Spina bifida is the most common birth defect affecting the central nervous system.1 The prevalence of spina bifida among children and adolescents 0 to 19 years of age in the United States is 3.1 cases per 10 000.2 Delayed achievement of ambulation is expected for all children with spina bifida, but most children achieve some form of ambulatory function.3 While ambulatory ability is related to the neurological level affected, large variations in ambulatory outcomes exist within a specific lesion group.4–7
Treadmill training provides task-specific stepping practice. Studies investigating the use of treadmill training to improve ambulatory skills in pediatric populations at risk for neuromotor delays, such as Down syndrome and cerebral palsy, have demonstrated promising initial results.8–19 In 2 small studies investigators found that treadmill training elicited stepping in infants with spina bifida.6,20 A case study used a combination of treadmill and overground gait training with an 18-month-old child with myelomeningocele.21 The child progressed from no stepping on the treadmill to ambulating 153 m over ground in her walker.21 A randomized controlled trial evaluated the effectiveness of a 12-week home-based, individualized, progressive treadmill training program with children 6 to 18 years of age with spina bifida who were able to ambulate in the community.22 A significant difference between the treadmill training group and the control group was found immediately after the training and 3 months postintervention in walking speed, duration, VO2 peak, and gross energy consumption.22
Despite this initial evidence, significant gaps continue to exist in the literature with regard to the use of treadmill training with the spina bifida population. Optimal training parameters including intensity, duration, patient selection, and location of practice (home vs clinic) have not been established. No studies have compared various intensities or durations of treadmill training to determine the most effective protocol. No investigation into the use of treadmill training in children 2 to 6 years of age, or in children older than 6 years who are at a level of ambulation other than community, has occurred. The use of treadmill training in a home setting versus a clinic setting has not been studied, although Charney et al4 found that a strong predictor of ambulation among children with spina bifida was involvement in a physical therapy program committed to teaching and achieving the specific goal of walking. In addition to determining optimal training parameters, authors of 3 systematic reviews have recommended further investigation into the effects of treadmill training on young children at risk for developmental delays.10,17,19
Case reports provide an opportunity to explore a new therapeutic approach with a different population.23 While they have limitations, they can be useful in generating new hypotheses or ideas.23,24 This information can lead to further, more rigorous investigation into a new technique or idea. The use of treadmill training has not been investigated with preschool-aged children with spina bifida. Preschool may be a critical time in walking development, as most children who have the ability to ambulate within the community do so between 2 and 5 years of age.3
The purpose of this case report was to describe and report the effect of an 8-week clinic-based, individualized, progressive treadmill training program on the ambulatory skills of a 4-year-old child with myelomeningocele who did not demonstrate functional ambulation despite almost 2 years of exposure to orthotic-assisted standing and walking practice. A secondary aim was to report the effect of the program on the child's overall independent functional mobility.
DESCRIPTION OF CASE
The 4-year-old child had spina bifida (lesion level L4-L5), hydrocephalus, and a Chiari malformation. She was born after 39 weeks gestation. A spinal closure was performed at 1 day of age, and a shunt was placed at 5 days of age. From the time of the original shunt placement until the initiation of the treadmill training program, the child had 8 shunt revisions. She had complications with her most recent shunt revision that occurred 5 months before she started the treadmill training program. The preschooler experienced bleeding into her ventricles, causing a seizure. External ventricular drains were placed to allow the blood to drain. Other pertinent surgical history included a Chiari decompression surgery and placement of a gastrostomy tube at 3 months of age secondary to vocal cord paralysis.
At the initiation of the treadmill training program, the preschooler did not demonstrate functional ambulation as defined by Hoffer et al.25 She was able to use a posterior walker and bilateral ankle-foot orthoses (AFOs) to ambulate short distances with standby assistance in therapy sessions and at her house. Her primary means of indoor and outdoor mobility, however, was propelling a manual wheelchair or creeping. Table 1 provides a history of her walking development.
The preschooler's parents expressed a goal of improving her speed, endurance, and steadiness within her walker. The family's goal was reasonable, given that 80% of children with lower-level lumbar lesions (L4-L5) gain community ambulation abilities with the mean age being 3 years 10 months.3
Prior to the initiation of the treadmill training program, the child had been participating in weekly outpatient physical therapy services that included overground ambulation, strength, postural control, and motor planning training without recent improvements in her ambulatory skills. The preschooler's young age and distraction level during physical therapy led to frequent stops and starts with overground ambulation training and the other interventions. Although dosage required for change among children with disabilities is not well defined, it is believed that training must be completed at a sufficient intensity, frequency, and duration to be effective.26 The preschooler's frequent stops and starts made it difficult for her to reach the level of consistency, intensity, and duration of training believed to be necessary to make improvements.
