Cerebral palsy (CP) is the most common neurologic condition seen by pediatric physical therapists (PTs)1 with recent studies reporting a prevalence rate of 3.6 per 1000.2 Physical therapy intervention for children with CP may include strengthening and task-specific training with the goal of helping them reach their full potential to participate in meaningful activities. In addition, PTs may recommend that children wear orthotic garments to control abnormal tone, stabilize posture, and improve function. Investigators evaluating the effects of orthotic garments in children with CP have reported increased confidence for attempting motor tasks,3,4 improved postural stability,3,5,6 improved gait kinematics,4 and some functional improvements.3,5,6 However, all of these investigators reported adverse effects from wearing the orthotic garments such as decreased respiratory function,3 heat and skin discomfort,4–6 and toileting difficulties.5,6 In addition, the majority of parents chose not to continue garment wear.5,6 In previous studies, the intervention consisted of wearing the orthotic garments 4 to 12 hours a day3–7 during typical daily schedules.
Recently, other types of orthotic garments or “suits” have been marketed (eg, TheraSuit, Adeli) that are to be worn, while participating in an intensive therapy program rather than during typical daily schedules. These suits incorporate bungee cords to provide resistance, and the makers claim the suit accelerates progress. One study reported the effects of an intensive suit therapy program on a sample of heterogeneous children with CP and found no difference between the intensive suit therapy group and children who received a neurodevelopmental approach with a similar intensity.8 A recent case report of 2 children with CP demonstrated minimal changes in motor function after an intensive suit therapy program with some positive changes measured with gait analysis.9 None of the investigators reported the effects of the suit itself.
Although evidence supporting intensive suit therapy is limited, families continue to seek these treatment programs for their children with the expectation of accelerating gross motor development even though they require significant resources. Aside from the intensity of the programs, several hours a day up to 5 days a week, the suits are expensive, time-consuming to don and doff, and uncomfortable. Damiano10 suggests “dissecting” programs such as suit therapy to examine the different components. In addition, Turner11 recommends a clinical trial in which both groups receive the same intervention except for the suit to determine its effectiveness. Further study is needed to determine whether using a suit is justified.
Therefore, the purpose of this randomized, controlled, single-blinded study was to determine the effect of wearing a suit during an intensive therapy program on motor function among children with CP, specifically to evaluate (a) the improvement of motor function, (b) to what extent the suit (TheraSuit) affects improvement in motor function, and (c) parent satisfaction with the intensive therapy program. This study was undertaken in response to multiple families who requested that a suit therapy program be offered at the authors' institution, in addition to therapists wanting evidence to understand this intervention. This study is the first to evaluate the effects of this suit on a group of similarly functioning children with CP, includes a control group receiving the same therapy with the exception of the suit, and uses assessors who are blind to group assignment.
Children were eligible to participate if they were between 3 and 8 years of age, had a diagnosis of CP, were classified as level III on the Gross Motor Functional Classification System (GMFCS),12 and had not previously participated in an intensive suit therapy program. In addition, the children had to be able to follow instructions and show no evidence of hip subluxation greater than 35% (migration index) and/or scoliosis greater than 25° (Cobb angle) on hip and spine x-rays, respectively, within 6 months of the start of intervention. The parent/guardian needed to speak and read English, and physician approval was obtained for each child to participate. Exclusion criteria were intrathecal baclofen pump therapy, history of selective dorsal rhizotomy, or Botox injections within the last 3 months, orthopedic surgery within the past year, serial casting within the past month, uncontrolled seizures, and a diagnosis of autism, attention-deficit disorder, other musculoskeletal diseases, progressive encephalopathies, and/or psychiatric or behavioral disorders.
Twenty children with CP aged 3 to 8 years were recruited between August 2005 and August 2007. Methods for recruitment included letters to parents of children diagnosed with CP receiving intervention at this institution, flyers to local parent support groups, and postings to relevant Web sites and magazines that parents of children with CP could access. All children who were recruited participated in the intervention at a 523-bed highly specialized quaternary care pediatric hospital and research center in the Midwest. This study was approved by the hospital's institutional review board and a parent of each participating child provided consent.
Participants were randomized to the experimental or control group by a computerized minimization program (Minimization software, Michael Conlon, Division of Biostatistics, Department of Statistics, University of Florida, Gainesville, May 1991). The minimization procedure controlled for potentially confounding variables, such as the patient's age, gender, and race. Age was categorized into 2 groups—3 years to less than 5 years, and 5 years and more. Therefore, systematic selection bias and chance skewing were reduced, and the 2 groups were comparable with minimized error variance of the covariates. See Figure 1 for study enrollment flow.
