Growth spurts are normal events in adolescence,1 but for an adolescent with spastic diplegic cerebral palsy (CP), the combination of accelerated growth rates and spasticity is problematic and a recurrent challenge to daily functional activities and mobility. In adolescents with CP, muscle groups surrounding the hips, knees, and ankles are at greater risk of shortening and restricting movements of the joints during accelerated growth.2 Without intervention, contractures may develop affecting the alignment of joints and consequently posture and may result in impaired balance, coordination, endurance, and gait making movement more difficult.2
When applying the International Classification of Functioning, Disability and Health (ICF) model to an adolescent with spastic diplegic CP with accelerated growth, the primary focus is on enabling the adolescent to fulfill roles of daily life. The ICF includes 3 domains: (1) body functions and structures, (2) activity, and (3) participation.3 To monitor treatment and family satisfaction outcomes, measures for each of these domains should be linked to the goals set by the adolescent and family.4
Recently, investigations into the value of traditional and innovative therapies, including strengthening in functional positions, task-related training,5 and hippotherapy,6 in children with CP have been reported. Traditional therapies and hippotherapy incorporating task and strength training have demonstrated positive effects on body structure and function and activity,5–8 with hippotherapy also demonstrating positive outcomes with respect to participation restrictions.9
The American Hippotherapy Association defines hippotherapy as a treatment strategy, used by physical, occupational, and speech language therapists, using the movement of the horse to address body structure and function, activity limitations, and participation restrictions in patients.10 Hippotherapy is recognized by the American Physical Therapy Association as an appropriate neuromuscular and therapeutic exercise activity for treating persons with neuromuscular disorders.11
Taking place in a nonclinical environment with outdoor and/or indoor arenas, hippotherapy incorporates multiple systems (ie, sensory, limbic, musculoskeletal, vestibular, and ocular) simultaneously in an open and unpredictable task that allows for mass practice. This benefits the child with internal feedback for carryover and problem solving in social, psychological, and educational situations across multiple settings.12 This experience of horse and human interaction is a powerful motivator for participation in therapy, which enhances the possibility of achieving the functional goals set by the adolescent and interdisciplinary team.10 Debuse et al9 found that hippotherapy increased self-efficacy, confidence, and self-esteem, which carried over into increased function in persons with CP.
In addition to psychological benefits, hippotherapy has displayed significant positive effects on impairments of body structures and activity limitations in children with CP.5–8 Hippotherapy research on children with CP has demonstrated improvements in balance and performance in daily activities,7 head and trunk stability, upper extremity reaching,13 postural control14 alignment of the pelvis, symmetrical hip movement, and tone reduction.8,15
To improve alignment during task-related training, therapists have used compression belts.16 The belts are used to provide increased tension at the pelvic region in an effort to enhance the sense of position and movement awareness in the joints surrounding the trunk, pelvis, and lower extremities (LEs).17 The design of some compression belts allows for the addition of weights at the pelvic region, to promote strengthening during functional task training. Information on the effectiveness of compression belts for alignment, with the addition of weights for strengthening, is antidotal, with no research on the efficacy of using them in combination. No studies could be identified that assessed changes in adolescents with spastic diplegic CP Gross Motor Function Classification System level II (GMFCS level II)18 during a period of accelerated growth. The purpose of this case report was to describe an adolescent's improvements across ICF components after the combined physical therapy (PT) interventions provided at home and through hippotherapy using a weighted compression belt.
DESCRIPTION OF THE CASE
Written consent/assent was obtained from the mother and adolescent to participate in this case report and to access his medical records.
