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1 This article was downloaded by: [Karolinska Institute Library] On: 1 September 2009 Access details: Access Details: [subscription number ] Publisher Informa Healthcare Informa Ltd Registered in England and Wales Registered Number: Registered office: Mortimer House, Mortimer Street, London W1T 3JH, UK Physical & Occupational Therapy In Pediatrics Publication details, including instructions for authors and subscription information: Feasibility of a Day-Camp Model of Modified Constraint-Induced Movement Therapy With and Without Botulinum Toxin A Injection for Children With Hemiplegia Ann-Christin Eliasson ab ; Karin Shaw b ; Eva Pontén c ; Roslyn Boyd de ; Lena Krumlinde-Sundholm a a Department of Woman and Child Health, Astrid Lindgren Children's Hospital, Karolinska Institute, Stockholm, Sweden b Habilitation Services for Children and Youth, Stockholm, Sweden c Department of Pediatric Orthopedic Surgery, Astrid Lindgren Children's Hospital, Karolinska Institute, Stockholm, Sweden d Brain Research Institute, Melbourne, Australia e Queensland Cerebral Palsy and Rehabilitation Research Centre, School of Medicine, University of Queensland, Brisbane, Australia Online Publication Date: 01 August 2009 To cite this Article Eliasson, Ann-Christin, Shaw, Karin, Pontén, Eva, Boyd, Roslyn and Krumlinde-Sundholm, Lena(2009)'Feasibility of a Day-Camp Model of Modified Constraint-Induced Movement Therapy With and Without Botulinum Toxin A Injection for Children With Hemiplegia',Physical & Occupational Therapy In Pediatrics,29:3, To link to this Article: DOI: / URL: PLEASE SCROLL DOWN FOR ARTICLE Full terms and conditions of use: This article may be used for research, teaching and private study purposes. Any substantial or systematic reproduction, re-distribution, re-selling, loan or sub-licensing, systematic supply or distribution in any form to anyone is expressly forbidden. The publisher does not give any warranty express or implied or make any representation that the contents will be complete or accurate or up to date. The accuracy of any instructions, formulae and drug doses should be independently verified with primary sources. The publisher shall not be liable for any loss, actions, claims, proceedings, demand or costs or damages whatsoever or howsoever caused arising directly or indirectly in connection with or arising out of the use of this material.

2 Feasibility of a Day-Camp Model of Modified Constraint-Induced Movement Therapy With and Without Botulinum Toxin A Injection for Children With Hemiplegia Ann-Christin Eliasson Karin Shaw Eva Pontén Roslyn Boyd Lena Krumlinde-Sundholm Ann-Christin Eliasson, PhD, is a registered occupational therapist and a professor at the Department of Woman and Child Health, Astrid Lindgren Children s Hospital, Karolinska Institute, at the Habilitation Services for Children and Youth, Stockholm, Sweden. Karin Shaw, MSc, is a registered physical therapist working with the Habilitation Services for Children and Youth, Stockholm, Sweden. Eva Pontén, PhD, MD, is a specialist in hand surgery and orthopedic surgery working with the Department of Pediatric Orthopedic Surgery, Astrid Lindgren Children s Hospital, Karolinska Institute, Stockholm, Sweden. Roslyn Boyd, PhD, is a registered physical therapist associated with the Brain Research Institute, Melbourne, and an associate professor in the Queensland Cerebral Palsy and Rehabilitation Research Centre, School of Medicine, at the University of Queensland, Brisbane, Australia. Lena Krumlinde-Sundholm, PhD, is a registered occupational therapist at the Department of Woman and Child Health, Astrid Lindgren Children s Hospital, Karolinska Institute, Stockholm, Sweden. The authors wish to express their gratitude to the participants at the day camp as well as their camp colleagues, Beatrice Bonnier, Åsa Karlsson, Karin Nyhlen, Annika Pahlén, Magnus Hansson, Sara Jonsson, and Barry Shaw, who made this project possible. They would also like to thank Frida Sandberg for collecting all the data and Brian Hoare for scoring all the videos. Finally they would like to acknowledge the Stockholm Habilitation Service research unit, Stiftelsen Sunnerdahls Handikappfond, Norrback-Eugeniastiftelsen, the Swedish Research Council and Stockholm County Council, the Center for Health Care Sciences, National Health and Medical Research Council (NHMRC) Career Development fellowship, and the Queensland Smart State award (Australia) for supporting this research. Address correspondence to: Ann-Christin Eliasson, Neuropediatric Research Unit Q2:07, Astrid Lindgren Children s Hospital, SE Stockholm, Sweden ( ann-christin.eliasson@ki.se). Physical & Occupational Therapy in Pediatrics, Vol. 29(3), 2009 Available online at C 2009 by Informa Healthcare USA, Inc. All rights reserved. doi: /