Use of a treadmill can provide task-specific stepping practice in an environment where the training parameters, including speed, duration, and consistency of stepping, can be controlled. Control over training parameters is important because task-specific training induces changes in the spinal reflexes that are dependent on the specific behavioral demands of the task.27 Treadmill training has shown promising initial results with regard to improving the ambulatory abilities in children with developmental disabilities associated with delayed or abnormal ambulation.6,8–20 Therefore, treadmill training was the intervention chosen to improve the preschooler's ambulatory abilities. With more controlled training parameters that encouraged her to work at a higher intensity on the specific task of stepping, it was hypothesized that the child would gain more ambulatory abilities.
Treadmill training provides task-specific training, which is important for neural plasticity and motor learning.27–29 In addition, the specific protocol used in this case report increased in intensity based on fatigue level and therefore had the potential to improve aerobic capacity and/or strength. Furthermore, a question exists as to whether treadmill training can improve general motor development in children at risk for neuromotor delays.17 Thus, outcome measures used in this case report were chosen to identify changes in ambulatory ability, aerobic capacity, and general motor function. They included the 2-minute walk test (2MWT), the Timed “Up and Go” (TUG), and the mobility section of the Pediatric Evaluation of Disability Index (PEDI). Strength was not directly assessed because the reliability and validity of hand-held dynamometry and manual muscle testing have not been investigated in the spina bifida population in children younger than 5 years.30
The 2MWT measures the distance walked in 2 minutes at a comfortable pace and has been shown to be a reliable and valid measure of aerobic capacity and functional mobility.31,32
The TUG is a reliable and valid test used as an outcome measure to assess functional ambulatory mobility in children as young as 3 years of age with disabilities.33 The TUG requires the child to rise from a seat, stand momentarily, walk 3 m, turn, return to the same seat, and sit down. It was performed with the modifications recommended by Williams et al33 for use in the pediatric population. The modifications included (1) asking the child to touch a target as compared with the more abstract instructions of the standard TUG, (2) repeating the instructions during the test, (3) using a seat with a backrest but without arms that allowed for the child's knees to be in 90° of flexion and her feet to be flat on the floor, (4) allowing the child to behave spontaneously, (5) starting the time as she left the seat, rather than on the instruction to “go,” and (6) stopping the time when her buttocks touched the seat to measure only movement time.33 The child used a posterior walker and bilateral AFOs to complete both the 2MWT and the TUG.
The mobility functional skills section of the PEDI was used to report change in overall independent functional mobility. The PEDI is a reliable and valid clinical assessment instrument that samples key functional capabilities and performances in children from 6 months to 7.5 years of age.34–37 It significantly correlates to neurological level, walking ability, and independence in activities of daily living in children with spina bifida.7
Baseline testing of the 2MWT and the TUG was completed 1 week before the start of the treadmill training program. The baseline PEDI was completed on the first day of training by the child's mother. Postintervention testing was completed during the last day of training. During the 6 weeks following the discontinuation of treadmill training, the child had only 1 physical therapy visit due to a family vacation and the visit did not include the use of the treadmill. Therefore, the 2MWT and the TUG were repeated 6 weeks after completion of the training program to determine whether she maintained the improvements overtime without direct therapeutic intervention.
DESCRIPTION OF INTERVENTION
The preschooler completed a modified version of the treadmill training program studied by de Groot et al.22 de Groot et al22 used a home-based, individualized, progressive treadmill training program performed 2 times per week for 12 weeks. Speed of walking and heart rate were measured during a 6-minute walk test (6MWT) performed over ground prior to the initiation of treadmill training. The 6MWT measures the distance walked in 6 minutes at a comfortable, self-selected pace.38 The child's speed on the 6MWT was used to set the initial treadmill speed as described in step 1 of the treadmill training progression plan given in Table 2.
