The Development of Patient-Reported Outcome Measures and an Observer-Reported Outcome Measure for Pediatric Patients with Inherited Retinal Disease
Bibliographic record
Abstract
Objective: This study aimed to devise valid patient-reported and observer-reported outcome measures for inherited retinal diseases (IRDs) in pediatric patients and caregivers. Methods: Guided by a literature review and IRD specialist focus groups, measures were developed for patients aged 6 to <13 years. Interviews were conducted with 26 children and 23 caregivers, using thematic analysis and qualitative description methdology. Existing adult measures were validated in 91 adolescents (13 to <18 years) with IRDs. Results: Children and their caregivers each identified 16 themes focused on the impact of their condition on daily life and psychosocial health. The adult measures were validated for use in adolescents. Conclusion: The study underlines the need for tailored outcome measures addressing the experiences of adolescents, children with IRDs, and caregivers. These tailored tools offer valuable resources for eye care providers to optimize the management of IRDs in pediatric patients, including both children and adolescents.
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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 | 0.057 | 0.102 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.001 | 0.001 |
| Bibliometrics | 0.002 | 0.002 |
| Science and technology studies | 0.001 | 0.001 |
| Scholarly communication | 0.002 | 0.002 |
| Open science | 0.001 | 0.002 |
| Research integrity | 0.001 | 0.001 |
| Insufficient payload (model declined to judge) | 0.001 | 0.000 |
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".