3 312 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS ABSTRACT. The objective of the study was to investigate the feasibility of modified constraint-induced (CI) therapy provided in a 2-week day-camp model with and without intramuscular botulinum toxin type A (BoNT-A) injections for children with congenital cerebral palsy. Sixteen children with congenital hemiplegia, Manual Ability Classification System (MACS) level I and II, aged 8 17 years, participated in a CI therapy day camp; of whom five participants (aged years) received intramuscular BoNT-A prior to CI therapy. Assessments were conducted 4 months and 2 weeks before (baselines 1 and 2), immediately after, and 6 months after the day camp. For the children who received BoNT- A, no statistical analyses were conducted due to the small size of the sample. In this group, consistent improvement was only found according to the Melbourne Unilateral Limb Assessment. The children who received only the CI therapy demonstrated improvements in the Jebsen Taylor Hand Function Test (p =.04) at posttest, but improvements were not sustained at 6-month follow-up. No significant improvement was obtained for the Melbourne Assessment or the Assisting Hand Assessment. Children in both groups improved on specially trained tasks: frisbee golf, stacking blocks, and in-hand manipulation. Feedback from the participants suggests that the day-camp model is a feasible intervention following intramuscular BoNT-A injections. The results suggest that children with congenital hemiplegia with varying severity of impairment in hand function may benefit from CI therapy, but not every child demonstrates improvements in hand function. The characteristics of children who respond the best to CI therapy are not clear. KEYWORDS. Botulinum toxin A, constraint-induced movement therapy, group therapy, hand function, hemiplegia, upper extremity Constraint-induced (CI) movement therapy has gained attention as an intervention for children with congenital hemiplegia. Despite its promise, the evidence concerning the effects is tentative at best (Charles & Gordon, 2005; Hoare, Wasiak, Imms, & Carey, 2007). It is difficult to compare the results between studies, as there are considerable variations in the methods and modes of delivery of interventions as well as the outcome measures used (Charles & Gordon, 2005). One challenge has been the development of a child-oriented approach, and different methods have been described (Bonnier, Eliasson, & Krumlinde-Sundholm, 2006; Eliasson, Krumlinde- Sundholm, Shaw, & Wang, 2005; Gordon, Charles, & Wolf, 2005). Another

4 Eliasson et al. 313 intervention for children with congenital hemiplegia is intramuscular injections of botulinum toxin type A (BoNT-A), which aims to diminish muscle overactivity (Corry, Cosgrove, Walsh, McClean, & Graham, 1997) and improve the quality of movement when combined with various models of upper-limb training (Fehlings, Rang, Glazier, & Steele, 2000). A systematic review concluded that there is uncertainty in the results (Park & Rha, 2006). Some studies indicated positive results of BoNT-A with usual training and care (Fehlings et al., 2000; Lowe, Novak, & Cusick, 2006), but no additional benefits over upper-limb training alone have been reported for others (Speth, Leffers, Janssen-Potten, & Vles, 2005; Wallen, O Flaherty, & Waugh, 2007). No studies have examined intensive training after BoNT-A injection; at most training was provided three times a week for 12 weeks (Speth et al., 2005). The protocols commonly included different aspects of occupational therapy in addition to specifically targeting hand training. The effectiveness of CI therapy of high intensity following intramuscular injections of BoNT-A has not been examined. A modified CI training paradigm, developed and used in our earlier studies and in clinical practice in the Stockholm region, has been enjoyed by the participants, and many have shown significant and sustained improvements in hand function (Bonnier et al., 2006; Eliasson et al., 2005). The key ingredient in the Stockholm paradigm was restraining the dominant hand by a stiff glove to elicit intensive use and training of the impaired hand. Furthermore, the content and structure of the training was developed to make it enjoyable and motivating and was adapted to the ability levels of individual children within the group, giving numerous opportunities for repetitive practice (Bonnier et al., 2006; Eliasson et al., 2005; Gordon et al., 2005). The training focused on hand use, practiced by performing complete tasks and motivating activities. Participants were encouraged to discover various strategies for solving tasks requiring different types of hand use (Skold, Josephsson, & Eliasson, 2004). The training was based on knowledge generated from the motor learning and motor control literature and also from knowledge of characteristics of the impaired hand (Eliasson, 2006; Haugen & Mathiowetz, 1989; Smith & Wrisberg, 2001). This training paradigm is somewhat different from the CI therapy described by Taub and colleagues, in which specific components or part practice of movements are practiced by using shaping techniques (Gordon et al., 2005; Taub et al., 1994). An issue of concern for intervention studies is the selection of assessments for optimal understanding and interpretation of the results. Interventions need to be evaluated from a broad perspective, and more importantly there is a need to measure the elements targeted in the intervention

