Mortality in Children with Optic Nerve Hypoplasia/Septo-Optic-Pituitary Dysplasia
Bibliographic record
Abstract
AIM: To quantify optic nerve hypoplasia/septo-optic-pituitary dysplasia (ONH/SOD) all-cause mortality rate and risk of death in Manitoba, Canada. METHOD: A retrospective population-based study with a case-control design was undertaken using the Manitoba Population Research Data Repository. Cases were 124 ONH/SOD patients diagnosed during 1990-2019, matched to 620 unrelated population-based controls on year of birth, sex and area of residence. Both cases and controls were followed until March 31, 2022, or until they moved out of the province or died. Crude mortality rate was estimated. Cox proportional hazards models were used to test for differences in all-cause mortality between cases and controls. Hazard ratios (HR) with 95% confidence intervals (CIs) were estimated. RESULTS: percentiles) age of death of ONH/SOD patients was 4.6 (2.7-9.1) years. The median duration of follow-up was 12.0 years for the cases and 11.4 years for the controls. The crude mortality rate (95% CIs) was 3.7 (1.7-8.3) per 1000 person-years in patients with ONH/SOD and 1.0 (0.5-2.1) per 1000 person-years in unrelated matched controls. All-cause mortality was significantly higher in ONH/SOD patients compared to unrelated controls (HR = 3.7, 95% CI = 1.3-10.5). CONCLUSION: Patients with ONH/SOD have a higher risk of death compared to unrelated controls. Healthcare professionals should be familiar with the morbidities and comorbidities associated with ONH/SOD and the complications that may lead to their demise, since they can be managed to reduce the mortality risk.
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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 | 0.000 | 0.001 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.000 | 0.000 |
| Bibliometrics | 0.001 | 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) | 0.001 | 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".