C A S E
R E P O R T
Daily Intervention for Young Children With Cerebral Palsy in GMFCS Level V: A Case Series Jill C. Heathcock, MPT, PhD; Kathleen Baranet, PT, DPT; Rachel Ferrante, PT, DPT; Sarah Hendershot, PT, DPT Division of Physical Therapy (Dr Heathcock), The Ohio State University, Columbus, Ohio; Center for Perinatal Research (Dr Heathcock) and Department of Clinical Therapies (Drs Baranet, Ferrante, and Hendershot), Nationwide Children’s Hospital, Columbus, Ohio.
Purpose: To describe a daily physical therapy (PT) intervention program and outcomes for 2 young children with spastic quadriplegia, Gross Motor Function Classification System (GMFCS) level V, and to evaluate the feasibility of using a daily program in an urban children’s hospital outpatient setting. Summary of Key Points: Two young children, GMFCS level V, received 2 hours of PT intervention based on motor learning principles 5 days a week for 4 consecutive weeks. Gross Motor Functional Measure (GMFM-66, GMFM-88) and the Bayley Scales of Infant Development, Third Edition, were used as pre- and postoutcome measures. The daily, high intensity intervention was well tolerated. Improvements in motor function, language, and cognitive skills were found. Statement of Conclusion: A daily PT program appears feasible and may improve overall development in young children with cerebral palsy in GMFCS level V. (Pediatr Phys Ther 2015;27:285–292) Key words: case report, cerebral palsy, child development, human, infant, female, male, outcomes assessment, physical therapy/methods/administration and dosage, quadriplegia
INTRODUCTION Children with spastic cerebral palsy (CP) account for 80% of the CP population, with 15% estimated to have spastic quadriplegia.1-3 Children with spastic quadriplegic CP typically have very limited mobility and are classified at higher Gross Motor Function Classification System (GMFCS) levels.4,5 The GMFCS is an age-based classification scale for describing motor abilities in children with CP from birth to 18 years old. The classification levels, I through V, indicate least severe to most severe impairments of motor
0898-5669/110/2703-0285 Pediatric Physical Therapy C 2015 Wolters Kluwer Health, Inc. All rights reserved. and Copyright Section on Pediatrics of the American Physical Therapy Association
Correspondence: Jill C. Heathcock, MPT, PhD, Division of Physical Therapy, The Ohio State University, 453 W. 10th Ave, Columbus, Ohio 43214 ([email protected]). The authors declare no conflicts of interest. At the time this article was written, Sarah Hendershot, PT, DPT, was a pediatric physical therapist resident at Nationwide Children’s Hospital and The Ohio State University. DOI: 10.1097/PEP.0000000000000149
Pediatric Physical Therapy
function, respectively.4-6 For example, children with CP classified as GMFCS level V typically do not have independent forms of mobility and are dependent on caregivers for most aspects of daily life. In infancy, children classified at level V have limited voluntary control and antigravity movements and cannot roll or sit independently.4-6 Perhaps as a result of more severe delay and lower skill level expectations, they are infrequently included in intervention studies, especially those with high intensity protocols. When children in GMFCS level V are included in intervention studies, they are commonly grouped with children in levels III to V to create a nonambulatory group. Children in GMFCS level V have unique motor skill development compared with other children with CP and show a plateau in motor skill development earlier than children in other GMFCS levels.6 The motor development curves described by Rosenbaum et al6 show the spread of gross motor skills between GMFCS level V and other GMFCS levels is closest from 0 to 3 years of age. Children in all 5 levels continue to make progress from 0 to 3 years of age, possibly indicating a window of opportunity to promote optimal future development. Direct interventions aimed at making meaningful changes in motor skill development during this time period may be justified.7 Nonefficacious intervention and service delivery means Intensive PT for Young Children With CP—GMFCS Level V 285
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
that the children with CP who are most seriously impaired may miss critical years for intervention when brain plasticity including formation of new neural pathways, and growth and development of synaptic formations may result in the most potential to change motor function. Importantly, children classified in GMFCS level V tend to remain in that level over time, and younger children in level V who are reclassified are more frequently classified to a lower functional level.8 It is unknown whether higher intensities of intervention at younger ages can improve GMFCS classification. These results support the need to provide effective interventions at a young age to optimize function and determine whether function is within or above the expected trajectory. Children in GMFCS level V often demonstrate multiple impairments and show delays and disabilities in other domains of development such as communication skills,9 language skills,10 self-determination,11 cognition, behavior, and social function.12 They show these delays and disabilities at a higher rate and severity than children who are typically developing and those in higher functioning GMFCS levels. Furthermore, early skill development, such as head control and independent sitting, influences how children perceive their world and interact with their environment and caregivers. Children with major motor atypicalities have less experience independently moving their bodies against gravity and exploring their environment. This limitation affects not only their immediate gross and fine motor, language, cognitive, and social skills, but also their developmental trajectory.13 Distribution and Dosing of Physical Therapy The delivery of physical therapy (PT) services for children with CP including frequency, intensity, and time varies greatly depending on the region in which the family resides and the environment in which the services are provided. These features of dosing may also be dependent on the type of intervention, the child’s characteristics, impairments, and family-centered goals.14 In general, 1 hour per week, for several months, a common model of delivery for traditional PT, is the most common model of care at major pediatric outpatient centers in the Midwestern United States and is typical of services covered by third-party reimbursement. Intensive PT programs, using a delivery model of several hours per day, multiple days per week, have demonstrated greater motor and cognitive improvements than programs of lower intensity in other populations, including children with CP at higher functioning GMFCS levels.15-18 Although children with CP commonly receive multiple episodes of care throughout their lifetime, the optimal frequency, intensity, time, and type of interventions that are most effective are unknown. Researchers have begun to address questions related to dosing, most specifically intensity in 2 types of interventions—treadmill training (TT) and constraint-induced movement therapy (CIMT).17,19-23
286
Heathcock et al
In brief, children who received higher intensities of TT and CIMT performed better than those who received lower intensities.24,25 One intensive therapy model is a daily model, in which the child receives therapy at least 5 consecutive days per week over the episode of care. Motor Learning Providing older children with CP with functional training based on principles of motor learning improves gross motor skills such as stepping behaviors,26 balance,27 sit to stand,28 and mobility,29 as well as strength.30 Upper limb training protocols based on principles of motor learning, including CIMT and bimanual training, show that both types of intervention result in improvements in upper extremity function with CIMT showing more unimanual improvements and Hand Arm Bimanual Intensive Therapy showing more bimanual improvements, demonstrating the importance of task specificity.31-33 In addition, manipulating intensity in protocols such as TT and CIMT, both based on principles of motor learning, has resulted in greater improvement with higher intensities. None of these studies, however, compared their outcomes to a traditional PT program with the same intensity. To our knowledge, no published studies exist on intensive daily programs, like TT or CIMT, that have been used to treat younger children with higher GMFCS levels. Given the percentage of children with spastic quadriplegic CP who receive PT services, their limited mobility, and the success of higher intensity protocols in other populations, higher intensity options may be warranted. In this case series, 2 young children with spastic quadriplegic CP classified at GMFCS level V were provided traditional outpatient PT intervention using principles of motor learning and goal setting.31 The objective of this intervention was to provide each child with functional PT focusing on repetition, active exploration with error, intensity, variable practice, and education delivered using an intensive daily model. Although evidence supports the use of daily PT with multiple hours of structured intervention per day, little evidence exists on the effectiveness of these programs with children in lower functioning GMFCS levels.31,34 This case series starts to address this gap by describing a therapist-delivered daily outpatient PT program used with 2 children with spastic CP classified in GMFCS level V. For children who are more severely involved it is unknown if daily intervention can improve outcomes and be tolerated by families and children.