5 314 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS (Majnemer & Mazer, 2004). Few assessments of hand function, however, are validated for individuals with hemiplegic cerebral palsy with adequate reliability and demonstrated responsiveness to change. The wide variation of assessments that have been used in research might be because of the lack of suitable assessments or consensus on what is important to measure (Charles & Gordon, 2005). Results from studies indicate that speed, force control, coordination, and dexterity are components of hand use that may improve following CI therapy (Charles & Gordon, 2005; Hoare et al., 2007; Taub et al., 2007). Typically there is a relationship between skillful hand use and speed of performance, demonstrated for example by age-related norms from upperlimb scales like the Bruininks Oseretsky Test of Motor Proficiency (Bruininks & Bruininks, 2006). Even though children with cerebral palsy do not follow typical development, the relation between skilled performance and speed of performance seems relevant so that a timed test would be useful. Another important issue is whether there is a transfer effect from unilateral training (in CI therapy) to bimanual performance, which would be the hoped-for outcome for participants with hemiplegia. For BoNT-A intervention the aims are to weaken relatively overactive muscles (agonists) so that the relatively weaker antagonists can act more normally on the joints. Botulinum toxin also reduces the spasticity in the injected muscles. Accordingly, muscle tone, quality of movements, grip strength, and speed are important to measure (Park & Rha, 2006; Reeuwijk, van Schie, Becher, & Kwakkel, 2006). Our previous day-camp model of CI therapy for adolescents with congenital hemiplegia was successful in that the participants gained improved hand function; however, the sample was small (n = 9) (Bonnier et al., 2006). The purpose of this study was first to investigate if a day-camp model of CI therapy is a feasible intervention in children and adolescents with congenital hemiplegia after intramuscular BoNT-A injections and second whether the results of our previous study (Bonnier et al., 2006) could be replicated. METHOD Design This study used a prospective design with two baselines with 4 months between assessments, posttreatment assessment, and a 6-month follow-up.

6 Eliasson et al. 315 Recruitment of Participants Written invitations to attend a day camp with CI therapy were distributed to potential participants in the Stockholm City County through their therapists. The target participants were persons with congenital hemiplegia, aged 8 18 years and attending mainstream schools, with reduced hand function but able to grasp and lift a 500-g bottle with the impaired hand, to ensure a minimal level of grasping ability and strength. Families of children suitable and interested in intramuscular injections of BoNT-A as a complementary treatment to CI therapy were referred to an experienced hand surgeon for decisions about suitability. The final decision of group assignment was based on the family s wishes and the professional judgments of the most suitable treatment (Rosenbaum, 2004). The groups were those of (a) participants who were suitable and willing to combine intramuscular BoNT-A injections with CI therapy (CI therapy and BoNT-A group) and (b) participants only interested in the day camp (CI therapy group). Informed consent was obtained from all participating families, and the study was conducted after approval from the research ethics committee at the Karolinska University Hospital in accordance with ethical standards of human experimentation and with the Helsinki Declaration of 1975 as revised in Participants Sixteen participants with congenital hemiplegia of functional levels I and II of Manual Ability Classification System (MACS; Eliasson et al., 2006), aged 8 17 years, were entered (Table 1). Based on parental preference children were entered into the following groups: (a) CI therapy and BoNT-A group: Five participants were suitable for being injected and willing to be injected with intramuscular BoNT-A (Botox R, Allergan, Inc., Irvine, CA) 2 weeks prior to CI therapy. (b) CI therapy group: Eleven participants were included in the group receiving CI therapy alone. Participant demographics are reported for both groups in Table 1. Measures The Jebsen Taylor Hand Function Test (JTHFT) was used to measure dexterity using the six timed (in seconds) subtests with the writing test