Pediatric Evaluation of Disability Inventory
The Pediatric Evaluation of Disability Inventory (PEDI) is a 197-item parental-report questionnaire that measures functional skills (FS) and caregiver assistance (CA) across the domains of self-care, mobility, and social functioning of infants and children aged 6 months to 7½ years. However, the PEDI can be used with older children if their abilities are less than expected for a 7½-year-old child without disabilities.13 In this study, the Self-care and Mobility domains within each scale were used, as it was expected that the intervention might change scores in these areas. For the FS scale, each item on the PEDI is scored as 0 (unable to perform) or 1 (able to perform), and a total score is obtained by adding the items.13 For the CA scale, the items are scored from 0 to 5 (0 = total assistance to 5 = completely independent). Internal consistency coefficients for the scales of the PEDI range from 0.95 to 0.99.13 Concurrent and construct validity of the PEDI has been reported.14 In addition, the PEDI is responsive to change in motor function of children with CP.15 The PEDI was scored according to test manual instructions and scaled scores were used in the analyses. In the current study, correlation coefficients for the PEDI over the 3 assessment times were 0.86 to 0.96, thus demonstrating good reliability.
Gross Motor Function Measure–66
The Gross Motor Function Measure–66 (GMFM-66) is a valid and reliable (ICC, 0.99)16 clinical evaluation tool that measures change in motor skills in children with CP.17 It consists of 66 items organized into 5 dimensions: (1) lying and rolling; (2) sitting; (3) crawling and kneeling; (4) standing; and (5) walking, running, and jumping. Item scores range from 0 to 3 (0 = does not initiate to 3 = completes). GMFM-66 total scores were calculated using the Gross Motor Ability Estimator computer program provided by the test developers, which converts item scores into a total interval level score. The GMFM-66 is sensitive to change in motor performance of children with CP over time18 and is commonly used in studies to evaluate effects of intervention in children with CP.19–22 In this study, correlation coefficients for the GMFM-66 over the 3 assessment times were 0.92 to 0.97, thus demonstrating good reliability.
Parent satisfaction was assessed by a nonstandardized investigator-developed questionnaire that was given to the parent at the last appointment. Frequencies were calculated and reported for parent responses to the following questions: (1) rate your child's level of comfort during the program (no discomfort, very minimal, mild, moderate, severe); (2) do you think wearing the garment helped your child? (yes, no, unsure); and (3) would you enroll your child in the intensive therapy program again? (yes, no, unsure).
All participants were assessed without the suit on, at baseline (3–10 days before the intervention), at 4 weeks (3–10 days after the intervention), and at 9 weeks (1 month after the intervention). During each assessment, the children were weighed and the GMFM-66 and the PEDI Self-care and Mobility domains were administered. Weight was measured at each time point using the same scale, because parents expressed concerns that the children might lose weight because of the program's intensity. Assessments were completed by 1 of the 2 PTs with 12 and 16 years of pediatric experience. The assessing therapists were blinded to group assignment and each child was assessed by the same therapist at all times. Parents and therapists were instructed regarding the importance of not discussing their thoughts on which group they believed the child was assigned. The therapists were trained and had used the measurement tools in the clinic prior to the study. In addition, procedures were reviewed prior to data collection and interrater agreement was established (ICCs: 0.97 GMFM-66, 0.99 PEDI) against a trained therapist (the first author).
Both groups received the therapy intervention for 4 hours daily, 5 days a week over a 3-week period. Physical therapists and occupational therapists employed by this institution provided intervention to both groups and were trained in the TheraSuit Method.9 Both groups followed the previously described activity sequence and description of the TheraSuit Method. During the intervention, the experimental group wore the TheraSuit with elastic bungee cords attached to the vest, shorts, kneepads, and shoes, as described in the training manual.23 The control group wore a “control suit,” which consisted of only the TheraSuit vest and shorts and did not have the elastic bungee cords attached. It was felt that the vest and shorts, which are made of canvas, did not provide added benefit when worn without the bungee cords and therefore could be the control. Each child's intervention was individualized to the goal of achieving the next functional activity level. Therapists maintained a daily log of all activities/exercises completed, behavior, rest breaks, and any adverse safety events. At the end of the 3-week intervention, each child was given an individualized home exercise program to perform not more than 1 hour daily from weeks 4 through 9. The participants did not receive any other direct occupational therapy or physical therapy services for the duration of the study protocol. Parent satisfaction with the program was assessed at the last assessment via a nonstandardized questionnaire.