Purpose The purposes of this case series were to (1) describe the use of a therapist-delivered high intensity PT program for young children with CP GMFCS level V, (2) describe motor and cognitive behaviors following participation in the high intensity PT program for the 2 young children in this case series, and (3) determine the feasibility of using
Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
a high intensity protocol at an urban outpatient setting for future clinical and research participants. Description of Case Series Participants Two children aged 19 months, 14 days (16 months, 14 days corrected age) and 21 months of age with spastic quadriplegic CP, GMFCS level V, are the focus of this case series. The children were born at 26 weeks 6 days and 39 weeks 0 days, respectively. Both children included in this study were Caucasian, had no siblings at the time of participation, and both parents of each child had a college education. Child A, a boy, was born by emergency caesarean section due to decreased fetal heart tones, placental abruption, and maternal amniotic fluid embolism. He weighed 2 pounds, 0.1 ounces and was hospitalized for 189 days. Child A’s APGAR scores at 1, 5, and 10 minutes were 1, 1, and 4, respectively. Since birth, child A had 3 magnetic resonance imaging (MRI) scans of the brain, all indicating bilateral cystic periventricular leukomalacia and severe white matter loss. Child A was diagnosed with CP at the age of 12 months (corrected age). His gross motor skills at the start of the high intensity protocol consisted of sitting with moderate to maximal assistance and maximum assistance in propping on elbows in prone without weight shift for 15 seconds. Child B, a girl, was also born by emergency caesarean section due to fetal distress and was hospitalized for 10 days. Child B’s birth weight was 6 pounds, 6.6 ounces and APGAR scores at 1, 5, and 10 minutes were 2, 3, and 3, respectively. Since birth, child B had 3 MRI scans of the brain indicating hypoxic ischemic injury, microcephaly, enlarging ventricles, and severe white matter loss. Child B was diagnosed with CP at 21 months of age. Her gross motor skills at the start of the high intensity protocol consisted of supported sitting with upright head control for 2 seconds, 45◦ of cervical extension in prone for 1 second, and rolling with maximal assist.
Description of the Intervention The intervention was designed to provide PT based on the principles of motor learning to enhance and train gross motor skill development focusing on functional goals using an intense delivery model. On the basis of previous research and clinical experience with young children with CP, as well as motor learning theory, we hypothesized that a high intensity protocol would advance motor and cognitive development. Goals were established at the first treatment session with collaboration between the family and therapist. Each child had 3 functional goals with 1 goal written by the family and 2 written by the therapists. These goals were used by the treating therapist to guide treatment activities during each daily session. Goals for child A included independent sitting, increased independence with floor mobility, and weight shifting in prone. Goals for child B included Pediatric Physical Therapy
antigravity head and trunk control in prone, sitting, and during rolling (see the Appendix). Participants received outpatient therapist-directed PT treatment for 2 hours a day, 5 days a week for 4 consecutive weeks, for a total of 40 hours. The daily program was designed to enhance gross motor skills through repetition and variable practice by encouraging participants to gain strength, mobility, and gross motor skills using various developmentally appropriate activities. The specific intervention activities were different for each participant based on the individual goals, but, in general, these activities involved placing the child in various developmental positions and encouraging antigravity movement and greater independence maintaining these positions using toys or other motivators. The therapist provided the minimal amount of support and cueing during these activities and then decreased the support throughout the intervention. The children often gained knowledge of results based on whether they reached or activated a toy. Variable practice was often achieved by varying the mat surfaces or other support surfaces used during the activities. Massed and random practices were used equally during each session. For child A, approximately 40% of the treatment time focused on sitting, 35% on prone skills, and 25% on floor mobility. For child B, approximately 70% of the treatment time focused on head and postural control in sitting and prone propping and 30% on rolling. Motor, speech, and cognitive skills were assessed using the Gross Motor Functional Measure (GMFM-66, GMFM-88) and the Bayley Scales of Infant Development (Bayley-III) at 2 time points, before and after intervention. All outcome measures have good intra- and interrater reliability.35-38
Description of Outcomes Daily intervention in an outpatient hospital setting was feasible and well tolerated by participants and families. Both children completed 19/20 (95%) of the planned sessions of the intensive program. Child A missed 1 day due to illness and child B missed 1 day due to a holiday. For child A and child B, GMFM-66 scores improved by 6.06 and 9.3 points, respectively (Figure 1A). These values are above established clinically meaningful differences for the GMFM-66.39 Total GMFM-88 score improved by 12 points for child A and 16 points for child B (Figure 1B). Bayley-III gross motor raw scores improved by 5 and 7 points, fine motor skills by 0 and 3 points, receptive language skills by 3 and 2 points, and cognitive skills by 0 and 5 points for child A and child B, respectively (Figure 2). In addition, both participants met their established motor goals, which included independent sitting, improved mobility, and improved head control. Interestingly, parents of both subjects reported observable improvements in global development including cognitive, language, and fine motor skills observed by emergence of visual preference, response to name being called, attention to play routines, Intensive PT for Young Children With CP—GMFCS Level V 287
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
the tests. Equipment setup, transition time between activities, and patient breaks accounted for less than 10% of each treatment session and was documented by the treating therapist.
DISCUSSION Children with CP, GMFCS level V, can participate in a daily therapist-driven PT program in an outpatient setting. The intervention program described has known efficacy in children with CP and has novel components as it focuses on skills meaningful to both the therapist and family and was at a much higher intensity than previously reported for this population. We speculate that daily treatment protocols may be effective in promoting motor and cognitive development for younger and more severely impaired children with CP. Furthermore, we anticipate that this type of service delivery may be used in several bursts throughout the child’s lifetime.40
Fig. 1. Gross Motor Function Measure (GMFM) changes for child A and child B on the GMFM-66 (A) and the GMFM-88 (B).
improved visual tracking, and preference for novel objects or pictures. Feasibility The intensive program and assessments used in this case series were feasible and well tolerated by the children, both younger than 2 years of age and classified as GMFCS level V, as well as their families. The children were referred to the intensive program on the basis of gross motor delays by a physical therapist, and both children had received monitoring and/or intervention from a physical therapist since early infancy. Appointments were scheduled with 1 of 3 physical therapists participating in the intensive program. The experience of the 3 therapists was 2, 5, and 5 years. A parent packet was provided to each family prior to the first day of therapy to confirm program purpose, schedule, location, attendance policy, and parking information. A parking pass for free covered parking for each visit was provided on the first day of the program. Insurance was billed per treatment session with all sessions reimbursed. The children completed the developmental assessments in the first 2 days of the program and tolerated both the GMFM and Bayley-III testing well. Assessments were administered by 1 of the 3 physical therapists participating in the program. Scoring of the assessments was completed after the session. Training and reliability of assessment tools is part of clinical practice at our hospital system and not associated with this specific case series. Our hospital system tests interrater reliability on the Bayley-III yearly for therapists using the test clinically. Therapists need 85% reliability with a reference standard to continue use of 288
Heathcock et al
High Intensity PT May Improve Motor and Cognitive Development Both young children in this case series demonstrated improved motor development during the study period. Gross Motor Functional Measure (GMFM-66 and GMFM88) scores for both children reached the minimal level of clinically meaningful change, defined as 1.58 points on the GMFM-66 and 1.29 points on the GMFM-88,39 suggesting a meaningful change in motor skills. Bayley-III raw scores for the gross motor subtest also increased for both children, suggesting gross motor change in a severely impaired population. Each child had a unique medical history. Their initial motor skill levels and progress during the intervention period were similar. Motor development is advanced with interventions focused on principles of motor learning and goal setting in pediatric PT practice at lower intensities.26,31,33,41,42 Motor learning interventions have been shown to be effective for older children with CP and lower GMFCS levels.11,26-29,43,44 This case series lends support for daily motor learning interventions for younger children classified at GMFCS level V. Each child showed improvement in at least 1 other domain of development measured with the Bayley-III. Child A showed improvements in receptive language but no change in fine motor, cognitive skills, or expressive language. Many of the items on the cognitive section of the Bayley-III require fine motor skills. Child B showed improvement in all sections of the Bayley-III except expressive language. Cognitive, fine motor, and language development advance rapidly over the second year of life. Since child B was older and a girl, variables such as age-related development, motivation, and sex could have contributed to her ability to make some changes in these domains. These results indicate that intensive daily PT could have an effect on multiple domains of development. Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
Fig. 2. Bayley III scores for gross motor (A), fine motor (B), receptive language (C), expressive language (D), and cognitive skills (E) for child A and child B.