The boy was 13 years old and cared for by his adoptive parents. His mother reported a possible growth spurt and was referred to PT by his pediatrician for evaluation and treatment to include hippotherapy. Other than being a twin and the fact his spastic diplegic CP was caused by cortical abnormalities and dysplasia, his birth history was unknown. His functional level was consistent with GMFCS level II.18 Medical records indicated a surgical history of selective dorsal rhizotomy at age 4 years, with postoperative spinal meningitis quickly resolved after antibiotic therapy. He wore prism glasses to correct right eye strabismus and reported seeing well with glasses. At the time of this intervention and over the prior 4 years, he took 200 mg of Provigil once daily to decrease drooling,19 spasticity, and improve gait.20 From age 4 to 13 years, a biweekly PT program of home therapy and hippotherapy led from ambulation with a posterior walker to walking without an assistive device on all surfaces in the home and community. He was discharged 6 months before this intervention period and received a once weekly home stretching and exercise program with a personal trainer. Medical equipment included bilateral arm splints and custom dynamic ankle foot orthoses (DAFOs), which had not been used for the past 6 months because of time constraints and questions of benefits, respectively.
The boy lived in a 2-story home with his parents and twin sister. He attended a regular high school and exceled in academics. During the school day, an assistant helped him negotiate the busy hallways, carry his books, and maintain his schedule. At the time of his initial visit, his mother reported a possible growth spurt, and noted increased pulling on the handrail to ascend and descend the stairs in the home, increased crouched posture in standing, and inability to rise from the floor without assistance.
Cardiopulmonary, Musculoskeletal, and Integumentary Observations
The September 24, 2013, baseline assessment and systems review revealed that his cardiopulmonary status was within normal limits for his age (Table 1) as were the gastrointestinal, urogenital, and endocrine systems. His height was 52 inches, revealing a height difference of 1 inch from his previous height measurement taken on August 17, 2012. His weight was 61 lb. His body mass index was 15.89. This body mass index was between the fifth and 10th percentiles for boys that are typically developing21 and between the 10th and 25th percentiles for boys with CP GMFCS II.22 With the exception of a well-healed 1.5-inch scar in the central lumbar region from his dorsal rhizotomy, skin integrity and color were not impaired. The adolescent denied any joint or muscle pain or discomfort.
Transfers from stand to sit to stand were accomplished without use of hands. He demonstrated a crouched position in standing, which he held independently for 30 seconds before moving to maintain balance. He walked on level surfaces in and outside of the home without physical assistance with both upper extremities (UEs) in flexion at his chest. He required moderate assistance to stand from the floor. Minimal decreased coordination was observed in the left UE compared with the right UE (Table 2). Sensation was intact to light touch throughout with no apparent ankle clonus or hyperreflexia noted.
Communication, Affect, Cognition, Language, and Learning Style
He was alert and oriented to person, place, and time. His oral motor control and verbal communication were impaired as exhibited by decreased management of oral secretions and difficulty with word pronunciation. He was motivated to participate in hippotherapy. His preferred learning style was to read written instructions for schoolwork and computer games and use visualization and mental practice when he learned a new physical skill.
Body Structure and Function Measures
The Modified Ashworth Scale (MAS) was used to measure spasticity (Table 2). The MAS has adequate to excellent test-retest reliability for hamstrings with an intraclass correlation coefficient (ICC) of 0.66 to 0.80, and adequate to excellent for hip adductors (ICC, 0.59-0.82).23 Although the MAS has limited reliability and validity, the MAS may be useful to measure muscle tone changes during strengthening programs.24
The reliability of goniometric measures is variable in children with CP.25 Goniometric measurements revealed that UE and LE passive range of motion (PROM) was within normal limits. Limitations were measured at his elbows, hips, and knees (Table 2). Functional mobility strength was assessed in place of manual muscle testing because of minimally increased tone and lack of isolated movement throughout.26
The 1-Minute Walk Test (1-MWT) was used as a measure of physiological health and to assess body function. It is a quick, reliable,27 and valid28 test in children with bilateral spastic CP. The 1-MWT test-retest reliability (ICC) of 0.9427 makes it a useful tool for repeated assessments. A fast 1-MWT has been shown to correlate to function (r = 0.92) in children with bilateral spastic CP who are ambulatory.28 The physiological cost index (PCI) is a valid measure of endurance in children with CP.29 Using a finger-tip oximeter and blood pressure cuff, direct measures of O2 saturation, heart rate, and blood pressure were taken before and after the 1-MWT to calculate the PCI. Speed and distance were measured with an iPod stopwatch and tape measure with the test repeated after a 5-minute rest.
