The research design of this study was a randomized control trial. It was approved by the Ethics Review Committee of the Ethics Review Committee of the Faculty of Applied Medical Sciences - Umm Al Qura University. This trial was registered in the ClinicalTrial.gov PRS (NCT04990193). Children’s parents signed a consent for permitting the engagement of their children in this study. The study was proceeded according to the Declaration of Helsinki of 1975, as revised in 2013. The study was conducted in the Department of Physical Therapy - Umm Al Qura University. The involved children were recruited from kingdom of Saudi Arabia - western region. The organization responsible for the integrity, conduct and financial support of this study was the Deanship of Scientific Research at Umm Al Qura University - Grand Code (19-MED-1-01-0004).
Children of a confirmed diagnosis of dyskinetic cerebral palsy of choreoathetosis type were selected to participate in this study. The diagnosis was confirmed by magnetic resonance images obtained from medical records or personal physicians. These children (of both genders) were between 12 and 16 years old. Their height and weight were more than 100 cm and 20 kg, respectively, to be eligible for the evaluation process on the Biodex stability system.
Children were able to comprehend and follow orders. Children with level I and II Gross Motor Functional Classification System (GMFCS) were recruited to participate in the study . They were able to walk 10 m openly without any orthoses. So that they were able to perform the gait evaluation procedures. Participating children were not subjected to any other physical therapy programs throughout the study period except the assigned treatment protocol.
The children were excluded from this study if they had fixed spinal deformities interfering with their spinal and limb functional mobility. Children were also excluded if their skin were sensitive or inflamed to any materials used. Children who had seizures, perceptual disorders, visual problems, and auditory deficits did not participate as well.
Sample size and randomization
A sample size of 40 participants was calculated for this study based on a preliminary power analysis (power = 0.8, α = 0.05, effect size = 0.5, groups = 2 and evaluation times = 2) [10,11,12,13]. The total number of participants was raised to 56 for anticipation of withdrawals. The participants were divided equally into two groups: control and study groups.
The randomization process was performed by an independent person who was blinded to the study procedures. It was performed simply by adding a specific identification number for each participating child. SPSS program (version 25) was used to randomly assign children to both groups.
Assessments of the children with dyskinesia were conducted by blinded evaluators who didn’t participate in the application of intervention and were also blinded from the randomization process. The evaluation was carried out to assess the following outcome measures: (1) Overall, anteroposterior and mediolateral stability indices (%) of Postural Stability Test from standing position at the static mode of platform tilt, (2) Pediatric Balance Scale score, and (3) gait parameters including step length (cm), gait cycle time (s), cadence (steps/min) and velocity (m/s). The evaluation process was conducted at both baseline and immediately after 12 weeks of intervention.
Biodex Balance System SD (BBS; Biodex Inc., Shirley, NY) measured stability indices (%) of postural stability test from standing position at the static mode of platform tilt with high reliability and validity . The postural stability indices were measured to evaluate the children’s capabilities to control their balance in different directions. The higher the indices values (%) were recorded, the less stability the children had, and the children had balance control difficulties.
The Pediatric Balance Scale is a valid and reliable test to assess and re-assess balance deficits in children with mild and moderate motor disabilities. The test includes fourteen tasks that evaluate balance abilities and motor performance in children. 0–4 is the rating score for each item, where 0 is the lowest score and 56 is the highest possible score for all tasks that indicate the best balance and motor performance ever .
An electronic walkway (GaitRite , CIR Systems Inc., New Jersey, USA) connected to a portable computer was used to measure the gait parameters (step length, gait cycle time, cadence, and velocity). The length of the GaitRite walking mat was about 5 m. The electronic walkway GaitRite is a reliable and valid tool for analyzing spatial and temporal gait parameters in children with cerebral palsy and Gross Motor Function Classification System—level I or II . Children were requested to walk barefoot along the walkway. Each participant was asked to start walking 3 m far from the walking mat to reach his/her natural walking pattern before the actual measurement. Three trials were performed and the average measurment of them was calculated.
Children in both groups (control and study) received the conventional therapeutic exercise training protocol, which was designed, to improve trunk steadiness during standing and walking. The exercise training protocol for both groups is fully outlined in Appendix 1
The conventional therapeutic exercise training protocol for every child was three sessions per week for 12 consecutive weeks. The interventions were performed by two experienced physical therapists. Every treatment session was conducted for 2 h with a 15-min rest between the two training hours.
The children in the study group received the conventional exercise protocol given to the control group. Moreover, they wore TheraTogs orthotic undergarment with the strapping system 8 h every day for 12 consecutive weeks.
A TheraTogs orthotic undergarment and its strapping system developed by Cusick, 1997 is composed of a sleeveless Tank-Top (vest) and two shorts (Hipster), each with two thighs and limb cuffs . All parts are fabricated from nylon and spandex with a foam layer and a variety of elasticized straps. They are designed to be worn directly on the skin as undergarments that allow unobstructed toileting. A TheraTogs system provides the wearer a vertical compression and stiffness to support posture, and reinforce stability.
Each child had his/her own TheraTogs orthotic undergarment and strapping materials. It was not allowed for any child to share his/her orthosis with others to avoid any infectious disease transfer. In this study, the participating children wore the TheraTogs and strapping system under their regular dresses, so it was not annoying or disturbing.
A pre-training period, before starting the treatment program, was given to all participating children for preparing them to get used and endure the wearing of the TheraTogs orthotic undergarment and strapping system. The pre-training period lasted for 14 days. During which the wearing period gradually increased from 1 h at the beginning of the course until reached 8 h by the fourteenth day. The rate of wearing period increment was 1 h every 2 days. This pre-training period was completed 1 week before starting the treatment program. If any participating child failed to follow this regimen, the training was repeated again until the child reached the 8 h of daily wearing the TheraTogs system. This pre-training course was completed one week before starting the treatment program.
Throughout this pre-training period, the therapist who is responsible for the strapping protocol trained one of the child’s family members on (1) how to fit the TheraTogs orthosis to his/her child, and (2) how to apply the prescribed strapping technique for the involved body parts. By the end of this training, the therapist made sure of the skills of this member in the application of the TheraTogs strapping protocol before the actual beginning of the treatment program.
It was not allowed to start the treatment program with TheraTogs and strapping until making sure of the family member’s skill and proficiency in providing the prescribed TheraTogs protocol. Any member who was not successful in this preparatory course was retrained again. The therapist daily followed up the process of wearing TheraTogs and strapping by telephone with this member and made sure that the process was completed successfully. This daily communication continued throughout the treatment period (12 weeks).
The fitting and strapping techniques of the TheraTogs system for all participating children in the study group are fully outlined in Appendix 2.
The statistical analysis for all recorded scores pre-and post-treatment was reported as mean ± standard deviation (SD). The Shapiro-Wilk test and the Levine's test were used to test the variables' normal distribution and variances homogeneity, respectively. The data of this study were homogeneous and normally distributed. The comparison between both groups, pre-and immediately post-treatment, was carried out by using unpaired t test. Recorded data were analyzed by using SPSS computer program version 25.00. P values less than 0.05 were accepted to represent a statistical significance.