High Intensity PT May Be an Option for Younger Children With More Severe Delays High intensity intervention programs have been shown to improve motor skills for children with CP in higher functioning GMFCS levels.45 Daily, home-based, and camp models for TT and CIMT have not been presented to families and clinicians as options for children classified as GMFCS level V as their level of impairment often excludes them from research studies and clinical programs.17,20,32,46 This is true at our hospital system too, where established intensive programs of TT and CIMT designed for older children and children who are ambuPediatric Physical Therapy
latory are offered. This case series joins others’ work in suggesting that dosing may be a central issue in understanding how to deliver PT services for children with CP most effectively.14,47 This case series cannot confirm that the high intensity program improved development in these young children or would be more beneficial than a lower intensity or distributed model of service delivery; however, it does support the need and feasibility for children with CP at GMFCS level V to participate in early and intensive intervention programs in early childhood. The potential benefit of a burst of intervention is a jump in motor skill development, which could propel infants with CP to a new developmental trajectory that may have lifelong positive Intensive PT for Young Children With CP—GMFCS Level V 289
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
repercussions across all domains of development. If similar changes and long-term outcomes are established with larger research studies, intensity could have a major effect on insurance reimbursement, frequency and time of interventions, appropriate support for families, and necessary planning by families. Children with quadriplegic CP GMFCS level V require lifelong care and commitment from family and health care professionals. High intensity, and in this case series, daily pediatric PT, was a successful option for advancing motor skill development. High intensity protocols may be beneficial in the treatment of motor impairment that are at a comparable or better level. In addition, this model of service delivery in an outpatient setting complements early intervention services for children aged 0 to 3 years, which are often provided in a distributed model of care. Intensive bursts of outpatient therapy aimed at gross motor development could be offered in addition to early intervention services, which provide ongoing parent education and address the changing needs of the child on a distributed basis. The time commitment and cost to the families should not be minimized. In our cases, young children with spastic quadriplegic CP GMFCS level V and their families could participate in an outpatient-delivered daily PT program every weekday for 4 weeks. To be clear, they came to outpatient PT every weekday for 2 hours for an entire month. Future work using high intensity models of practice could include the benefit of several intense bursts of intervention, long-term follow-up, and assessment and comparisons with equal amounts (in this case series 40 hours) of intervention in a distributed model. These types of comparisons would allow planning of necessary resources including financial, time, and scheduling of both the family and other health care professionals.
Future Research
Clinical Implications
ACKNOWLEDGMENTS
Although the most common age for a CP diagnosis by a physician is 2 years, infants who are more severely affected are often diagnosed with CP earlier, representing an important window of opportunity to treat motor and other developmental impairments in the first year of life. Infants who have been diagnosed with CP before 1 year of age may benefit from a higher intensity intervention at younger ages than the young children in this case series. Future program development may look at high intensity PT with infants in GMFCS level V through the first year of life, as motor development curves described by Rosenbaum and colleagues are the closest for children in GMFCS level V and other GMFCS levels at younger ages.4,6,8 We know from human and animal models that brain plasticity including neuronal growth and refinement might be most amenable to intervention at younger ages.6,48-51 Intervening at this early age has the potential to positively affect the motor developmental trajectory for children with CP.
The authors thank the families who participated in this project. We also thank the research staff in the Infant Lab at The Ohio State University and Helen Carey, PT, PCS, for their support and facilitation of this study.
290
Heathcock et al
Studying interventions for young children in GMFCS level V is important and necessary to optimize service delivery and promote function to the greatest extent possible. Since this report is a case series, the necessary next step is to examine the effect of high intensity PT based on motor learning principles for young children classified as GMFCS level V. Future work could include randomized controlled trials comparing a distributed model to the high intensity model used in this case series.14 The second step is to determine the minimum intensity necessary for positive changes in children classified as GMFCS level V across domains of development. Clinical trials that test components of intensity, frequency, and timing (including multiple episodes of high intensity intervention) should all be investigated. Other clinical assessment tools, such as Goal Attainment Scaling and the Canadian Occupational Performance Measure, may be beneficial in capturing child-specific outcomes that norm-referenced assessment scales do not measure.52-54 We feel that future outcome measures should span multiple domains of development, including activities and participation in society, and be tested at short- and long-term intervals.55 CONCLUSIONS The findings from this case series, while preliminary in nature, support the feasibility of providing a higher intensity program based heavily on motor learning principles for young children classified as GMFCS level V. The positive changes in motor, cognitive, and language development suggest the need for a larger study to evaluate the efficacy of an intensive outpatient PT program to potentially help young infants with CP achieve a new developmental trajectory.
REFERENCES 1. Hidecker MJ, Ho NT, Dodge N, et al. Inter-relationships of functional status in cerebral palsy: analyzing gross motor function, manual ability, and communication function classification systems in children. Dev Med Child Neurol. 2012;54(8):737-742. 2. Richards CL, Malouin F. Cerebral palsy: definition, assessment and rehabilitation. Handb Clin Neurol. 2013;111:183-195. 3. Stanley F, Blair E, Alberman E. Cerebral Palsies: Epidemiology and Causal Pathways. London, United Kingdom: MacKeith Press; 2000. 4. Palisano R, Rosenbaum P, Bartlett D, Livingston M. Gross Motor Function Classification System—Expanded and Revised. Hamilton, ON, Canada: CanChild Centre for Childhood Disability Research; 2007. 5. Palisano R, Rosenbaum P, Walter S, Russell D, Wood E, Galuppi B. Development and reliability of a system to classify gross motor function in children with cerebral palsy. Dev Med Child Neurol. 1997;39(4):214-223. Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
6. Rosenbaum PL, Walter SD, Hanna SE, et al. Prognosis for gross motor function in cerebral palsy: creation of motor development curves. JAMA. 2002;288(11):1357-1363. 7. Bartlett DJ, Palisano RJ. Physical therapists’ perceptions of factors influencing the acquisition of motor abilities of children with cerebral palsy: implications for clinical reasoning. Phys Ther. 2002;82(3):237248. 8. Palisano RJ, Cameron D, Rosenbaum PL, Walter SD, Russell D. Stability of the gross motor function classification system. Dev Med Child Neurol. 2006;48(6):424-428. 9. Coleman A, Weir KA, Ware RS, Boyd RN. Relationship between communication skills and gross motor function in preschool-aged children with cerebral palsy. Arch Phys Med Rehabil. 2013;94(11):22102217. 10. Sigurdardottir S, Vik T. Speech, expressive language, and verbal cognition of preschool children with cerebral palsy in Iceland. Dev Med Child Neurol. 2011;53(1):74-80. 11. Chang HJ, Chiarello LA, Palisano RJ, Orlin MN, Bundy A, Gracely EJ. The determinants of self-determined behaviors of young children with cerebral palsy. Res Dev Disabil. 2014;35(1):99-109. 12. van Schie PE, Siebes RC, Dallmeijer AJ, et al. Development of social functioning and communication in school-aged (5-9 years) children with cerebral palsy. Res Dev Disabil. 2013;34(12):4485-4494. 13. Lobo MA, Harbourne RT, Dusing SC, McCoy SW. Grounding early intervention: physical therapy cannot just be about motor skills anymore. Phys Ther. 2013;93(1):94-103. 14. Gannotti ME, Christy JB, Heathcock JC, Kolobe TH. A path model for evaluating dosing parameters for children with cerebral palsy. Phys Ther. 2013;94:411-421. 15. Auld ML, Johnston LM. “Strong and steady”: a community-based strength and balance exercise group for children with cerebral palsy. Disabil Rehabil. 2014;36:2065-2071. 16. Brandao MB, Ferre C, Kuo HC, et al. Comparison of structured skill and unstructured practice during intensive bimanual training in children with unilateral spastic cerebral palsy. Neurorehabil Neural Repair. 2013;28(5):452-461. 17. Mattern-Baxter K, McNeil S, Mansoor JK. Effects of home-based locomotor treadmill training on gross motor function in young children with cerebral palsy: a quasi-randomized controlled trial. Arch Phys Med Rehabil. 2013;94(11):2061-2067. 18. DeLuca SC, Case-Smith J, Stevenson R, Ramey SL. Constraintinduced movement therapy (CIMT) for young children with cerebral palsy: effects of therapeutic dosage. J Pediatr Rehabil Med. 2012;5(2):133-142. 19. Begnoche DM, Pitetti KH. Effects of traditional treatment and partial body weight treadmill training on the motor skills of children with spastic cerebral palsy. A pilot study. Pediatr Phys Ther. 2007;19(1): 11-19. 20. Mattern-Baxter K, Bellamy S, Mansoor JK. Effects of intensive locomotor treadmill training on young children with cerebral palsy. Pediatr Phys Ther. 2009;21(4):308-318. 21. Willoughby KL, Dodd KJ, Shields N. A systematic review of the effectiveness of treadmill training for children with cerebral palsy. Disabil Rehabil. 2009;31(24):1971-1979. 22. Brady K, Garcia T. Constraint-induced movement therapy (CIMT): pediatric applications. Dev Disabil Res Rev. 2009;15(2):102-111. 23. Gordon AM. To constrain or not to constrain, and other stories of intensive upper extremity training for children with unilateral cerebral palsy. Dev Med Child Neurol. 2011;53(suppl 4):56-61. 24. Looper J, Ulrich DA. Effect of treadmill training and supramalleolar orthosis use on motor skill development in infants with Down syndrome: a randomized clinical trial. Phys Ther. 2010;90(3):382-390. 25. Sakzewski L, Ziviani J, Boyd R. Systematic review and meta-analysis of therapeutic management of upper-limb dysfunction in children with congenital hemiplegia. Pediatrics. 2009;123(6):e1111-e1122. 26. Bar-Haim S, Harries N, Nammourah I, Oraibi S, et al. Effectiveness of motor learning coaching in children with cerebral palsy: a randomized controlled trial. Clin Rehabil. 2010;24(11):1009-1020. 27. Katz-Leurer M, Rotem H, Keren O, Meyer S. The effects of a ‘homebased’ task-oriented exercise programme on motor and balance perPediatric Physical Therapy
28.