The Pediatric Balance Scale (PBS) was used as a measure of balance. The PBS is designed for children aged 5 to 15 years with mild to moderate motor impairments and has a high test-retest reliability (ICC [3,1] = 0.998) and inter-rater reliability (ICC [3,1] = 0.997).30
The Timed Up and Down Stairs (TUDS) was used to measure activity in life situations. The TUDS measures functional mobility strength and balance during the course of PT.31 The TUDS is a reliable test for children with CP and demonstrated both high intrarater and interrater reliability (ICC [2,1] = 0.99) and test-retest reliability (ICC [2,1] = 0.94).31 The task was timed in seconds with an iPod stopwatch as the boy ascended and descended a 14-step flight of stairs with 1 handrail as instructed in the TUDS. Each step measured 7 inches in height, and between the 2 flights of stairs a 180° turn was required where the steps narrowed at the axis.
The Pediatric Evaluation of Disability Inventory Computer Adaptive Test (PEDI-CAT)32 was used to measure activity and participation. The PEDI-CAT measures the 4 domains of Daily Activities, Mobility, Social/Cognitive, and Responsibility from birth to 20 years of age.32
The Content-Balanced (“Comprehensive”) CAT version was chosen because the 30 items per domain provides normative scores, scaled scores, and item maps to monitor patient progress.33 The PEDI-CAT Mobility Domain has been shown to correlate with the original PEDI Functional Skills Mobility Scale (r = 0.82) for concurrent validity (ICC = 0.3390-1.000) and agreement of 60% to 100% for 8 specific items in children with CP.34 The PEDI-CAT was completed through interview of the mother by the therapist at baseline, 5, and 10 weeks.
The Dimensions of Master Questionnaire (DMQ 17), designed to assess the outcome of persistence in mastering a task,35 was used to measure participation and motivation in life situations. The DMQ 17 consists of a questionnaire rated separately by the adolescent and parent and scored on 3 scales: instrumental, expressive, and general competence. The DMQ 17 has internal consistency alphas .70 or more for all items on the instrumental and expressive scales in adult and adolescent versions for CP.35
PROGNOSIS, PLAN OF CARE, AND GOALS
On the basis of the examination, which revealed impairments of PROM, strength, balance, functional activities, mobility, and gait, participation in weekly sessions consisting of 1 hour of home-based PT and 1 hour of hippotherapy for a 10-week period (2 hours per week, 20 hours total) was recommended for the boy. The plan of care was designed to improve functional activities and mobility to his previous level of function in GMFCS level II. His plan of care also included once a week home stretching and strengthening program with a personal trainer. The PT-directed home exercise program was initiated with his mother to reestablish the use of UE splints and DAFOs for preventative positioning, and home balance activities: standing at the computer desk, negotiating the 6-inch step in and out of the home, ascending/descending 2 flights of stairs without use of the handrail, and transfers up from the floor. The mother and boy participated in the development of short-term goals (Table 3).
DESCRIPTION OF INTERVENTION
The 10 weeks of therapy occurred between September and November 2013. In weeks 7 and 8, the adolescent missed his hippotherapy sessions because of illness of family members. These were rescheduled for week 10. The physical therapist consistently provided the interventions and collected all the measurements at baseline, 5, and 10 weeks. The Appendix (Supplemental Digital Content 1, available at http://links.lww.com/PPT/A94) provides the activities and progression of the interventions for both settings. Treatment in the home environment focused on improved functional activities, mobility, strength, balance, and walking in daily life situations. The intervention of hippotherapy focused on stretching for improved PROM, therapeutic postural responses, and core balance-strengthening activities for carryover across different settings.
