Association between work productivity and characteristics of adults with X-linked hypophosphatemia: an analysis of the XLH disease monitoring program
Why this work is in the frame
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Bibliographic record
Abstract
Abstract X-linked hypophosphatemia (XLH) is a rare, genetic, progressive, phosphate-wasting disorder that causes skeletal morbidities, stiffness, pain, and impaired physical function. This study used baseline data from the XLH Disease Monitoring Program to evaluate relationships between work productivity and patient characteristics (demographics, medical history, patient-reported, and functional outcomes) in adults with XLH. Bivariate analysis guided the selection of variables for multivariate analysis after adjustment for multicollinearity and conceptual overlap. The analysis comprised 281 subjects (75.4% female; 80.8% from USA; median age 39.2 yr); 53.4% were employed full-time and 31.3% were not employed; 15.3% were receiving disability payments; 47.0% were taking burosumab at study entry. Most employed subjects were working full-time outside the home (69.9%) and in light or sedentary roles (59.6%). In multivariate analyses, patients with fewer orthopedic surgeries (odds ratio [OR] 0.88; 95% confidence interval [CI], 0.81–0.96; p=.002) and better Patient-Reported Outcomes Measurement Information System Physical Function scores (OR 1.08; 95% CI, 1.02–1.15; p=.013) were more likely to be in full-time employment than not employed. Younger patients (OR 0.97; 95% CI, 0.94–0.99; p=.014) and those with fewer orthopedic surgeries (OR 0.83; 95% CI, 0.73–0.95; p=.008) were more likely to be in medium than light or sedentary work. Those with worse WOMAC Pain scores were more likely to be doing heavy/very heavy than light or sedentary activity (OR 1.04; 95% CI, 1.01–1.07; p=.006). Full-time employment levels are low in adults of working age with XLH, and unemployment and disability payment rates are high, suggesting that XLH has a substantial impact on work productivity. Worse physical function and a greater number of orthopedic surgeries are associated with lower work productivity. Worse pain, higher number of orthopedic surgeries, and younger age are associated with heavier work roles; however, causality was not specifically investigated.
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Full frame distilled prediction
Teacher imitationNot calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.
Codex and Gemma teacher scores by category
| Category | Codex | Gemma |
|---|---|---|
| Metaresearch | 0.000 | 0.000 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.000 | 0.000 |
| Bibliometrics | 0.000 | 0.001 |
| Science and technology studies | 0.000 | 0.000 |
| Scholarly communication | 0.000 | 0.000 |
| Open science | 0.000 | 0.000 |
| Research integrity | 0.000 | 0.000 |
| Insufficient payload (model declined to judge) | 0.000 | 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 it