Delivering a Home-Based Exercise Program to Youth With Osteogenesis Imperfecta: Protocol for a Comparative-Approach Study
Bibliographic record
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 health care through technology to provide care at a distance, may be well suited for this population. OBJECTIVE: The main objectives are (1) to investigate the feasibility and cost-effectiveness of 2 telemedicine approaches for the delivery of an exercise intervention for youth with OI and (2) to assess the impact of the exercise intervention on muscle function and cardiopulmonary fitness in youth with OI. METHODS: Patients with OI type I (the mildest form of OI; n=12, aged 12-16 years) from a pediatric orthopedic tertiary hospital will be randomized to receive a 12-week remote exercise intervention in either (1) a supervised group (n=6), monitored every session, or (2) a follow-up group (n=6), receiving monthly progress update appointments. Participants will undergo the following pre- and postintervention 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 12-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 discuss their progress with the kinesiologist every 4 weeks over a teleconferencing video call. Feasibility will be assessed by recruitment, adherence, and completion rates. A cost-effectiveness analysis of both approaches will be computed. Changes in muscle function and cardiopulmonary fitness will be examined between the 2 groups, pre- and postintervention. RESULTS: It is anticipated that 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, this study may serve as a basis for providing increased access to specialized adjunct therapies for individuals with rare disorders. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID): PRR1-10.2196/40262.
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How this classification was reachedexpand
Full frame machine prediction
Teacher imitationNot calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.
Distilled classifier scores by category (both heads)
| Category | Codex | Gemma |
|---|---|---|
| Metaresearch | 0.027 | 0.018 |
| Meta-epidemiology (narrow) | 0.004 | 0.002 |
| Meta-epidemiology (broad) | 0.007 | 0.004 |
| Bibliometrics | 0.003 | 0.003 |
| Science and technology studies | 0.004 | 0.002 |
| Scholarly communication | 0.002 | 0.002 |
| Open science | 0.004 | 0.002 |
| Research integrity | 0.006 | 0.005 |
| Insufficient payload (model declined to judge) | 0.057 | 0.008 |
Machine scores (provisional)
The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.
Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.
score_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from itClassification
machine, unvalidatedMachine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.
How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".