Integration of a compression belt with progressive weight at the sacral region was used in both home and hippotherapy interventions. The Com-Pressor Belt (OPTP, Minneapolis, MN) was placed around the pelvis below the ilia and across the sacral region, above greater trochanters and below anterior superior iliac spines (Figures 1A and 1B). This allowed for progression of core and gluteal muscle strengthening in alignment during exercises in both interventions (Figure 1D).16
Each home PT session consisted of learning a new functional skill in his own environment. This was accomplished by breaking down a task into parts, then advancing to performance of the whole task. Each session included floor-to-stand transfers, gait training on level or uneven surfaces and ascending or descending a 6-inch step and 2 flights of stairs with tactile cues for facilitation or inhibition. This was performed with explicit learning and repeated distributed practice. For example, as he advanced his skills, he was able to merge the parts and complete the whole functional task with increased weight attached to the Com-Pressor Belt and fewer tactile and verbal cues. He was also instructed in mental practice during the home program with his mother because this was one of his methods of learning. These problem-solving abilities were generalized to different environments.
The hippotherapy intervention was carried out by the same physical therapist who was a Registered Therapist with Professional Association of Therapeutic Horsemanship International (PATH, Intl.) and member of the American Hippotherapy Association. A trained hippotherapy horse with symmetrical 3-dimensional movements and a barrel measuring 18 inches wide was selected. The horse equipment included a soft vaulting pad, Anti-Cast Roller Web surcingle with 1 hoop (Figure 1C), halter, reins, and lead rope. The boy wore an American Society for Testing and Materials-approved helmet.
The horse was led in the arena by an experienced horse handler, and the adolescent was assisted or supervised by the physical therapist and an experienced side walker for safety. The handler was directed by the therapist to adjust the movements of the horse while walking to affect the intrinsic feedback received by the adolescent. For instance, each of the positions of quadruped, tall kneel, half kneel, and standing on the 3-dimensional moving support surface of the horse was accompanied by additional visual and vestibular challenges. This could be misleading to the adolescent's proprioceptive and visual input and may lead to both inputs being reweighted in order to maintain postural control with noted increased ankle strategies.36 As these strategies advanced, increased weight was applied to the Com-Pressor Belt and he required fewer tactile and verbal cues. Gait training was performed after the hippotherapy session, with emphasis on taking advantage of the increased range of motion and motor activation as he walked to the car.
DESCRIPTION OF OUTCOMES
Because the 1-MWT and TUDS were timed speed tests that could be affected by fatigue, they were performed before the treatment session, whereas all other measurements were taken postsession. Although his DAFOs were reestablished into his daily life, the adolescent was not wearing his DAFOs at baseline measure. Therefore, for consistency, DAFOs were not worn during the 5- and 10-week examinations.
Body Structure/Function and Activity
The MAS score of 1 remained the same, and PROM increased throughout the interventions (Table 2). By the 10th-week assessment, his functional reach had increased 1 inch, which allowed him to reach the handle and close the door without assistance while sitting in the car. His overall increased PROM was demonstrated as he functionally reached higher from a standing position into the refrigerator to retrieve a drink or snack.
The 1-MWT demonstrated a significantly decreased heart rate by 0.17 bpm at 5 weeks and 0.26 bpm at 10 weeks. The decrease in the PCI of 0.26 bpm during the walk test did meet the minimal detectable change (MDC) of 0.22 m/s for GMFCS I and II (Table 1).37 By week 10, his increased endurance, as measured by PCI values, was reflected by the adolescent who exceeded the goal of standing at the computer station for 1 hour while using the keyboard and the mouse (Figure 1B). He was able to maintain straight alignment of hips and legs in unsupported standing during conversations with his mother, standing in lines and participating in community outings with his family including walking for 1 to 3 hours. His 1-MWT distance (Figure 2A) did not meet the MDC of 13 m for GMFCS levels I, II, and III,28, nor was his 0.012 m/s change in gait speed significant.