29.
30.
31.
32.
33.
34.
35.
36.
37.
38.
39.
40.
41.
42.
43.
44.
45.
46.
47.
formance in children with spastic cerebral palsy and severe traumatic brain injury. Clin Rehabil. 2009;23(8):714-724. Kumban W, Amatachaya S, Emasithi A, Siritaratiwat W. Effects of task-specific training on functional ability in children with mild to moderate cerebral palsy. Dev Neurorehabil. 2013;16(6):410-417. Salem Y, Godwin EM. Effects of task-oriented training on mobility function in children with cerebral palsy. NeuroRehabilitation. 2009;24(4):307-313. Lee JA, You JH, Kim DA, Lee MJ, et al. Effects of functional movement strength training on strength, muscle size, kinematics, and motor function in cerebral palsy: a 3-month follow-up. NeuroRehabilitation. 2013;32(2):287-295. Novak I, McIntyre S, Morgan C, Campbell L, et al. A systematic review of interventions for children with cerebral palsy: state of the evidence. Dev Med Child Neurol. 2013;55(10):885-910. Gordon AM, Hung YC, Brandao M, et al. Bimanual training and constraint-induced movement therapy in children with hemiplegic cerebral palsy: a randomized trial. Neurorehabil Neural Repair. 2011;25(8):692-702. Gordon AM, Schneider JA, Chinnan A, Charles JR. Efficacy of a handarm bimanual intensive therapy (HABIT) in children with hemiplegic cerebral palsy: a randomized control trial. Dev Med Child Neurol. 2007;49(11):830-838. Arpino C, Vescio MF, De Luca A, Curatolo P. Efficacy of intensive versus nonintensive physiotherapy in children with cerebral palsy: a meta-analysis. Int J Rehabil Res. 2010;33(2):165-171. Ko J, Kim M. Reliability and responsiveness of the gross motor function measure-88 in children with cerebral palsy. Phys Ther. 2013;93(3):393-400. Mahasup N, Sritipsukho P, Lekskulchai R, Keawutan P. Interrater and intra-rater reliability of the gross motor function measure (GMFM-66) by Thai pediatric physical therapists. J Med Assoc Thai. 2011;94(suppl 7):S139-S144. Chang YJ, Han WY, Tsai YC. A Kinect-based upper limb rehabilitation system to assist people with cerebral palsy. Res Dev Disabil. 2013;34(11):3654-3659. Yu YT, Hsieh WS, Hsu CH, et al. A psychometric study of the Bayley Scales of Infant and Toddler Development—3rd Edition for term and preterm Taiwanese infants. Res Dev Disabil. 2013;34(11):3875-3883. Wang HY, Yang YH. Evaluating the responsiveness of 2 versions of the gross motor function measure for children with cerebral palsy. Arch Phys Med Rehabil. 2006;87(1):51-56. Charles JR, Gordon AM. A repeated course of constraint-induced movement therapy results in further improvement. Dev Med Child Neurol. 2007;49(10):770-773. Molina-Rueda F, Aguila-Maturana AM, Molina-Rueda MJ, Miangolarra-Page JC. Treadmill training with or without partial body weight support in children with cerebral palsy: systematic review and meta-analysis [in Spanish]. Rev Neurol. 2010;51(3):135-145. Prosser LA, Ohlrich LB, Curatalo LA, Alter KE, Damiano DL. Feasibility and preliminary effectiveness of a novel mobility training intervention in infants and toddlers with cerebral palsy. Dev Neurorehabil. 2012;15(4):259-266. Willoughby KL, Dodd KJ, Shields N, Foley S. Efficacy of partial body weight-supported treadmill training compared with overground walking practice for children with cerebral palsy: a randomized controlled trial. Arch Phys Med Rehabil. 2010;91(3):333-339. Dos Santos AN, da Costa CS, Golineleo MT, Rocha NA. Functional strength training in child with cerebral palsy GMFCS IV: case report. Dev Neurorehabil. 2013;16(5):308-314. Shamir M, Dickstein R, Tirosh E. Intensive intermittent physical therapy in infants with cerebral palsy: a randomized controlled pilot study. Isr Med Assoc J. 2012;14(12):737-741. Smania N, Bonetti P, Gandolfi M, Cosentino A, et al. Improved gait after repetitive locomotor training in children with cerebral palsy. Am J Phys Med Rehabil. 2011;90(2):137-149. Kolobe TH, Christy JB, Gannotti ME, et al. Section on pediatrics research summit III. research summit III proceedings on dosing in children with an injured brain or cerebral palsy. Phys Ther. 2014;94:907-920.
Intensive PT for Young Children With CP—GMFCS Level V 291
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
48. Morgan C, Novak I, Badawi N. Enriched environments and motor outcomes in cerebral palsy: systematic review and meta-analysis. Pediatrics. 2013;132(3):e735-e746. 49. Park JW, Bang MS, Kwon BS, et al. Early treadmill training promotes motor function after hemorrhagic stroke in rats. Neurosci Lett. 2010;471(2):104-108. 50. Klintsova AY, Scamra C, Hoffman M, Napper RM, Goodlett CR, Greenough WT. Therapeutic effects of complex motor training on motor performance deficits induced by neonatal binge-like alcohol exposure in rats: II. A quantitative stereological study of synaptic plasticity in female rat cerebellum. Brain Res. 2002;937(1-2):83-93. 51. Hadders-Algra M. Early brain damage and the development of motor behavior in children: clues for therapeutic intervention? Neural Plast. 2001;8(1-2):31-49.
52. Steenbeek D, Gorter JW, Ketelaar M, Galama K, Lindeman E. Responsiveness of Goal Attainment Scaling in comparison to two standardized measures in outcome evaluation of children with cerebral palsy. Clin Rehabil. 2011;25(12):1128-1139. 53. Sorsdahl AB, Moe-Nilssen R, Kaale HK, Rieber J, Strand LI. Change in basic motor abilities, quality of movement and everyday activities following intensive, goal-directed, activity-focused physiotherapy in a group setting for children with cerebral palsy. BMC Pediatr. 2010;10:26. 54. Palisano RJ, Haley SM, Brown DA. Goal attainment scaling as a measure of change in infants with motor delays. Phys Ther. 1992;72(6):432-437. 55. Jette A. Toward a common language for function, disability, and health. Phys Ther. 2006;86(5):726-734.
APPENDIX Examples of Goals Set by Family and Therapist Goal Type
Child A
Family goal
Child A will sit by himself and will have a form of independent floor mobility Child A will sit independently for 10 s in 2/3 trials Child A will weight shift in prone and reach in 5/10 attempts
Therapist goal Therapist goal
292
Heathcock et al
Child B To increase child B’s neck and core strength to progress independence with sitting and rolling Child B will maintain 45◦ of cervical extension in prone prop for 5 s, 2/3 trials in 1 session Child B will demonstrate improved head control in supported sitting through the ability to maintain a neutral cervical position for 5 s 2/3 trials in 1 session
Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
R E P O R T
Daily Intervention for Young Children With Cerebral Palsy in GMFCS Level V: A Case Series Jill C. Heathcock, MPT, PhD; Kathleen Baranet, PT, DPT; Rachel Ferrante, PT, DPT; Sarah Hendershot, PT, DPT Division of Physical Therapy (Dr Heathcock), The Ohio State University, Columbus, Ohio; Center for Perinatal Research (Dr Heathcock) and Department of Clinical Therapies (Drs Baranet, Ferrante, and Hendershot), Nationwide Children’s Hospital, Columbus, Ohio.