Descriptive statistics were used to summarize the sample demographics and outcome variables. Student t tests and exact tests were used to compare demographic and baseline measurements of the experimental and control groups. Linear mixed models for repeated measures were conducted to (1) check differences in mean GMFM-66 and PEDI scores over time within and between groups; (2) calculate effect sizes between groups for GMFM-66 and PEDI outcomes; and (3) evaluate the change in weight for each group over the 9 weeks. Also, intention-to-treat analysis was considered. Tukey pairwise adjustments were used for all significant variables. Effect sizes were interpreted as follows: small, 0.20 to 0.50; moderate, 0.50 to 0.80; and large, greater than 0.80.24 Power estimates were not performed prior to data collection because it was not known how much change to expect with this intervention. Therefore, from this study, one could calculate effect sizes for the outcome measures and inform future studies. All the analyses were conducted using Statistical Analysis System (SAS 9.1). A 2-sided significance level was set for α < 0.05.
Demographic and baseline characteristics of the sample are presented in Table 1. No significant differences were found between groups at baseline with regard to age, gender, race, weight, and initial GMFM-66 and PEDI scores. Mean GMFM-66 and PEDI scores for both groups are presented in Table 2 across the 3 time points. All scores were observed to improve across time.
Between-group P values and effect sizes were calculated and are presented in Table 3. After controlling for age, gender, race, and baseline scores, no statistical significant differences were found between groups on the GMFM-66 or any domains of the PEDI. A small negative effect size was found for the PEDI CA Mobility domain. All other effect sizes were small and positive.
Results of the linear mixed modes for repeated measures within each group showed a significant difference for the control group on the GMFM-66 at week 9 versus baseline (see Table 4). The experimental group demonstrated significant differences for 4 of the outcome measures, GMFM-66, PEDI FS self-care, PEDI CA self-care, and PEDI FS mobility.
Mean weight for the experimental group was 17.27 kg (SD = 4.53) at baseline, 17.54 kg (SD = 4.76) at week 4, and 17.93 kg (SD = 4.91) at week 9. For the control group, mean weight was 17.19 kg (SD = 4.59) at baseline, 17.49 kg (SD = 4.68) at week 4, and 18.23 kg (SD = 4.76) at week 9. Weight significantly increased across all time points for both the control group and the experimental group, F 3,16 = 47.31 and F 3,16 = 49.43, P < .0001.
One child in the control group was not able to complete the intervention and withdrew at day 12 because of combative behaviors that posed a safety risk. However, the child attended both follow-up assessments and the data were used in the analysis.
No serious safety events were encountered or occurred during the study such as fractures, hip dislocations, or skin abrasions.
Nineteen questionnaires were returned at the end of the study. Ten of these were from parents whose child was in the experimental group and 9 were from the control group. All parents reported that their children experienced some level of discomfort during the program. For example, 7 parents (4 control and 3 experimental) reported minimal discomfort, 7 parents (3 control and 4 experimental) reported mild discomfort, 3 parents (experimental) reported moderate discomfort, and 2 parents (control) reported severe discomfort. When parents were asked whether they thought wearing the suit helped their children, 4 parents in the control group said no and 5 parents in the control group were unsure. In contrast, 3 in the treatment group were unsure and 7 in treatment group answered yes. Also, 17 parents reported that they would enroll their children in the program again and 2 were unsure (1 in the control group and 1 in the treatment group). The 2 parents who were unsure also commented that the program was too intense.
The results from this study demonstrate that children with CP who wore the TheraSuit with attached bungee cords during an intensive therapy program did not increase function more than children who wore a control suit (TheraSuit vest and shorts) during the same intensive therapy program. This was the first study to examine the different components of suit therapy and study the effects of the suit itself. Strengths of this study include the following: subjects were randomized to an experimental or control group; all children were of similar functional level (GMFCS III); the age range of the children was narrow; and the assessors were blinded to group assignment.
The effect sizes obtained in this study were positive and in the small range with the exception of a negative small effect size for PEDI CA mobility. The negative effect size for PEDI CA mobility suggests that the control group did better than the experimental group after intervention on the CA scale. This is may be due to the greater variation in the experimental group scores on the PEDI CA Mobility domain, which has been reported elsewhere in the literature.6 An alternative explanation would be that the difference is real, and that the experimental group needed more assistance with mobility skills following the intervention.