Timed scores of the TUDS from baseline measurement increased 1 second at 5 weeks, but decreased by 9.6 seconds at 10 weeks (Figure 2B). The TUDS has been found to be an independent predictor of community ambulation in children with CP who walk without support. Children at GMFCS level II/III have reported speeds of 24.5 seconds (standard error of the mean = ± 3.83). Based on these data, this boy would not have demonstrated community ambulation, with scores of 31 seconds at baseline and 32 seconds at week 5 (Figure 2B).31 However, at week 10, although he did not reach the cutoff for community ambulation,31 he performed the TUDS in 21.4 seconds, which correlates with a classification of GMFCS level II/III.31 This is consistent with his functional classification of GMFCS II as ambulatory without an assistive device and an indicator of his increased functional strength.31 This is in direct contrast to his 10 week 1-MWT test distance, which suggested his GMFCS level as III.28 This demonstrates that although there is some correlation between TUDS scores, 1-MWT distances, and GMFCS classifications, not all adolescents, especially during growth spurts, will perform according to predictions. Therefore, therapists need to use all information before classifying GMFCS levels in adolescents.
His static PBS score improved by 5 points, with a score of 14 at baseline and 19 at week 10, whereas the dynamic score increased by 3 points (baseline score = 22; week 10 = 25), for a total 8-point improvement after 10 weeks of treatment (Figure 2C). Because MDC values have not been established for 13-year-old youth with CP on the PBS and children who are typically developing reach the ceiling score by age 7 years,38 Chen et al's39 recently validated PBS MDC and minimal clinically important differences (MCID) in children 1.6 to 6.4 years old with CP were used for comparison. The established PBS from baseline to follow-up for static, dynamic, and total values of MDC was 0.79, 0.96, and 1.59, and the MCID ranges were 1.47 to 2.92, 2.23 to 2.92, and 3.66 to 5.83, respectively.39 The fact that the adolescent's increase in PBS scores exceeded the MDC and MCID ranges could be considered clinically relevant (Figure 2C).
Generally, his PEDI-CAT daily activity and responsibility domain-scaled scores had the most improvement between weeks 5 and 10 (Table 4). Using the 95% confidence interval for these domains, the 2- and 5-point improvement of his scaled score in daily activities from baseline to 5 weeks and 10 weeks, respectively, is significant. A clinically significant change was also seen in the responsibility domain at 10 weeks (56) in comparison to his baseline (48) and 5-week scores (50).32 The mobility domain had a 2-point improvement at 5 weeks, whereas the social/cognitive domain improved the most between baseline and 10 weeks (6 points), but these improvements were not clinically significant.
Both the adolescent and his mother self-reported on their separate questionnaires improvements in all domains of the DMQ 17 after the intervention. The adolescent reported the most improvement in social persistence with other children at 10 weeks (3.0 points), and the mother observed the greatest improvement in gross motor persistence (1.6 points) (Table 5). The adolescent's DMQ 17 baseline self-report score for gross motor persistence was 0.7 greater than his mothers. This difference might be explained as the adolescent's perception of working hard at a challenging task, whereas his mother perceived he had low motivation for mastering challenging tasks. However, at 5 and 10 weeks, they were in closer agreement, which could suggest the adolescent's improved strength, endurance, and balance translated to his ability to persist with tasks that his mother interpreted as improved motivation. More importantly, these clinical changes manifested in behavioral changes. After 8 weeks of treatment, his mother shared that he exited the car himself, retrieved a 32-ounce bottle of activity drink, and successfully carried the bottle from the car into the house by ascending the 6-inch step at the entrance. At the end of the 10 weeks, his outward negative reactions to failure, which included folding his arms and refusing to continue to participate in a challenging task, had decreased and evolved into problem-solving techniques that correlated to his increased gross motor persistence scores and decreased negative reaction scores. Increase in social persistence with other children was demonstrated when the adolescent saw a flyer in school to sign up for a chess club. He retrieved the information himself, brought the information to his mother, and asked to participate in the chess club. His mother noted informally with excitement that he had never done any of these tasks independently (Table 5).
