JMIR Publications

ABSTRACT

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. 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.