On the first day of training, the children in the de Groot et al22 study walked for 2 minutes at 70% of their walking speed on the 6MWT, followed by 4 minutes at 100% of their walking speed on the 6MWT. This interval was repeated 3 times for a total of 18 minutes of treadmill training. The training was progressively increased in intensity following a treadmill training progression plan given in Table 2. Progression to the next level of training was based on fatigue level as measured by heart rate and the OMNI Rating of Perceived Exertion. The OMNI Rating of Perceived Exertion is a 0 to 10 scale of perceived physical exertion.39 The children moved to the next level of intensity on the treadmill training progression plan when their heart rates fell below 66% of peak heart rate measured during the 6MWT and/or their scores reached 5 or below on the OMNI Rating of Perceived Exertion.
The treadmill training protocol followed by the child in this case report was based on the de Groot et al22 protocol but was modified to meet her individual needs. Figure 1 provides an overview of the treadmill training protocol followed by the child in this case report.
Many of the components of the de Groot et al22 treadmill training protocol were used in the current protocol, including (1) use of an overground walking test prior to treadmill training to calculate walking speed and heart rate, (2) use of walking speed on the pretraining overground walking test to set the initial treadmill speed as specified in step 1 of the treadmill training progression plan (see Table 2), (3) use of heart rate as a measure of fatigue and an indication of the need to move to the next level of intensity, (4) progression of training intensity using the same treadmill training progression plan described by de Groot et al22 and given in Table 2, and (5) use of a training frequency of 2 times per week.
To individualize the treadmill training program to meet the unique needs of the child in this case report, the following modifications were made to the de Groot et al22 protocol: (1) the 2MWT was used to calculate heart rate and speed rather than the 6MWT; (2) heart rate alone was used to signal the need to progress to the next level of intensity of training versus a combination of heart rate and/or OMNI Rating of Perceived Exertion; (3) progression to the next level of intensity on the treadmill training progression plan occurred when heart rate fell below 70% versus 66% of peak heart rate; (4) manual palpation of radial pulse was used to determine heart rate versus a heart rate monitor; (5) the training duration was reduced to 8 weeks from 12 weeks; and (6) the treadmill training was completed in the clinic versus home environment.
The child's speed of walking and peak heart rate were obtained prior to treadmill training during a 2MWT performed over ground rather than the 6MWT used in the de Groot et al22 protocol because of the child's limited walking abilities preventing her from completing the 6MWT. The 2WMT and the 6MWT have a high intertest correlation when used for walking assessment in adults with neurological impairments,31,32 and heart rate is an acceptable tool to evaluate exercise capacity in children with spina bifida.40
Heart rate alone was used to progress the training program rather than a combination of heart rate and the OMNI Rating of Perceived Exertion because the validity of the OMNI Rating of Perceived Exertion has not been studied in children younger than 6 years.39 Therefore, the preschooler in this case report was progressed to the next level of intensity when her heart rate reached 70% of her peak heart rate on the 2MWT.
The program was progressed in intensity when the child's heart rate fell below 70% of her peak heart rate on the 2MWT rather than 66% used in the de Groot et al22 protocol. Because of the shorter duration of the 2MWT, the heart rate measured as her maximum might not have reached the same level as those individuals in the de Groot et al22 study, where heart rate was measured during the longer 6MWT. To ensure that the intensity of the program was high enough to produce physiological benefits, the child's training program was progressed at a higher percentage of her maximum heart rate. Exercise intensities used in programs designed to improve walking ability or aerobic fitness in children with disabilities range from 50% to 80% of maximum heart rate.41–43
The child did not tolerate the use of a pulse oximeter and a heart rate monitor, thus heart rate was measured via manual palpation of her radial pulse. It was measured for 30 seconds at rest, following the 2MWT, and after each repetition on the treadmill. Thirty-second intervals have been found to be the most accurate and efficient time period to manually count radial pulse.44
The training program was reduced in duration from 12 to 8 weeks to accommodate the family's scheduling needs while still following pediatric training recommendations of 6 to 16 weeks.26 Training was completed within the clinic setting rather than in the home setting. The family did not own a treadmill for home use, and the child required use of the WalkAble pediatric LiteGait WK 100 device for safety.
The child had no additional therapy services or physical activities during the 8 weeks of treadmill training. The family was instructed to practice stepping daily within her posterior walker at home during both the treatment and postintervention periods. They reported practicing stepping for 10 to 15 minutes per day but did not keep a systematic record of the practice.