Before this 10-week intervention period, the adolescent was in a maintenance program with a personal trainer. His mother observed a sudden decline in function and asked for a referral to PT because the personal training program was no longer working. During the examination the physical therapist noted that the adolescent had grown 1 inch over the prior year, and determined this might have been 1 of the reasons his maintenance program was not working. The growth spurt may have caused him to be less functional in daily activities, resulting in the need for PT intervention. Because the adolescent liked working with the personal trainer, he continued with the weekly maintenance program during this 10-week PT intervention period.
This case report demonstrates that a growth spurt can be a critical period for PT intervention. A physical therapist's understanding of the effects of tight, spastic muscles on body structure and function, activity and participation allows the therapist to create an eclectic intervention program that is not only engaging for the adolescent, but also encourages compliance. Design of an intervention plan combining home PT, hippotherapy, a weighted compressor belt, and a home program, and continuation of a personal training program were successful in this case. After 10 weeks of a combination of home and hippotherapy, this 13-year-old adolescent improved in all domains of the ICF. More importantly, he met all the short-term goals set by him and his mother in conjunction with the physical therapist.
One of the rationales for using hippotherapy for this case was that equine movement allows for decreased tone and less asymmetrical spasticity,8 which may make stretching more effective and less uncomfortable than standard stretching methods. Another characteristic of motor weakness in CP is the inability to isolate muscle groups because of co-contraction.40 As this co-contraction is taking place, the intervention of hippotherapy permits the therapist to include stretching in conjunction with strengthening in alignment with the Com-Pressor Belt.16,17 The reason for using the Com-Pressor Belt with progressive weight added to the sacral region was to increase the power of the weak antagonist muscles with the corresponding spastic agonist muscles in the trunk, pelvis, and LEs.16,17,40 His increased daily activity PEDI-CAT-scaled score from baseline to week 10 (Table 4) suggests that improvements in trunk, UE and LE strength, active and PROM, and functional activities may have led to improved social skills and responsibility, even though the social skills improvement was not considered clinically significant.
Studies have suggested that it is not only the passive stretching and active strengthening program performed while sitting astride the barrel of the horse, but the rhythmical movement that improves symmetry and activity in agonist and antagonist muscle groups in children with spastic CP.8 Damiano et al40 found that when there was more muscle activity in both agonist and antagonist muscle groups in children with spastic CP they demonstrated more energy efficiency. One of the key findings revealed in this case report was the increase in energy efficiency confirmed by the significant change in PCI scores of 0.60 bpm at baseline to 0.34 bpm at 10 weeks (Table 1). This finding suggests the use of equine movement may have contributed to the positive improvement in his PCI scores. In addition, these changes were taking place in a motivating, nonclinical environment where the adolescent was actively participating in fun and challenging activities for successful carryover into functional activities in the home environment. Combining PT in the home and hippotherapy reinforced gains of PROM, strength, balance, and endurance for use in daily activities.
As in clinical practice, there are many variables operating in this case study. Although the personal training component was separate from the PT intervention, the continued sessions could have influenced the outcome measures, and improvements in PROM, strength, balance, and endurance may have improved the outcomes of the training sessions.
Because of time constraints, the adolescent only wore his arm braces 1 hour per week and declined use at night because of sleep disturbances. This could have increased PROM in his UEs and led to changes in outcome scores, such the PEDI-CAT that required UE use for some items. There were also varying intensities in the hippotherapy treatment over the last 4 sessions, especially the 10th week where the adolescent participated in 1 home therapy and 3 hippotherapy sessions. The 10th week could be considered intensive therapy and possibly had an influence on the outcome measures.
Future Implications and Conclusions
This case report has demonstrated the effects on body function/structure and activity of short-term intensive PT using a combination of home-base and hippotherapy, and how these improvements led to this boy's ability to fulfill his roles of daily life. In the future, researchers could examine changes in PCI scores, the underlying motivational factors of hippotherapy and PT interventions, and participation in these interventions for adolescents with CP transitioning to adulthood.
The authors thank the staff from Rehab Essentials in collaboration with the University of South Florida transitional DPT program for making this case report possible.
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