This is a pilot study to evaluate the feasibility and effectiveness of a telerehabilitation intervention for youths with AMC.

Youths aged between 8 and 21 years with a confirmed clinical diagnosis of multiple congenital contractures, or AMC, will be invited to participate. Inclusion criteria include the ability to communicate in English or French and residence in Canada. Youths living in another country will be excluded as rehabilitation therapists participating in this study hold only a Canadian professional license. To align with postintervention precautions, those having undergone a recent surgery (ie, 3 months for soft tissue and 6 months for bony surgery) will be excluded. Youths with cognitive deficits or unstable health will also be excluded, to ensure participants can take part in the HEP. To assess the level of cognition, medical records will be reviewed for the presence of central nervous involvement or intellectual impairment. In addition, the treating physician, therapists, and parents will be consulted to determine eligibility based on type of schooling and ability to follow instructions. No restriction will be made about the degree of severity of contractures or physical impairment.

Youths with AMC followed at Shriners Hospital for Children - Canada (SHC-C) will be recruited during their clinic visit. Potential youths with the appropriate clinical diagnosis who do not have an upcoming visit to SHC-C will be contacted by postal mail and phone. For youths with AMC who are not followed at SHC-C, an advertisement describing the study will be posted on social media of a Canadian AMC support group (ie, Facebook). Those interested in participating will be asked to contact the clinical research coordinator for more information. Prior to participating in the study, informed consent will be sought from parents as well as from youths aged 14 years and older, as per provincial regulations. Youths between the ages of 8 and 13 years will be asked to provide assent. For these younger participants, the research team will encourage parents to be available during the telerehabilitation sessions to provide support during the sessions and throughout the HEP. As we expect to complete this study on 10 youths, 13 youths will be recruited to account for a 30% dropout rate [19].

A rehabilitation team including a physical therapist, an occupational therapist, and a physical rehabilitation therapist will collaborate to provide the intervention. A physical rehabilitation therapist is defined as a professional among the interdisciplinary team whose role is to obtain necessary prerequisite information from a leading physical therapist in order to develop treatment plans and provide appropriate interventions to reduce activity limitations and participation restrictions.

The intervention will consist of a 12-week HEP. Prior to the assessment with the therapists, youths will complete an online questionnaire consisting of the Physical Activity Questionnaire for Adolescents (PAQ-A), the Pediatrics Outcomes Data Collection Instrument (PODCI), and demographic questions. The occupational therapist and physical therapist will conduct the initial assessment of each youth using ZOOM Pro (Zoom Video Communications, Inc), a videoconferencing platform allowing an encrypted connection. During this assessment, the therapists will perform an active joint range-of-motion (AROM) assessment of the upper and lower limbs, evaluate overall function, assess pain using the Adolescent and Pediatric Pain Tool (APPT), and establish individualized goals with each youth using the Goal Attainment Scale (GAS). The information gathered through this initial assessment will be used by the rehabilitation team to develop an individualized 12-week HEP. A week after the initial assessment, the physical rehabilitation therapist will exchange with the youth using ZOOM to explain and deliver the HEP as well as ensure comprehension and safe execution of the exercises. Youths will be asked to perform their HEP three times a week for approximately 15-30 minutes each time. They will be sent a physical activity monitor (POLAR watch A370) by postal mail and will be instructed to wear it on their wrist during HEP sessions, as it will be used to capture heart rate and the duration of the exercise sessions. Youths will have the possibility to wear the physical activity monitor at all times if they desire. If needed for the HEP, exercise materials such as resistant elastic bands or TheraPutty will also be sent to them by postal mail. A follow up with the physical rehabilitation therapist will be provided through ZOOM every 3 weeks (ie, at weeks 3, 6, and 9) to address any questions and to adjust the HEP as needed. At the end of the 12-week intervention, the occupational therapist and physical therapist will re-evaluate the youth using the same outcome measures as the initial evaluation. Youths will be asked to complete the same questionnaire, with the addition of questions about their satisfaction regarding the intervention. Figure 1 provides a summary of the intervention. The outcome measures used in this study are described in the following sections. In addition, parents will be asked to complete a cost questionnaire (direct and indirect costs incurred in relation to their child’s condition), which will be described elsewhere as it extends beyond the scope of this study.

Given this is a pilot study with a small sample size and a heterogeneous population, nonparametric and descriptive statistics will be used. To assess feasibility, the compliance to the HEP and to the telerehabilitation meetings, recruitment and completion rates, missing data, and closed-ended questions on satisfaction will be analyzed using descriptive statistics and compared with the established feasibility criteria. Table 1 describes the feasibility criteria for operationalization that will be used in the study. Youths’ experience about use of technology and overall satisfaction with the program will be reported as collected in the open-ended questions. The cost of the telerehabilitation intervention (ie, therapists’ time, federal professional license, and teleconferencing cost), recruitment source, and technical issues experienced during the program will be described. For the effectiveness of the HEP, raw GAS scores will be converted to GAS T-scores for each youth and for the global HEP. Preintervention and postintervention data of the range of motion, APPT, PAQ-A, and PODCI will be compared using the Wilcoxon signed-rank test to measure change among the same youth.

The proposed telerehabilitation pilot study is designed to provide an HEP to youths with AMC. Establishing the feasibility of this study has the potential to inform service delivery models using technology, thereby overcoming geographical barriers, to provide an individualized HEP to youths living with a rare condition.

To date, little is known about the potential effectiveness of exercises in adolescents with AMC [5]. Using this type of technology in a study that includes frequent follow ups as proposed in this protocol has the potential to reach more participants because it transcends geographical barriers, improving the sample size needed for research with heterogeneous populations such as AMC. Telerehabilitation allows inclusion of youths from across Canada, regardless of proximity to a specialized health care center. Although telerehabilitation has previously been used in studies on children with asthma and autism spectrum disorders [26,27], the only musculoskeletal disorder in which a telerehabilitation intervention was used is with children with cerebral palsy [18]. However, the results were variable among those studies which justify studies with other populations such as AMC [28,29]. In addition, little is known about the cost efficiency of this type of intervention [26,30].

This pilot study has some limitations. Because of the remoteness of the assessment, even if passive range of motion is normally measured in the clinic, it will not be assessed in this study. Despite passive range of motion being important to measure, it is expected that therapists will have sufficient information to develop the individualized HEP from the initial telerehabilitation assessment. Another consideration is that youths will be instructed to wear the activity monitor on their wrists, even in the presence of upper extremity contractures that may interfere with the movement capture, therefore with the accuracy of step count. As the purpose of the activity monitor in this study is to measure heart rate and duration of the exercise sessions, and not to record steps, all youths will wear the watch at the wrist. Importantly, the goal of using telerehabilitation is not to replace face-to-face clinic visits or in-person therapy, but rather to propose a complementary intervention therapists can offer youths and their families during adolescence, at which time rehabilitation has been shown to decrease [12]. The aim to provide an individualized HEP is to help youths maintain their physical gains and assist them in reaching new goals that may arise as they grow. This telerehabilitation pilot study will also inform the possible pitfalls, beneficial effects, and cost associated with this new method of care, thus informing its use in other populations with musculoskeletal conditions.

Establishing the feasibility of using telerehabilitation for children with AMC will inform us for a future clinical trial. Information about the potential effectiveness of using telerehabilitation to deliver an HEP for children with AMC will be provided, thus leading to the development of a novel approach in this population. If proven successful, this service delivery model can be tailored to other pediatric musculoskeletal conditions, such as osteogenesis imperfecta and juvenile rheumatoid arthritis.