7 316 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS TABLE 1. Characteristics of Children With Hemiplegia, Ordered From the Mildest to the Severest Hand Function Based on the Jebsen Taylor Hand Function Test Subject Gender Age Side First Baseline (s) Strength (N) Sensibility a MACS CI therapy group 1 F 16 L Good 1 2 M 12 R Good 2 3 M 12 R Decreased 2 4 M 17 R Poor 2 5 M 10 R Decreased 2 6 M 11 R Good 2 7 b M 8 L Good 2 8 c F 10 R Good 2 9 M 13 L Good 2 10 F 8 L Poor 2 11 M 12 R Poor 2 BoNT-A and CI therapy group 12 F 13 R Poor 2 13 b F 16 R Good 2 14 c M 14 L Good 2 15 M 11 R Good 2 16 c F 15 L Poor 2 a See the Method section for explanation. b Did not participate in 6-month follow-up. c Have had surgery more than 2 years ago. excluded (Taylor, Sand, & Jebsen, 1973). If a subtest was not completed within 2 min a score of 120 s was assigned on that subtest (Gordon, Charles, & Wolf, 2006). Maximal time was consequently limited to 720 s for each individual. The test retest data of the JTHFT is fairly strong (Taylor et al., 1973). The Melbourne Unilateral Upper Limb Assessment (Melbourne Assessment) is a standardized test for children, aged 5 15 years, with neurological impairment (Randall, Carlin, Chondros, & Reddihough, 2001). It measures quality of movement related to reach, grasp, release, and manipulation in 16 tasks. The test is video recorded, and the raw score sum is converted to a percentage scale (1 100). The test is validated for children with cerebral palsy with strong intra- and inter-rater reliability as well as with test retest validity (Randall et al., 2001) The Assisting Hand Assessment (AHA) is a standardized test for children with unilateral upper-limb impairments, which measures the effectiveness with which a child makes use of his/her impaired hand in

8 Eliasson et al. 317 bimanual activities (Krumlinde-Sundholm & Eliasson, 2003). The AHA is designed and validated for children with unilateral impairment, between the ages of 18 months and 12 years. Its intra- and inter-rater reliability is strong (Holmefur, Krumlinde-Sundholm, & Eliasson, 2007; Krumlinde- Sundholm, Holmefur, Kottorp, & Eliasson, 2007). For participants over 12 years of age, an adolescent version of the test was used. The AHA is scored from video with 22 items rated on a 4-point scale, resulting in a raw score range; the higher the score, the higher the child s performance. Grip strength was measured using the electronic measuring device Grippit and recorded in N (Nordenskiöld & Grimby, 1993). The instrument is validated for use with children, and there are norms for Swedish children (Hager-Ross & Rosblad, 2002). The two-point discrimination test (2PD) was used to measure sensibility (Krumlinde-Sundholm & Eliasson, 2002). Sensibility was considered good when 2PD was possible at 3 mm, reduced if 7 mm of spacing was discriminated, and poor when it was not possible to discriminate 7 mm spacing. The in-hand manipulation task (Exner, 2001) involves moving a pen linearly (from the point to the top of the pen) along the pads of the fingers (pen shift task). The task was practiced for approximately 5 min each day during the day camp. Through the use of video analysis, the shift task was scored from 0 to 8 (0 = no transfer of the pen, 8 = translation in both directions) (Bonnier et al., 2006). Frisbee golf is a game in which the goal is to traverse a 350-foot long course, ending with the frisbee in a basket in the fewest number of throws (Bonnier et al., 2006). Each consecutive throw is made from where the frisbee comes to rest after the last throw. On the first and last days of the camp the total number of throws needed to complete the course was recorded. The participants practiced frisbee golf on five occasions during the day camp. The ability to stack blocks in towers was assessed on the first and last days of the camp and measured as the total height in cm. The task was practiced for 10 min a day. Any form of construction was allowed while practicing, but the measured height was only legitimate for towers built in single file with wooden blocks (with sides of 3 cm). Procedure The assessments were conducted by an occupational therapist not involved in the day camp. Another independent occupational therapist,

