Association between work productivity and characteristics of adults with X-linked hypophosphatemia: an analysis of the XLH disease monitoring program
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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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 | 0.001 | 0.003 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.000 | 0.001 |
| Bibliometrics | 0.001 | 0.001 |
| Science and technology studies | 0.000 | 0.000 |
| Scholarly communication | 0.001 | 0.000 |
| Open science | 0.000 | 0.001 |
| Research integrity | 0.000 | 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".