Significant within-group differences were found in this study, which is similar to a recent randomized clinical trial of a cycling intervention in children with CP,25 where no significant between-group differences were found but significant differences were found within the experimental group. Intrasubject variability, even within the same GMFCS level, may make it difficult to detect between-group statistical significance. Therefore, future studies may choose multiple baseline sessions as a part of studies to evaluate interventions in children with CP.
Comparison among our findings and other studies is difficult because no other studies have investigated the effects of the TheraSuit alone. Our findings are similar to those reported by Bar-Haim,8 who compared a similar intensive program with a similar suit to neurodevelopmental treatment in children with CP. Both groups in the Bar-Haim study showed significant changes in the GMFM-66 1 month following the intervention. Like the Bar-Haim study, our study suggests that intensity is a principal factor in improvement. Bar-Haim examined the children again at 10 months when the changes were not maintained. Our study followed the children for only 1 month and the children continued to show improvement. Longer-term follow-up is needed to determine whether children's motor skills are maintained over time following this intervention.
Comparison between our findings and previous studies of orthotic garments is also difficult for several reasons. The previous studies4–7 used a variety of garment types, garments were often worn at home for longer periods of time during each day with or without intervention being performed, and postassessments were performed with the garment on after achieving several hours of wear time. Some previous investigators reported improvements in GMFM scores,7 stability,6 and PEDI scores,5 whereas others3,4 with postassessments that included both wearing and not wearing the garment reported some improvements in both conditions using varied outcome measures. The procedure of the TheraSuit Method includes suit wear during the therapy intervention and not necessarily at other times during the day. In addition, postassessments in this study were completed with the suit off. We did not find any differences in GMFM-66 or PEDI scores between groups, suggesting that the suit itself did not contribute to the gains made.
Results of this study are similar to those of studies of other garments that reported general discomfort and difficulty with wear.5–7 However, in the current study, most parents in both the experimental and control groups reported that their children had some level of discomfort during the program. This finding suggests that at least some of their discomfort might be due to the 4-hour length of each session and 5-day-a-week frequency, as opposed to suit wear.
This study has several limitations related to the small sample size and lack of blinding of families and treating therapists. Strict enrollment criteria to ensure a homogenous group also limited our sample size. Although we did not tell the families which suit was the experimental suit, families verbalized knowledge of the TheraSuit and/or TheraSuit Method or sought information outside of the study; therefore, we were not able to assume that parents were blinded to group assignment. This most likely biased our PEDI results since PEDI is a parent report. In addition, the parent satisfaction survey was not validated and may not have been comprehensive enough to determine the benefits perceived by the families.
The GMFM-66 and PEDI are functional measures and may not detect changes in gait, quality of movement, or energy efficiency, which may be important areas to consider. In a recent case study, 2 children who participated in the TheraSuit Method showed improvements in gait.9 The outcome measures used for this study also did not account for changes in assistive devices. For example, as reported by a treating therapist, one child transitioned from a walker to a single tripod cane and one child transitioned from a walker to forearm crutches. Although the experimental group was not shown to produce greater functional benefits than the control group, therapists reported that they preferred using the suit because it gave them more “hands” to achieve better alignment during activities.
This study could have been strengthened if a control group of children that did not participate in any therapy were included in the comparisons. Finally, results from this study cannot be generalized to children with CP of other ages, other GMFCS levels, and/or in combination with other interventions such as Botox or casting.
This study does not provide statistical evidence that the use of the TheraSuit improves motor function during intensive therapy programs more than an intensive therapy program wearing a control suit for children with CP classified as GMFCS level III. Future studies may examine postural changes and gait efficiency with and without suit wear in children with CP of varying functional abilities. Single-subject studies with multiple assessments may be able to detect changes that this study could not. Until further studies are completed, therapists should use this information to guide families who are considering participating in intensive suit therapy programs.
The authors thank the following for their contributions: families and children who graciously agreed to participate in this study; intervention therapists Shannon Brausch, PT, Michelle Menner, MPT, Shannon Teeters, OTR/L, Russell Garrision, OTR/L, and Emily Owens OTR/L; blinded assessors Cathy Lowe, PT, and Terri Dinkelaker, PT; administrative support at intervention site: Michelle Kiger, OTR/L; soft knee splints for participants fabricated by the CCHMC Sewing Room; equipment purchased by the Smiles for Kids Foundation; and 2 TheraSuits donated by TheraSuit LLC.
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Keywords:© 2011 Lippincott Williams & Wilkins, Inc.
ADL; cerebral palsy; cerebral palsy/classification; cerebral palsy/rehabilitation; child; child/preschool; clothing; patient satisfaction; physical therapy/modalities; movement; psychomotor performance; randomized control trial; treatment outcome