Purpose: To describe a daily physical therapy (PT) intervention program and outcomes for 2 young children with spastic quadriplegia, Gross Motor Function Classification System (GMFCS) level V, and to evaluate the feasibility of using a daily program in an urban children’s hospital outpatient setting. Summary of Key Points: Two young children, GMFCS level V, received 2 hours of PT intervention based on motor learning principles 5 days a week for 4 consecutive weeks. Gross Motor Functional Measure (GMFM-66, GMFM-88) and the Bayley Scales of Infant Development, Third Edition, were used as pre- and postoutcome measures. The daily, high intensity intervention was well tolerated. Improvements in motor function, language, and cognitive skills were found. Statement of Conclusion: A daily PT program appears feasible and may improve overall development in young children with cerebral palsy in GMFCS level V. (Pediatr Phys Ther 2015;27:285–292) Key words: case report, cerebral palsy, child development, human, infant, female, male, outcomes assessment, physical therapy/methods/administration and dosage, quadriplegia
INTRODUCTION Children with spastic cerebral palsy (CP) account for 80% of the CP population, with 15% estimated to have spastic quadriplegia.1-3 Children with spastic quadriplegic CP typically have very limited mobility and are classified at higher Gross Motor Function Classification System (GMFCS) levels.4,5 The GMFCS is an age-based classification scale for describing motor abilities in children with CP from birth to 18 years old. The classification levels, I through V, indicate least severe to most severe impairments of motor
0898-5669/110/2703-0285 Pediatric Physical Therapy C 2015 Wolters Kluwer Health, Inc. All rights reserved. and Copyright Section on Pediatrics of the American Physical Therapy Association
Correspondence: Jill C. Heathcock, MPT, PhD, Division of Physical Therapy, The Ohio State University, 453 W. 10th Ave, Columbus, Ohio 43214 ([email protected]). The authors declare no conflicts of interest. At the time this article was written, Sarah Hendershot, PT, DPT, was a pediatric physical therapist resident at Nationwide Children’s Hospital and The Ohio State University. DOI: 10.1097/PEP.0000000000000149
Pediatric Physical Therapy
function, respectively.4-6 For example, children with CP classified as GMFCS level V typically do not have independent forms of mobility and are dependent on caregivers for most aspects of daily life. In infancy, children classified at level V have limited voluntary control and antigravity movements and cannot roll or sit independently.4-6 Perhaps as a result of more severe delay and lower skill level expectations, they are infrequently included in intervention studies, especially those with high intensity protocols. When children in GMFCS level V are included in intervention studies, they are commonly grouped with children in levels III to V to create a nonambulatory group. Children in GMFCS level V have unique motor skill development compared with other children with CP and show a plateau in motor skill development earlier than children in other GMFCS levels.6 The motor development curves described by Rosenbaum et al6 show the spread of gross motor skills between GMFCS level V and other GMFCS levels is closest from 0 to 3 years of age. Children in all 5 levels continue to make progress from 0 to 3 years of age, possibly indicating a window of opportunity to promote optimal future development. Direct interventions aimed at making meaningful changes in motor skill development during this time period may be justified.7 Nonefficacious intervention and service delivery means Intensive PT for Young Children With CP—GMFCS Level V 285
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
that the children with CP who are most seriously impaired may miss critical years for intervention when brain plasticity including formation of new neural pathways, and growth and development of synaptic formations may result in the most potential to change motor function. Importantly, children classified in GMFCS level V tend to remain in that level over time, and younger children in level V who are reclassified are more frequently classified to a lower functional level.8 It is unknown whether higher intensities of intervention at younger ages can improve GMFCS classification. These results support the need to provide effective interventions at a young age to optimize function and determine whether function is within or above the expected trajectory. Children in GMFCS level V often demonstrate multiple impairments and show delays and disabilities in other domains of development such as communication skills,9 language skills,10 self-determination,11 cognition, behavior, and social function.12 They show these delays and disabilities at a higher rate and severity than children who are typically developing and those in higher functioning GMFCS levels. Furthermore, early skill development, such as head control and independent sitting, influences how children perceive their world and interact with their environment and caregivers. Children with major motor atypicalities have less experience independently moving their bodies against gravity and exploring their environment. This limitation affects not only their immediate gross and fine motor, language, cognitive, and social skills, but also their developmental trajectory.13 Distribution and Dosing of Physical Therapy The delivery of physical therapy (PT) services for children with CP including frequency, intensity, and time varies greatly depending on the region in which the family resides and the environment in which the services are provided. These features of dosing may also be dependent on the type of intervention, the child’s characteristics, impairments, and family-centered goals.14 In general, 1 hour per week, for several months, a common model of delivery for traditional PT, is the most common model of care at major pediatric outpatient centers in the Midwestern United States and is typical of services covered by third-party reimbursement. Intensive PT programs, using a delivery model of several hours per day, multiple days per week, have demonstrated greater motor and cognitive improvements than programs of lower intensity in other populations, including children with CP at higher functioning GMFCS levels.15-18 Although children with CP commonly receive multiple episodes of care throughout their lifetime, the optimal frequency, intensity, time, and type of interventions that are most effective are unknown. Researchers have begun to address questions related to dosing, most specifically intensity in 2 types of interventions—treadmill training (TT) and constraint-induced movement therapy (CIMT).17,19-23
286
Heathcock et al
In brief, children who received higher intensities of TT and CIMT performed better than those who received lower intensities.24,25 One intensive therapy model is a daily model, in which the child receives therapy at least 5 consecutive days per week over the episode of care. Motor Learning Providing older children with CP with functional training based on principles of motor learning improves gross motor skills such as stepping behaviors,26 balance,27 sit to stand,28 and mobility,29 as well as strength.30 Upper limb training protocols based on principles of motor learning, including CIMT and bimanual training, show that both types of intervention result in improvements in upper extremity function with CIMT showing more unimanual improvements and Hand Arm Bimanual Intensive Therapy showing more bimanual improvements, demonstrating the importance of task specificity.31-33 In addition, manipulating intensity in protocols such as TT and CIMT, both based on principles of motor learning, has resulted in greater improvement with higher intensities. None of these studies, however, compared their outcomes to a traditional PT program with the same intensity. To our knowledge, no published studies exist on intensive daily programs, like TT or CIMT, that have been used to treat younger children with higher GMFCS levels. Given the percentage of children with spastic quadriplegic CP who receive PT services, their limited mobility, and the success of higher intensity protocols in other populations, higher intensity options may be warranted. In this case series, 2 young children with spastic quadriplegic CP classified at GMFCS level V were provided traditional outpatient PT intervention using principles of motor learning and goal setting.31 The objective of this intervention was to provide each child with functional PT focusing on repetition, active exploration with error, intensity, variable practice, and education delivered using an intensive daily model. Although evidence supports the use of daily PT with multiple hours of structured intervention per day, little evidence exists on the effectiveness of these programs with children in lower functioning GMFCS levels.31,34 This case series starts to address this gap by describing a therapist-delivered daily outpatient PT program used with 2 children with spastic CP classified in GMFCS level V. For children who are more severely involved it is unknown if daily intervention can improve outcomes and be tolerated by families and children.