9 318 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS not aware of the order of assessments and treatment groups, scored the participants performance on the video-based assessments. The assessment for the first baseline occurred 4 months before the day camp. The second, preintervention baseline assessment was performed 2 weeks before the day camp commenced. The third, postintervention assessment was performed the week after the day camp ended, and the fourth assessment was performed 6 months later, i.e., at follow-up. Measures of the in-hand manipulation task, frisbee golf, and stacking blocks were only performed at the second baseline and directly after the day camp. BoNT-A Injection The five children receiving BoNT-A were injected after the second baseline assessment, 2 weeks prior to the day camp. This ensured that the peak effect of the BoNT-A injection had been reached before entering the camp. The 4-month baseline period thus served as a pretreatment control period. Dosage of BoNT-A was calculated on the basis of body weight, and selection of injected muscles was determined by observation of the participant s individual motor patterns in functional tasks (Table 2). Target muscles for injections (diluted to 100 U Botox/ml normal saline) were located using an EMG amplifier and muscle stimulator. A total dose of 2 4 U/kg body weight was given. The muscles were injected through the same insulated 27-gauge needle (Allergan) that was used for the electrical stimulation to locate the target muscle. Topical dermal anesthetic (EMLA R ) combined with nitrous oxide inhalation or midazolam was used to minimize pain due to the injections. Day Camp The duration of the day camp was 7 hr per day, over 9 days, for a 2- week summer period (total 63 hr). The camp was held in a favorable open recreational area and was conducted by occupational therapists, physical therapists, and volunteers. The training program primarily consisted of daily recreational activities of interest to the participants. Each participant was given a restraining glove to be worn on the unimpaired dominant hand. The glove prevented finger flexion and fine manipulation but enabled gross hand movements and stabilizing for bimanual gross grasp. The day-camp model was a replication of our earlier day camp (Bonnier et al., 2006; Eliasson, Bonnier, & Krumlinde-Sundholm, 2003). Activities were specifically

10 Eliasson et al. 319 TABLE 2. Muscles Injected With BoNT-A and Change in Grip Strength Grip Strength (N) Subject Indication Injection Baseline 2 Posttreatment 12 Diminished thumb abduction during initiation of grip. Impaired supination. 13 Volar flexion of wrist during grip. Impaired supination. 14 Volar flexion of wrist and flexion of thumb during initiation of grip. Adduction and internal rotation of shoulder. 15 Volar flexion of wrist and adduction of thumb during initiation of grip. Difficulty in extending fingers and release. Impaired supination. Diminished reaching capacity. 16 Flexed thumb and fingers flexed in MCP, extended in PIP and DIP during initiation of grip. Upper arm adducted to thorax. AddP,FPB,PQ FCU, FCR, PT, PQ FCU, FPB, subscapularis FCU, FCR, FDP, AddP, PQ, biceps FPB, opponens, interosseus muscles, AddP, TM, pectoralis Note: AddP = adductor pollicis, FPB = flexor pollicis brevis, PT = pronator teres, PQ = pronator quadratus, FCU = flexor carpi ulnaris, FCR = flexor carpi radialis, FDP = flexor digitorum profundus, TM = teres major, MCP = metacarpophalangeal joint, PIP = proximal interphalangeal joint, DIP = distal interphalangeal joint. selected to include both gross motor function of the upper extremity and hand manipulation tasks. Examples of gross motor activities are playing frisbee golf, basketball, boule, and water games. Specific fine-motor board games and dice and card games were incorporated into daily activities. Furthermore, participants engaged in activities of daily living such as the preparation of meals, eating, and washing of dishes. The program included challenging activities that require use of both hands such as rock climbing and canoeing. During these tasks the glove was not used. Overall, the attendance in the modified training program was good; the participants were engaged in the training and completed the expected tasks. Following completion of the day camp, all participants were asked five open questions about their perceptions of the day camp in a paper questionnaire: The questions were as follows: What was the best? What was the most important? What was the hardest? Which activity was the

11 320 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS most boring? Which activity was the most fun? Participants recorded their responses anonymously. Data Analysis The data were not normally distributed; therefore, nonparametric statistics were used. The median and percentiles were computed for each standardized measure. For the CI therapy group (n = 11) the overall effect was calculated by the Friedman ANOVA, and the Wilcoxon matched-pair test was used to measure the difference between scores at different assessment times. To assess whether the treatment effect was related to the severity of the impairment in hand use as measured by the JTHFT, an approach suggested by Kirkwood and Sterne (2003) was used. The correlation between the effect of treatment (pretreatment score on the JTHFT minus posttreatment score on the JTHFT) and the average of pre- and posttreatment measures (the sum of pretreatment and posttreatment scores on the JTHFT divided by two) were calculated. The level of statistical significance was set at p<.05. For the CI therapy and BoNT-A group, individual results and medians are reported due to the small number of participants (n = 5). No comparisons between groups were made. RESULTS The median and percentiles for each standardized measure at each of the four assessments are reported by group in Table 3. Data were missing for two participants, one from each group, at 6-month follow-up (Table 1). Children Who Received CI Therapy and BoNT-A Standardized Measures As only five participants received BoNT-A prior to CI therapy, no group comparisons could be made, and individual results are reported. Two of the five participants improved on the JTHFT (lower time score) directly after the day camp. The scores were sustained results at 6-month follow-up (Figure 1a). All five participants had improved in the quality of movement on the Melbourne Assessment immediately posttraining; however, the effect was not sustained at 6-month follow-up (Figure 2a and Table 3).

