BACKGROUND
Osteogenesis imperfecta (OI) is a rare bone fragility disorder associated with muscle weakness. Individuals with OI may therefore benefit from exercise interventions aiming to improve muscle and bone strength. Given the rarity of OI, many patients do not have access to exercise specialists who are familiar with the disorder. As such, telemedicine, the provision of healthcare through technology to provide care at a distance, may be well suited for this population.
OBJECTIVE
(i) to investigate the feasibility and cost-effectiveness of two telemedicine approaches for the delivery of an exercise intervention for youth with OI and (ii) to assess the impact of the exercise intervention on muscle function and cardiopulmonary fitness in youth with OI.
METHODS
Patients with OI type I (mildest form of OI; n=12, ages: 12-16 years old), from a pediatric orthopedic tertiary hospital, will be randomized to receive a twelve-week remote exercise intervention either in the (i) supervised group (n = 6), monitored every session, or (ii) follow-up group (n = 6), receiving monthly progress update appointments. Participants will undergo the following pre- and post-intervention evaluations: sit-to-stand test, push-up test, sit-up test, single-legged balance test, and a heel-rise test. Both groups will be given the same twelve-week exercise regimen which includes cardiovascular, resistance, and flexibility training. For each exercise training session involving the Supervised group, a kinesiologist will provide instructions to participants through live video sessions using a teleconferencing application. On the other hand, the Follow-up group will be discussing their progress with the kinesiologist every four weeks over a teleconferencing video call. Feasibility will be assessed by recruitment, adherence, and completion rates. A cost-effective analysis of both approaches will be computed. Changes in muscle function and cardiopulmonary fitness will be examined between the two groups, pre- and post-intervention.
RESULTS
It is anticipated the supervised group will have higher adherence and completion rates compared to the follow-up group, which may be associated with greater physiological benefits, however, it may not be as cost-effective compared to the follow-up approach.
CONCLUSIONS
By determining the most feasible telemedicine approach, the current study may serve as a basis for providing increased access to specialized adjunct therapies for individuals with rare disorders.
CLINICALTRIAL