Purpose The purposes of this case series were to (1) describe the use of a therapist-delivered high intensity PT program for young children with CP GMFCS level V, (2) describe motor and cognitive behaviors following participation in the high intensity PT program for the 2 young children in this case series, and (3) determine the feasibility of using
Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
a high intensity protocol at an urban outpatient setting for future clinical and research participants. Description of Case Series Participants Two children aged 19 months, 14 days (16 months, 14 days corrected age) and 21 months of age with spastic quadriplegic CP, GMFCS level V, are the focus of this case series. The children were born at 26 weeks 6 days and 39 weeks 0 days, respectively. Both children included in this study were Caucasian, had no siblings at the time of participation, and both parents of each child had a college education. Child A, a boy, was born by emergency caesarean section due to decreased fetal heart tones, placental abruption, and maternal amniotic fluid embolism. He weighed 2 pounds, 0.1 ounces and was hospitalized for 189 days. Child A’s APGAR scores at 1, 5, and 10 minutes were 1, 1, and 4, respectively. Since birth, child A had 3 magnetic resonance imaging (MRI) scans of the brain, all indicating bilateral cystic periventricular leukomalacia and severe white matter loss. Child A was diagnosed with CP at the age of 12 months (corrected age). His gross motor skills at the start of the high intensity protocol consisted of sitting with moderate to maximal assistance and maximum assistance in propping on elbows in prone without weight shift for 15 seconds. Child B, a girl, was also born by emergency caesarean section due to fetal distress and was hospitalized for 10 days. Child B’s birth weight was 6 pounds, 6.6 ounces and APGAR scores at 1, 5, and 10 minutes were 2, 3, and 3, respectively. Since birth, child B had 3 MRI scans of the brain indicating hypoxic ischemic injury, microcephaly, enlarging ventricles, and severe white matter loss. Child B was diagnosed with CP at 21 months of age. Her gross motor skills at the start of the high intensity protocol consisted of supported sitting with upright head control for 2 seconds, 45◦ of cervical extension in prone for 1 second, and rolling with maximal assist.
Description of the Intervention The intervention was designed to provide PT based on the principles of motor learning to enhance and train gross motor skill development focusing on functional goals using an intense delivery model. On the basis of previous research and clinical experience with young children with CP, as well as motor learning theory, we hypothesized that a high intensity protocol would advance motor and cognitive development. Goals were established at the first treatment session with collaboration between the family and therapist. Each child had 3 functional goals with 1 goal written by the family and 2 written by the therapists. These goals were used by the treating therapist to guide treatment activities during each daily session. Goals for child A included independent sitting, increased independence with floor mobility, and weight shifting in prone. Goals for child B included Pediatric Physical Therapy
antigravity head and trunk control in prone, sitting, and during rolling (see the Appendix). Participants received outpatient therapist-directed PT treatment for 2 hours a day, 5 days a week for 4 consecutive weeks, for a total of 40 hours. The daily program was designed to enhance gross motor skills through repetition and variable practice by encouraging participants to gain strength, mobility, and gross motor skills using various developmentally appropriate activities. The specific intervention activities were different for each participant based on the individual goals, but, in general, these activities involved placing the child in various developmental positions and encouraging antigravity movement and greater independence maintaining these positions using toys or other motivators. The therapist provided the minimal amount of support and cueing during these activities and then decreased the support throughout the intervention. The children often gained knowledge of results based on whether they reached or activated a toy. Variable practice was often achieved by varying the mat surfaces or other support surfaces used during the activities. Massed and random practices were used equally during each session. For child A, approximately 40% of the treatment time focused on sitting, 35% on prone skills, and 25% on floor mobility. For child B, approximately 70% of the treatment time focused on head and postural control in sitting and prone propping and 30% on rolling. Motor, speech, and cognitive skills were assessed using the Gross Motor Functional Measure (GMFM-66, GMFM-88) and the Bayley Scales of Infant Development (Bayley-III) at 2 time points, before and after intervention. All outcome measures have good intra- and interrater reliability.35-38
Description of Outcomes Daily intervention in an outpatient hospital setting was feasible and well tolerated by participants and families. Both children completed 19/20 (95%) of the planned sessions of the intensive program. Child A missed 1 day due to illness and child B missed 1 day due to a holiday. For child A and child B, GMFM-66 scores improved by 6.06 and 9.3 points, respectively (Figure 1A). These values are above established clinically meaningful differences for the GMFM-66.39 Total GMFM-88 score improved by 12 points for child A and 16 points for child B (Figure 1B). Bayley-III gross motor raw scores improved by 5 and 7 points, fine motor skills by 0 and 3 points, receptive language skills by 3 and 2 points, and cognitive skills by 0 and 5 points for child A and child B, respectively (Figure 2). In addition, both participants met their established motor goals, which included independent sitting, improved mobility, and improved head control. Interestingly, parents of both subjects reported observable improvements in global development including cognitive, language, and fine motor skills observed by emergence of visual preference, response to name being called, attention to play routines, Intensive PT for Young Children With CP—GMFCS Level V 287
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
the tests. Equipment setup, transition time between activities, and patient breaks accounted for less than 10% of each treatment session and was documented by the treating therapist.
DISCUSSION Children with CP, GMFCS level V, can participate in a daily therapist-driven PT program in an outpatient setting. The intervention program described has known efficacy in children with CP and has novel components as it focuses on skills meaningful to both the therapist and family and was at a much higher intensity than previously reported for this population. We speculate that daily treatment protocols may be effective in promoting motor and cognitive development for younger and more severely impaired children with CP. Furthermore, we anticipate that this type of service delivery may be used in several bursts throughout the child’s lifetime.40
Fig. 1. Gross Motor Function Measure (GMFM) changes for child A and child B on the GMFM-66 (A) and the GMFM-88 (B).
improved visual tracking, and preference for novel objects or pictures. Feasibility The intensive program and assessments used in this case series were feasible and well tolerated by the children, both younger than 2 years of age and classified as GMFCS level V, as well as their families. The children were referred to the intensive program on the basis of gross motor delays by a physical therapist, and both children had received monitoring and/or intervention from a physical therapist since early infancy. Appointments were scheduled with 1 of 3 physical therapists participating in the intensive program. The experience of the 3 therapists was 2, 5, and 5 years. A parent packet was provided to each family prior to the first day of therapy to confirm program purpose, schedule, location, attendance policy, and parking information. A parking pass for free covered parking for each visit was provided on the first day of the program. Insurance was billed per treatment session with all sessions reimbursed. The children completed the developmental assessments in the first 2 days of the program and tolerated both the GMFM and Bayley-III testing well. Assessments were administered by 1 of the 3 physical therapists participating in the program. Scoring of the assessments was completed after the session. Training and reliability of assessment tools is part of clinical practice at our hospital system and not associated with this specific case series. Our hospital system tests interrater reliability on the Bayley-III yearly for therapists using the test clinically. Therapists need 85% reliability with a reference standard to continue use of 288
Heathcock et al
High Intensity PT May Improve Motor and Cognitive Development Both young children in this case series demonstrated improved motor development during the study period. Gross Motor Functional Measure (GMFM-66 and GMFM88) scores for both children reached the minimal level of clinically meaningful change, defined as 1.58 points on the GMFM-66 and 1.29 points on the GMFM-88,39 suggesting a meaningful change in motor skills. Bayley-III raw scores for the gross motor subtest also increased for both children, suggesting gross motor change in a severely impaired population. Each child had a unique medical history. Their initial motor skill levels and progress during the intervention period were similar. Motor development is advanced with interventions focused on principles of motor learning and goal setting in pediatric PT practice at lower intensities.26,31,33,41,42 Motor learning interventions have been shown to be effective for older children with CP and lower GMFCS levels.11,26-29,43,44 This case series lends support for daily motor learning interventions for younger children classified at GMFCS level V. Each child showed improvement in at least 1 other domain of development measured with the Bayley-III. Child A showed improvements in receptive language but no change in fine motor, cognitive skills, or expressive language. Many of the items on the cognitive section of the Bayley-III require fine motor skills. Child B showed improvement in all sections of the Bayley-III except expressive language. Cognitive, fine motor, and language development advance rapidly over the second year of life. Since child B was older and a girl, variables such as age-related development, motivation, and sex could have contributed to her ability to make some changes in these domains. These results indicate that intensive daily PT could have an effect on multiple domains of development. Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
Fig. 2. Bayley III scores for gross motor (A), fine motor (B), receptive language (C), expressive language (D), and cognitive skills (E) for child A and child B.