Six-Week Postintervention Period
During the 6-week postintervention period, the child did not have any physical therapy services or additional physical activities with the exception of one, 45-minute physical therapy session, that focused on strengthening and overground ambulation but did not include the use of treadmill training. This physical therapy session occurred 5 weeks after the completion of the treadmill training when the family returned from vacation. Outcome measures for this case report could not be completed during the first sessions because of time constraints and the child's fatigue from taking a late night flight across the country the night before, adjusting to a time change, and having a magnetic resonance scan on the day of the physical therapy visit. The retention measurements were taken at the next physical therapy session, which occurred 6 weeks after the completion of the treadmill training.
The WalkAble pediatric LiteGait WK 100 device was used over a zero-start Nautilus NTR 700 treadmill. The child did not use the harness typically worn with the WalkAble but did use the hand railings. The child was too heavy to be manually suspended over the treadmill, as has been described by other authors.15,16,21 The child did not use the harness typically worn with the WalkAble because it was difficult to get her to bear weight through her lower extremities when wearing the harness. Therefore, the hand railings, but not the harness, were used to assist with balance and provide a safe stepping environment that encouraged weight-bearing through her lower extremities. The use of the hand rails also follows a task-oriented approach in which the training matches the functional goal.45 Because the primary goal of the treadmill training was to improve her ambulatory skills with her walker, use of the hand railings on the treadmill provided an environment that closely simulated the child ambulating with her walker. The therapist provided contact guard assistance at the child's hips for safety.
Moerchen et al21 recommend that treadmill training in the spina bifida population be attempted in the following order: barefoot, shoes, orthoses (plus shoes), and with weights to determine under which conditions a child can step. Use of orthoses can create a constraint to range of motion, add additional weight, and alter the sensory input, therefore barefoot or only the use of shoes is preferred.21 However, the child in this case report was not able to safely step without the use of her AFOs. Therefore, she wore tennis shoes and bilateral AFOs throughout the training program.
DESCRIPTION OF OUTCOMES
The preschooler completed 15 of 16 (93.75%) planned treadmill training sessions. She averaged 47.5 minutes of treadmill training time per week and reached step 8 on the treatment protocol. It should be noted that because of the child's slow walking speed on the 2MWT, progression to the next level of the program did not always result in an increased speed of the treadmill. For example, in step 3, she walked at 110% of her walking speed on the 2MWT, which was 0.4873 miles per hour (mph). In step 4, she walked at 120%, which was 0.5316 mph. Because the treadmill adjusted in 0.1-mph increments, both were rounded to 0.5 mph. Table 3 provides details of the specific treadmill training program followed by the child in this case report.
The preschooler demonstrated improved ambulatory abilities as measured by the 2MWT and the TUG immediately after the 8-week treadmill training program. She maintained the improvements at the 6-week follow-up without physical therapy, additional treadmill training, or the start of a new activity. Table 4 provides a summary of the results.
Changes in overall functional mobility were measured using the functional mobility section of the PEDI. A summary of the results of the PEDI is available in Table 5. The preschooler did not demonstrate a significant improvement in her overall functional mobility, and she continued to have significant delays as compared with her peers.
The aim of this article was to describe and report the effect of an 8-week clinic-based, individualized, progressive treadmill training program on the ambulatory abilities of a 4-year-old child with spina bifida. The treadmill training program provided task-oriented ambulatory training that incorporated pediatric exercise training principles.
The results of this case report demonstrated that the use of the treadmill training program described here appeared to have improved the child's ambulatory skills as measured by the 2MWT and the TUG. She appeared to have maintained these improvements over a 6-week period of time without additional physical therapy or treadmill training intervention.
Although she did not demonstrate a significant improvement in her functional mobility score on the PEDI, it should be noted that the 2 areas of improvement on the PEDI were in the indoor locomotion distance and speed section. After the training, she was able to move within and between rooms with difficulty, which she was not able to perform before the treadmill training.
The fact that the preschooler in this case report appeared to improve her ambulatory abilities, but not her overall functional mobility as measured by the PEDI, could be secondary to the specificity of treadmill training. Treadmill training provides repetitive stepping practice in an attempt to improve ambulatory ability. This is consistent with the training principle of task specificity, which refers to training the specific task that you would like to improve. The child practiced the specific skill of stepping and showed improvement in her ambulatory skills within her posterior walker as measured by the 2MWT and the TUG. Although she improved her indoor locomotion distance and speed on the PEDI, she did not improve on other functional mobility skills that she had not been trained to complete such as toilet transfers, chair transfers, car transfers, and stair climbing.
