setting and because of the possibility of a negative psychologic impact on them. We were concerned about complications resulting from prolonged immobilization of the upper arm in short-arm Scotchcasts, such as joint contracture, muscle atrophy, skin problem, and deterioration of hand function. However, we observed none of these during this study. Our study had some methodologic limitation. We did not select the ideal control group of patients. Their average age was older than the experimental group, although the difference was not statistically significant. In addition, because we have used this FUT program for children with hemiplegic CP since 1997, we knew its effectiveness. It was difficult to recruit additional participants for the control group because most parents of hemiplegic CP children wanted to join the FUT program earlier. CONCLUSIONS FUT appears to be an effective therapeutic method for improving function of the affected upper extremity in children with hemiplegic CP. Additional research is needed to determine the long-term effects and to ascertain the underlying mechanism of this new therapeutic approach. References 1. Stempien LM, Deborah GS. Rehabilitation of children and adults with cerebral palsy. In: Braddom RL, Buschbacher RM, Dumitru D, Johnson EW, Matthews D, Sinaki M, editors. Physical medicine and rehabilitation. Philadelphia: WB Saunders; 1996. p 1113-32. 2. Taub E, Uswatte G, Pidikiti R. Constraint-Induced Movement Therapy: a new family of techniques with broad application to physical rehabilitation—a clinical review. J Rehabil Res Dev 1999;36:237-51. 3. Sung IY, Park JM. A pilot evaluation of cast immobilization of intact upper extremity: a new therapeutic approach for spastic hemiplegic cerebral palsy. J Korean Acad Rehabil Med 1998;22: 828-32. Arch Phys Med Rehabil Vol 86, November 2005
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4. Taub E, Wolf SL. Constraint induced movement techniques to facilitate upper extremity use in stroke patients. Top Stroke Rehabil 1997;3:38-61. 5. Andrew K, Stewart J. Stroke recovery: he can but does he? Rheumatol Rehabil 1979;18:43-8. 6. Nudo RJ, Wise BM, Sifuentes F, Milliken GW. Neural substrates for the effects of rehabilitative training on motor recovery after ischemic infarction. Science 1996;272:1791-4. 7. Willis JK, Morello A, Davie A, Rice JC, Bennett JT. Forced use treatment of childhood hemiparesis. Pediatrics 2002;110:94-6. 8. Taub E, Ramey SL, DeLuca S, Echols K. Efficacy of constraintinduced movement therapy for children with cerebral palsy with asymmetric motor impairment. Pediatrics 2004;113:305-12. 9. Bower E, McLellan DL, Arney J, Campbell MJ. A randomised controlled trial of different intensities of physiotherapy and different goal-setting procedures in 44 children with cerebral palsy. Dev Med Child Neurol 1996;38:226-37. 10. Lee RG, van Donkelaar P. Mechanism underlying functional recovery following stroke. Can J Neurol Sci 1995;22:257-63. 11. Taub E. Movement in nonhuman primates deprived of somatosensory feedback. Exerc Sports Sci Rev 1977;4:335-74. 12. Taub E, Miller NE, Novack TA, et al. Technique to improve chronic motor deficit after stroke. Arch Phys Med Rehabil 1993; 74:347-54.
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13. Page SJ, Sisto SA, Levine P. Modified constraint-induced therapy in chronic stroke. Am J Phys Med Rehabil 2002;81:870-5. 14. Butefisch C, Hummelsheim H, Denzler P, Mauritz KH. Repetitive training of isolated movements improves the outcome of motor rehabilitation of the centrally paretic hand. J Neurol Sci 1995;130: 59-68. 15. Winstein CJ, Rose DK. Recovery and rehabilitation of arm use after stroke [abstract]. J Stroke Cerebrovasc Dis 2001;10:197. 16. Nakayama H, Jorgensen HS, Raaschou HO, Olsen TS. Recovery of upper extremity function in stroke patients: the Copenhagen Stroke Study. Arch Phys Med Rehabil 1994;75:394-8. 17. van der Lee JH, Wagenaar RC, Lankhorst GJ, Vogelaar TW, Deville WL, Bouter LM. Forced use of the upper extremity in chronic stroke patients: results from a single-blind randomized clinical trial. Stroke 1999;30:2369-75. 18. 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/behavior. In: Toole J, editor. Imaging and neurologic rehabilitation. New York: Demos; 1996. p 133-54. Suppliers a. 3M Health Care, St Paul, MN 55144-1000. b. SPSS Inc, 233 S Wacker Dr, 11th Fl, Chicago, IL 60606.