High Intensity PT May Be an Option for Younger Children With More Severe Delays High intensity intervention programs have been shown to improve motor skills for children with CP in higher functioning GMFCS levels.45 Daily, home-based, and camp models for TT and CIMT have not been presented to families and clinicians as options for children classified as GMFCS level V as their level of impairment often excludes them from research studies and clinical programs.17,20,32,46 This is true at our hospital system too, where established intensive programs of TT and CIMT designed for older children and children who are ambuPediatric Physical Therapy
latory are offered. This case series joins others’ work in suggesting that dosing may be a central issue in understanding how to deliver PT services for children with CP most effectively.14,47 This case series cannot confirm that the high intensity program improved development in these young children or would be more beneficial than a lower intensity or distributed model of service delivery; however, it does support the need and feasibility for children with CP at GMFCS level V to participate in early and intensive intervention programs in early childhood. The potential benefit of a burst of intervention is a jump in motor skill development, which could propel infants with CP to a new developmental trajectory that may have lifelong positive Intensive PT for Young Children With CP—GMFCS Level V 289
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
repercussions across all domains of development. If similar changes and long-term outcomes are established with larger research studies, intensity could have a major effect on insurance reimbursement, frequency and time of interventions, appropriate support for families, and necessary planning by families. Children with quadriplegic CP GMFCS level V require lifelong care and commitment from family and health care professionals. High intensity, and in this case series, daily pediatric PT, was a successful option for advancing motor skill development. High intensity protocols may be beneficial in the treatment of motor impairment that are at a comparable or better level. In addition, this model of service delivery in an outpatient setting complements early intervention services for children aged 0 to 3 years, which are often provided in a distributed model of care. Intensive bursts of outpatient therapy aimed at gross motor development could be offered in addition to early intervention services, which provide ongoing parent education and address the changing needs of the child on a distributed basis. The time commitment and cost to the families should not be minimized. In our cases, young children with spastic quadriplegic CP GMFCS level V and their families could participate in an outpatient-delivered daily PT program every weekday for 4 weeks. To be clear, they came to outpatient PT every weekday for 2 hours for an entire month. Future work using high intensity models of practice could include the benefit of several intense bursts of intervention, long-term follow-up, and assessment and comparisons with equal amounts (in this case series 40 hours) of intervention in a distributed model. These types of comparisons would allow planning of necessary resources including financial, time, and scheduling of both the family and other health care professionals.
Future Research
Clinical Implications
ACKNOWLEDGMENTS
Although the most common age for a CP diagnosis by a physician is 2 years, infants who are more severely affected are often diagnosed with CP earlier, representing an important window of opportunity to treat motor and other developmental impairments in the first year of life. Infants who have been diagnosed with CP before 1 year of age may benefit from a higher intensity intervention at younger ages than the young children in this case series. Future program development may look at high intensity PT with infants in GMFCS level V through the first year of life, as motor development curves described by Rosenbaum and colleagues are the closest for children in GMFCS level V and other GMFCS levels at younger ages.4,6,8 We know from human and animal models that brain plasticity including neuronal growth and refinement might be most amenable to intervention at younger ages.6,48-51 Intervening at this early age has the potential to positively affect the motor developmental trajectory for children with CP.
The authors thank the families who participated in this project. We also thank the research staff in the Infant Lab at The Ohio State University and Helen Carey, PT, PCS, for their support and facilitation of this study.
290
Heathcock et al
Studying interventions for young children in GMFCS level V is important and necessary to optimize service delivery and promote function to the greatest extent possible. Since this report is a case series, the necessary next step is to examine the effect of high intensity PT based on motor learning principles for young children classified as GMFCS level V. Future work could include randomized controlled trials comparing a distributed model to the high intensity model used in this case series.14 The second step is to determine the minimum intensity necessary for positive changes in children classified as GMFCS level V across domains of development. Clinical trials that test components of intensity, frequency, and timing (including multiple episodes of high intensity intervention) should all be investigated. Other clinical assessment tools, such as Goal Attainment Scaling and the Canadian Occupational Performance Measure, may be beneficial in capturing child-specific outcomes that norm-referenced assessment scales do not measure.52-54 We feel that future outcome measures should span multiple domains of development, including activities and participation in society, and be tested at short- and long-term intervals.55 CONCLUSIONS The findings from this case series, while preliminary in nature, support the feasibility of providing a higher intensity program based heavily on motor learning principles for young children classified as GMFCS level V. The positive changes in motor, cognitive, and language development suggest the need for a larger study to evaluate the efficacy of an intensive outpatient PT program to potentially help young infants with CP achieve a new developmental trajectory.
REFERENCES 1. Hidecker MJ, Ho NT, Dodge N, et al. Inter-relationships of functional status in cerebral palsy: analyzing gross motor function, manual ability, and communication function classification systems in children. Dev Med Child Neurol. 2012;54(8):737-742. 2. Richards CL, Malouin F. Cerebral palsy: definition, assessment and rehabilitation. Handb Clin Neurol. 2013;111:183-195. 3. Stanley F, Blair E, Alberman E. Cerebral Palsies: Epidemiology and Causal Pathways. London, United Kingdom: MacKeith Press; 2000. 4. Palisano R, Rosenbaum P, Bartlett D, Livingston M. Gross Motor Function Classification System—Expanded and Revised. Hamilton, ON, Canada: CanChild Centre for Childhood Disability Research; 2007. 5. Palisano R, Rosenbaum P, Walter S, Russell D, Wood E, Galuppi B. Development and reliability of a system to classify gross motor function in children with cerebral palsy. Dev Med Child Neurol. 1997;39(4):214-223. Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
6. Rosenbaum PL, Walter SD, Hanna SE, et al. Prognosis for gross motor function in cerebral palsy: creation of motor development curves. JAMA. 2002;288(11):1357-1363. 7. Bartlett DJ, Palisano RJ. Physical therapists’ perceptions of factors influencing the acquisition of motor abilities of children with cerebral palsy: implications for clinical reasoning. Phys Ther. 2002;82(3):237248. 8. Palisano RJ, Cameron D, Rosenbaum PL, Walter SD, Russell D. Stability of the gross motor function classification system. Dev Med Child Neurol. 2006;48(6):424-428. 9. Coleman A, Weir KA, Ware RS, Boyd RN. Relationship between communication skills and gross motor function in preschool-aged children with cerebral palsy. Arch Phys Med Rehabil. 2013;94(11):22102217. 10. Sigurdardottir S, Vik T. Speech, expressive language, and verbal cognition of preschool children with cerebral palsy in Iceland. Dev Med Child Neurol. 2011;53(1):74-80. 11. Chang HJ, Chiarello LA, Palisano RJ, Orlin MN, Bundy A, Gracely EJ. The determinants of self-determined behaviors of young children with cerebral palsy. Res Dev Disabil. 2014;35(1):99-109. 12. van Schie PE, Siebes RC, Dallmeijer AJ, et al. Development of social functioning and communication in school-aged (5-9 years) children with cerebral palsy. Res Dev Disabil. 2013;34(12):4485-4494. 13. Lobo MA, Harbourne RT, Dusing SC, McCoy SW. Grounding early intervention: physical therapy cannot just be about motor skills anymore. Phys Ther. 2013;93(1):94-103. 14. Gannotti ME, Christy JB, Heathcock JC, Kolobe TH. A path model for evaluating dosing parameters for children with cerebral palsy. Phys Ther. 2013;94:411-421. 15. Auld ML, Johnston LM. “Strong and steady”: a community-based strength and balance exercise group for children with cerebral palsy. Disabil Rehabil. 2014;36:2065-2071. 16. Brandao MB, Ferre C, Kuo HC, et al. Comparison of structured skill and unstructured practice during intensive bimanual training in children with unilateral spastic cerebral palsy. Neurorehabil Neural Repair. 2013;28(5):452-461. 17. Mattern-Baxter K, McNeil S, Mansoor JK. Effects of home-based locomotor treadmill training on gross motor function in young children with cerebral palsy: a quasi-randomized controlled trial. Arch Phys Med Rehabil. 2013;94(11):2061-2067. 18. DeLuca SC, Case-Smith J, Stevenson R, Ramey SL. Constraintinduced movement therapy (CIMT) for young children with cerebral palsy: effects of therapeutic dosage. J Pediatr Rehabil Med. 2012;5(2):133-142. 19. Begnoche DM, Pitetti KH. Effects of traditional treatment and partial body weight treadmill training on the motor skills of children with spastic cerebral palsy. A pilot study. Pediatr Phys Ther. 2007;19(1): 11-19. 20. Mattern-Baxter K, Bellamy S, Mansoor JK. Effects of intensive locomotor treadmill training on young children with cerebral palsy. Pediatr Phys Ther. 2009;21(4):308-318. 21. Willoughby KL, Dodd KJ, Shields N. A systematic review of the effectiveness of treadmill training for children with cerebral palsy. Disabil Rehabil. 2009;31(24):1971-1979. 22. Brady K, Garcia T. Constraint-induced movement therapy (CIMT): pediatric applications. Dev Disabil Res Rev. 2009;15(2):102-111. 23. Gordon AM. To constrain or not to constrain, and other stories of intensive upper extremity training for children with unilateral cerebral palsy. Dev Med Child Neurol. 2011;53(suppl 4):56-61. 24. Looper J, Ulrich DA. Effect of treadmill training and supramalleolar orthosis use on motor skill development in infants with Down syndrome: a randomized clinical trial. Phys Ther. 2010;90(3):382-390. 25. Sakzewski L, Ziviani J, Boyd R. Systematic review and meta-analysis of therapeutic management of upper-limb dysfunction in children with congenital hemiplegia. Pediatrics. 2009;123(6):e1111-e1122. 26. Bar-Haim S, Harries N, Nammourah I, Oraibi S, et al. Effectiveness of motor learning coaching in children with cerebral palsy: a randomized controlled trial. Clin Rehabil. 2010;24(11):1009-1020. 27. Katz-Leurer M, Rotem H, Keren O, Meyer S. The effects of a ‘homebased’ task-oriented exercise programme on motor and balance perPediatric Physical Therapy
28.