12 TABLE 3. Results of Standardized Measures for Children Receiving CI Therapy and Children Receiving Both CI Therapy and BoNT-A. Median and Percentiles Are Reported CI Therapy, n = 11 CI Therapy and BoNT-A, n = 5 Follow-Up, Follow-Up, Test Baseline 1 Baseline 2 Postintervention n = 10 Baseline 1 Baseline 2 Postintervention n = 4 Jebsen (s) 165 ( ) 160 ( ) 109 (78 260) a,b 142 (84 322) ac2 201 ( ) 145 ( ) 122 ( ) 122 ( ) Melbourne (%) 77 (63 84) 77 (65 86) 78 ( (65 87) 77 (55 81) 76 (57 82) 81(65 85) 72 (58 85) AHA (raw score) 60 (52 62) 60 (54 62) 60 (57 63) 58 (54 60) 58 (50 62) 59 (53 62) 57 (52 62) 57.5 ( ) Grip strength (N) 44 (24 72) 38 (21 82) 40 (24 84) 42 (25 76) 54 (40 78) 50 (45 91) 67 (36 78) 52 ( ) a p <.05. b The Wilcoxon matched-pair test was calculated between baseline 2 and postintervention. c Calculated between baseline 1 and follow-up. 321

13 322 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS FIGURE 1. Dexterity measured in seconds by the JTHFT in the involved hand obtained on four occasions: two baseline assessments before intervention, postintervention and at 6-month follow-up. Individual data for (a) individual children who received both CI therapy and BoNT-A (n = 5) and (b) the CI therapy group alone (n = 11; n = 10 at 6-month follow-up). The thick dotted line represents the median results.

14 Eliasson et al. 323 FIGURE 2. Quality of movements as measured by the Melbourne Unilateral Upper Limb Assessment on four occasions: two baseline assessments before intervention, after intervention and at 6-month follow-up. Individual data for (a) individual children who received both CI therapy and BoNT-A (n = 5, with one participant missing at 6-month follow-up) and (b) the CI therapy group alone (n = 11; n = 10 at 6-month follow-up). The thick dotted line represents the median results.

15 324 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS FIGURE 3. Number of frisbee golf throws required before and after intervention for individual children (n = 16). The dotted line indicates children who received both CI therapy and BoNT-A. There were no notable changes in the AHA results on any occasion. Small grip-strength fluctuations occurred with minor increases in strength in three participants and decrease in strength in two other participants (Table 2). Trained Tasks Four of the five children demonstrated a decrease the number of throws needed to get the frisbee into its basket, and one participant showed no improvement (Figure 3). All three children who participated in the assessment from the first to the last day during the day camp improved on the stacking blocks task from the first to the last day of the day camp (Figure 4). Two of the five children demonstrated improvement in in-hand manipulation on the pen shift task (Figure 5). Children Who Received CI Therapy Standardized Measures There was no statistically significant difference between the first and second baseline scores for any of the standardized measures, indicating

16 Eliasson et al. 325 FIGURE 4. Height in cm of stacked blocks obtained before and after intervention for individual children (n = 14). The dotted lines indicate children who received both CI therapy and BoNT-A. FIGURE 5. Score for in-hand manipulation, i.e, vertical pen transition along the fingers before and after the intervention (n = 16). The dotted lines indicate children who received both CI therapy and BoNT-A.

17 326 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS the stability of performance in the control period (no training). The children demonstrated a significant improvement in speed and dexterity on the JTHFT (overall effect chi-square = 8.93, p =.03) with a decrease in the median time from 160 to 109 s between the pre- and postcamp assessments (p =.04). Wilcoxon matched-pair analysis indicated that the improvements were not sustained at 6-month follow-up (p =.95) (Figure 1b and Table 3). The children did not demonstrate an improvement in quality of movement on the Melbourne Assessment (overall effect chi-square = 0.23, p =.9; Figure 2a and Table 3). There were no changes in bimanual performance on the AHA (overall effect chi-square = 4.48, p =.21) or the task measuring grip strength (p =.2; Table 3). The correlation between severity of impairment in hand use (using the mean of pretest and posttest of the JTHFT) and improvement on the JTHFT (difference between pre- and postintervention; see the Method section) was r = The correlation indicates that the participant s severity of impairment in hand use was not related to change in hand use following intervention. Trained Tasks Children who received CI therapy demonstrated significant improvements in the specifically practiced tasks. In frisbee golf, the number of throws needed to get the frisbee into the basket decreased from the first day of camp to the last day of camp from a median of 33 throws to 26 throws (p =.003; Figure 3). Stacking blocks as high as possible also improved from a median of 27 cm to 43 cm (p =.005) from the first to the last day of the day camp (Figure 4). In-hand manipulation increased from a median of 2 points to 3 points postintervention (p =.02; Figure 5). Participants Experience of the Day Camp Overall, the participants enjoyed the camp. They reported that the best thing about the day camp was meeting new friends with the same problems as themselves; the feeling of not being alone with this type of disability was commonly mentioned. Several participants reported conflicting feelings, saying that the training was both the most important and the most difficult issue. Typically, they had not expected that wearing the glove would be as difficult as it turned out to be. Several participants reported that it was fun or very encouraging to recognize the improvement, for example, when playing frisbee golf. One child reported that not noticing any improvement