Comparison With the de Groot et al22 Study
The child in this case report completed 93.75% of the scheduled treadmill training sessions as compared with a mean of 94.17% sessions completed by the treadmill training group in the de Groot et al22 study. She averaged 47.5 minutes of treadmill training per week as compared with the 49.4 minutes averaged by the participants in the de Groot et al22 study but had less total treadmill training time because she completed only an 8-week program.
Despite the fact that the child had less ambulatory abilities and used a modified version of the treadmill training program studied by de Groot et al,22 she had a similar improvement in walking speed. The children who completed the treadmill training program in the study conducted by de Groot et al improved walking distance by 38.7 m on the 6MWT. This translates into ambulating an additional 21.16 ft per minute or an increase in walking speed of 0.24 mph. In this case report, the 2MWT was used and the child walked an additional 25 ft per minute or an increase in walking speed of 0.284 mph.
There were a number of limitations in this case report. The primary limitation was that the same physical therapist performed the assessments and treatments, which could have introduced bias into the results. Second, given that this was a case report and not a controlled study, it was not possible to determine whether the improvements in the child's ambulatory abilities were the direct result of the treadmill training program or due to other external causes. In addition, the outcome measures used were capacity-based rather than performance-based, with the exception of the PEDI. No direct measure of her ability to ambulate within the home or community setting was evaluated. The outcome measures used were not designed to identify the physiological cause for the improvements noted. Outcome measures that can differentiate central nervous system changes, strength changes, and cardiovascular changes would be helpful in identifying the mechanism for change. Breaks in the planned protocol were made on training days 5, 6, and 8, which may have affected the results. Furthermore, the time spent performing overground gait practice was not systematically measured, so the effect on the improvements found in the preschooler's ambulatory abilities could not be evaluated. In addition, completion of 1 physical therapy session during the 6-week postintervention period of time may have confounded the retention results. Finally, the 2MWT was used rather than the 6MWT because of the child's more limited ambulatory abilities. While the 2WMT and the 6MWT have a high intertest correlation when used for walking assessment in adults with neurological impairments, the 2MWT has not been evaluated for use in the pediatric population.31,32
Although this case report has a number of limitations, it demonstrates that a protocol, which was found to increase ambulatory abilities in a previous study, can be modified and applied for a child with the same diagnosis but different needs and functional level.
FUTURE IMPLICATIONS AND CONCLUSIONS
This case demonstrates that an 8-week clinic-based, individualized, progressive treadmill training program might have improved the ambulatory abilities of a 4-year-old with an L4-L5 lesion and associated conditions who did not demonstrate functional ambulation prior to the intervention. Although the protocol was tested with just 1 child, the protocol modified according to de Groot et al22 was individually progressed such that application to other children with similar ambulatory experience and needs might be possible.
The use of treadmill training in children with spina bifida, aged 2- to 6-year-old, had not been formally investigated at the time of this case report. The positive results in this case support further investigation into the use of treadmill training in children with spina bifida who are at a critical time in their walking development, using more rigorous randomized controlled study designs. It is important to investigate the effects of treadmill training on preschool-aged children with spina bifida because most children who have the potential to achieve community ambulation begin walking between 2 and 5 years of age.3
Further investigation into the ideal candidate for treadmill training is needed. This case demonstrated treadmill training with a child without functional ambulation might have improved her ambulatory abilities using a posterior walker. Moerchen et al21 provided initial support for the use of the treadmill in young children to work toward ambulation with an assistive device, and de Groot et al22 demonstrated that treadmill training improved the ambulatory skills in children who were able to demonstrate community ambulation. Studies designed to examine the effects of treadmill training on ambulatory function with children at various ambulatory abilities are needed.
The protocol developed for this case was based on training principles that have been used with children with disabilities. However, these training principles have not been specifically examined within the spina bifida population and the recommendations include wide ranges of values. For example, duration training recommendations range from 6 to 16 weeks,26 intensity recommendations range from 50% to 80% maximum heart rate,41–43 and locations vary from the clinic setting to the home setting. Therefore, randomized controlled trials that compare various intensities, durations, and locations of training would be beneficial.
Other possible research opportunities include the use of the treadmill to prevent the loss of ambulatory abilities,3 investigation into the mechanisms underlying changes observed with treadmill training, and studying the ideal combination of overground and treadmill training.
The authors thank Jessica Brock, PT, DPT, for collecting many of the research articles used in the introduction of this article. The authors also thank William L. Rice III, PT, MPT, for his contribution to the planning stages of this article.
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