29.
30.
31.
32.
33.
34.
35.
36.
37.
38.
39.
40.
41.
42.
43.
44.
45.
46.
47.
formance in children with spastic cerebral palsy and severe traumatic brain injury. Clin Rehabil. 2009;23(8):714-724. Kumban W, Amatachaya S, Emasithi A, Siritaratiwat W. Effects of task-specific training on functional ability in children with mild to moderate cerebral palsy. Dev Neurorehabil. 2013;16(6):410-417. Salem Y, Godwin EM. Effects of task-oriented training on mobility function in children with cerebral palsy. NeuroRehabilitation. 2009;24(4):307-313. Lee JA, You JH, Kim DA, Lee MJ, et al. Effects of functional movement strength training on strength, muscle size, kinematics, and motor function in cerebral palsy: a 3-month follow-up. NeuroRehabilitation. 2013;32(2):287-295. Novak I, McIntyre S, Morgan C, Campbell L, et al. A systematic review of interventions for children with cerebral palsy: state of the evidence. Dev Med Child Neurol. 2013;55(10):885-910. Gordon AM, Hung YC, Brandao M, et al. Bimanual training and constraint-induced movement therapy in children with hemiplegic cerebral palsy: a randomized trial. Neurorehabil Neural Repair. 2011;25(8):692-702. Gordon AM, Schneider JA, Chinnan A, Charles JR. Efficacy of a handarm bimanual intensive therapy (HABIT) in children with hemiplegic cerebral palsy: a randomized control trial. Dev Med Child Neurol. 2007;49(11):830-838. Arpino C, Vescio MF, De Luca A, Curatolo P. Efficacy of intensive versus nonintensive physiotherapy in children with cerebral palsy: a meta-analysis. Int J Rehabil Res. 2010;33(2):165-171. Ko J, Kim M. Reliability and responsiveness of the gross motor function measure-88 in children with cerebral palsy. Phys Ther. 2013;93(3):393-400. Mahasup N, Sritipsukho P, Lekskulchai R, Keawutan P. Interrater and intra-rater reliability of the gross motor function measure (GMFM-66) by Thai pediatric physical therapists. J Med Assoc Thai. 2011;94(suppl 7):S139-S144. Chang YJ, Han WY, Tsai YC. A Kinect-based upper limb rehabilitation system to assist people with cerebral palsy. Res Dev Disabil. 2013;34(11):3654-3659. Yu YT, Hsieh WS, Hsu CH, et al. A psychometric study of the Bayley Scales of Infant and Toddler Development—3rd Edition for term and preterm Taiwanese infants. Res Dev Disabil. 2013;34(11):3875-3883. Wang HY, Yang YH. Evaluating the responsiveness of 2 versions of the gross motor function measure for children with cerebral palsy. Arch Phys Med Rehabil. 2006;87(1):51-56. Charles JR, Gordon AM. A repeated course of constraint-induced movement therapy results in further improvement. Dev Med Child Neurol. 2007;49(10):770-773. Molina-Rueda F, Aguila-Maturana AM, Molina-Rueda MJ, Miangolarra-Page JC. Treadmill training with or without partial body weight support in children with cerebral palsy: systematic review and meta-analysis [in Spanish]. Rev Neurol. 2010;51(3):135-145. Prosser LA, Ohlrich LB, Curatalo LA, Alter KE, Damiano DL. Feasibility and preliminary effectiveness of a novel mobility training intervention in infants and toddlers with cerebral palsy. Dev Neurorehabil. 2012;15(4):259-266. Willoughby KL, Dodd KJ, Shields N, Foley S. Efficacy of partial body weight-supported treadmill training compared with overground walking practice for children with cerebral palsy: a randomized controlled trial. Arch Phys Med Rehabil. 2010;91(3):333-339. Dos Santos AN, da Costa CS, Golineleo MT, Rocha NA. Functional strength training in child with cerebral palsy GMFCS IV: case report. Dev Neurorehabil. 2013;16(5):308-314. Shamir M, Dickstein R, Tirosh E. Intensive intermittent physical therapy in infants with cerebral palsy: a randomized controlled pilot study. Isr Med Assoc J. 2012;14(12):737-741. Smania N, Bonetti P, Gandolfi M, Cosentino A, et al. Improved gait after repetitive locomotor training in children with cerebral palsy. Am J Phys Med Rehabil. 2011;90(2):137-149. Kolobe TH, Christy JB, Gannotti ME, et al. Section on pediatrics research summit III. research summit III proceedings on dosing in children with an injured brain or cerebral palsy. Phys Ther. 2014;94:907-920.
Intensive PT for Young Children With CP—GMFCS Level V 291
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
48. Morgan C, Novak I, Badawi N. Enriched environments and motor outcomes in cerebral palsy: systematic review and meta-analysis. Pediatrics. 2013;132(3):e735-e746. 49. Park JW, Bang MS, Kwon BS, et al. Early treadmill training promotes motor function after hemorrhagic stroke in rats. Neurosci Lett. 2010;471(2):104-108. 50. Klintsova AY, Scamra C, Hoffman M, Napper RM, Goodlett CR, Greenough WT. Therapeutic effects of complex motor training on motor performance deficits induced by neonatal binge-like alcohol exposure in rats: II. A quantitative stereological study of synaptic plasticity in female rat cerebellum. Brain Res. 2002;937(1-2):83-93. 51. Hadders-Algra M. Early brain damage and the development of motor behavior in children: clues for therapeutic intervention? Neural Plast. 2001;8(1-2):31-49.
52. Steenbeek D, Gorter JW, Ketelaar M, Galama K, Lindeman E. Responsiveness of Goal Attainment Scaling in comparison to two standardized measures in outcome evaluation of children with cerebral palsy. Clin Rehabil. 2011;25(12):1128-1139. 53. Sorsdahl AB, Moe-Nilssen R, Kaale HK, Rieber J, Strand LI. Change in basic motor abilities, quality of movement and everyday activities following intensive, goal-directed, activity-focused physiotherapy in a group setting for children with cerebral palsy. BMC Pediatr. 2010;10:26. 54. Palisano RJ, Haley SM, Brown DA. Goal attainment scaling as a measure of change in infants with motor delays. Phys Ther. 1992;72(6):432-437. 55. Jette A. Toward a common language for function, disability, and health. Phys Ther. 2006;86(5):726-734.
APPENDIX Examples of Goals Set by Family and Therapist Goal Type
Child A
Family goal
Child A will sit by himself and will have a form of independent floor mobility Child A will sit independently for 10 s in 2/3 trials Child A will weight shift in prone and reach in 5/10 attempts
Therapist goal Therapist goal
292
Heathcock et al
Child B To increase child B’s neck and core strength to progress independence with sitting and rolling Child B will maintain 45◦ of cervical extension in prone prop for 5 s, 2/3 trials in 1 session Child B will demonstrate improved head control in supported sitting through the ability to maintain a neutral cervical position for 5 s 2/3 trials in 1 session
Pediatric Physical Therapy
Copyright © 2015 Wolters Kluwer Health, Inc. and the Section on Pediatrics of the American Physical Therapy Association. Unauthorized reproduction of this article is prohibited.