18 Eliasson et al. 327 during the camp was tough for her. The activities reported to be the most fun were rock climbing, building and climbing on crates, and water games. When asking about the most boring activities several participants answered that they could not come up with anything, whereas others mentioned that the structured hand training including the in-hand manipulation task was boring. Once a day, there was a group discussion in which the participants could discuss the training and how they felt about it. This conversation was mentioned as important, with some participants emphasizing that it was both important and very hard to talk about themselves. Although the training was mentioned as important, it was the whole concept including the environment for the day camp friends and activities which made this event important for the participants. DISCUSSION All participants could successfully participate in the modified CI therapy day-camp program. There was no difference in the carrying through, engagement, or participation between the training activities for the participants with or without BoNT-A. The main results from the CI therapy alone group support the results from the previous day camp organized in a similar way (Bonnier et al., 2006; Eliasson et al., 2003), demonstrating improved speed of performance and improvements in specifically trained tasks. In the participants who also received injections of BoNT-A, a consistent change was only found oi the Melbourne Assessment. Children who received only the CI therapy, however, did not demonstrate changes in the Melbourne Assessment. To our knowledge, this is the first report of the combination of intramuscular BoNT-A injections followed by intensive training. The results varied among the five children. The only outcome in which a trend was seen that could be related to effects from BoNT-A was in the Melbourne Assessment. All five participants demonstrated improved quality of movements directly after the day camp, but the effect was not sustained at the 6-month follow-up, a result probably related to the transitory effects of BoNT-A. Decreased grip strength after BoNT-A has been reported (Fehlings et al., 2000) but did not occur in this study. Children demonstrated almost no change in grip strength, and no evidence of fatigue was reported. The finding that the children who received BoNT-A injections could complete the training sessions is encouraging. The improvements in speed and dexterity measured by the JTHFT support the outcomes previously reported (Bonnier et al., 2006; Charles,

19 328 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS Wolf, Schneider, & Gordon, 2006; Gordon et al., 2006). In our study, however, none of the children in the CI therapy group sustained the improvements at the 6-month follow-up. Among the 14 participants available for follow-up, 9 had improved JTHFT scores compared to before the day camp (baseline 2). As illustrated in Figure 1, the performance of participants both with and without BoNT-A varied considerably. This highlights the question that which children with congenital hemiplegia are most likely to benefit from CI therapy? Unfortunately, the combination of BoNT-A and CI therapy does not, so far, provide any new insights into this question. The findings suggest that children with a wide spectrum of hand use may benefit from CI therapy. The participants in this study had more severe limitations on hand function than the participants in the first day camp (Bonnier et al., 2006; Eliasson et al., 2003). The median time to perform the JTHFT before the day camp was 160 s compared with 72 s in the previous study (Bonnier et al., 2006; Eliasson et al., 2003). Time to perform the JTHFT improved 32% among children in this study, compared with a 31% improvement in our previous study (Eliasson et al., 2003). Our perspective is also supported by Gordon and coauthors, who reported a JTHFT baseline above 240 s and significant and sustained improvements (Charles et al., 2006; Gordon et al., 2006). There was no transfer of the improvement from one-handed training to bimanual performance as measured by the AHA. The AHA has previously demonstrated responsiveness to change following a longer treatment period of CI therapy in younger children (Eliasson et al., 2005). It is possible that a 2-week unimanually oriented treatment duration is too brief to change the habitual use of the hands in bimanual activities or that changes in bimanual function are harder to achieve in this sample of older individuals. The AHA has been used in a study with an intensive, 2-week bimanual treatment approach (hand arm bimanual intensive therapy or HABIT) in which improvements in bimanual functioning were demonstrated (Gordon et al., 2007). Further studies are needed to evaluate which treatments lead to improved bimanual performance, probably the most important aspect of hand function in people with unilateral disabilities. In contrast to the varying results of the standardized assessments, most of the children demonstrated improvement in the specifically practiced tasks (mostly novel tasks) of in-hand manipulation, throwing the frisbee, and stacking blocks. Individuals receiving BoNT-A improved within the same range as children who received only CI therapy. This finding emphasizes the premise You learn what you practice as suggested in the outcome of our earlier day camp (Bonnier et al., 2006). It also suggests that improvements

20 Eliasson et al. 329 in hand use were quite specific, and transfer effects to a more generally improved hand function as measured by the different standardized tests are not obvious. Appraisal of Measures Our proposal that BoNT-A might change other aspects of hand function than CI therapy alone was supported, because it was just the BoNT-A group that demonstrated changes in quality of movements as measured with the Melbourne Assessment. This assessment has been used in BoNT-A studies (Boyd, 2004; Speth et al., 2005; Wallen et al., 2007) but not for evaluation of CI therapy, which suggests that other investigators share our assumption that this instrument may be sensitive to changes related to effects of BoNT-A. Testing speed with a timed test such as the JTHFT is recommended as a general measure of (unilateral) hand capacity, which is addressed in CI therapy. Baseline measures however varied, suggesting that stability is a potential issue for children with congenital hemiplegia. The most functionally important aspect of hand function in people with unilateral impairments is probably the ability to perform daily activities, which mostly involve bimanual performance. In this study children did not demonstrate change in the AHA; nevertheless we recommend including bimanual function as an outcome. Limitations To investigate the feasibility of the CI therapy in combination with BoNT-A, we invited families with children potentially suitable for the intramuscular BoNT-A treatment and interested in CI therapy. The number of potential participants interested in receiving injections of BoNT-A was lower than expected. The parents were given the opportunity to decide the treatment methods after being informed by experts about suitable options for their children. Most families and children were interested in just attending the CI therapy program. This resulted in the major limitation of this study: uneven numbers of participants in the two groups and the low numbers in the BoNT-A group, which made statistical comparisons impossible. The two-baseline prospective design aimed to examine stability over time when no change was expected and to serve as a pretreatment control period. The fluctuation in baseline indicates that a single subject design with repeated baseline measurements is preferable. The data from the

21 330 PHYSICAL & OCCUPATIONAL THERAPY IN PEDIATRICS first and second baseline for the JTHFT were not stable and unexpected, because the test has been the primary outcome measure in several studies (Bonnier et al., 2006; Charles, Lavinder, & Gordon, 2001; Charles et al., 2006; Gordon et al., 2006). We did not expect changed performance on the JTHFT in a 4-month period for this age group. From clinical experience we know that assessments measuring speed may be difficult to use in people with varying muscle tone. Clearly, the JTHFT needs further investigation of both stability and sensitivity for change in persons with cerebral palsy. Implication for Practice The results suggest that children and adolescents with congenital hemiplegia with varying impairment of the hand may benefit from CI therapy, but not every child demonstrates improvements in hand function. The principal features of the best responders to CI therapy are not clear. However, it seems important to ensure that the training is intensive, with the duration of the program long enough and the content carefully selected so as to engage the participants. It is an enormous challenge for the participants in terms of effort and time; so the families and participants have to be prepared and well motivated for the program. Day camps may provide social and personal benefits for participants such as meeting new friends and learning a lot about themselves and their disability. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper. REFERENCES Bonnier, B., Eliasson, A. C., & Krumlinde-Sundholm, L. (2006). Effects of constraintinduced movement therapy in adolescents with hemiplegic cerebral palsy: A day camp model. Scandinavian Journal of Occupational Therapy, 13, Boyd, R. N. (2004). Central and peripheral effects of botulinum toxin A in children with cerebral palsy. Australia: La Trobe University. Bruininks, R., & Bruininks, B. (2006). Bruininks Oseretsky Test of Motor Proficiency: Second edition. Minneapolis, MN: Pearson Education. Charles, J., & Gordon, A. M. (2005). A critical review of constraint-induced movement therapy and forced use in children with hemiplegia. Neural Plasticity, 12, Charles, J., Lavinder, G., & Gordon, A. M. (2001). Effects of constraint-induced therapy on hand function in children with hemiplegic cerebral palsy. Pediatric Physical Therapy, 13, Charles, J. R., Wolf, S. L., Schneider, J. A., & Gordon, A. M. (2006). Efficacy of a child-friendly form of constraint-induced movement therapy in hemiplegic cerebral

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