Airway obstruction in Robin sequence: Evaluating longitudinal evolution
Bibliographic record
Abstract
Airway obstruction of various degrees is one of the most important problems in infantswith Robin sequence. Various tools are used to evaluate airway obstruction in these infantsand monitor treatment, but polysomnography is the gold standard. Routine use ofpolysomnography is however limited by cost and availability. The general purpose of thestudy was to document the evolution of airway obstruction in the first 18 months of life in acohort of infants afflicted with Robin sequence. More specifically, we wanted to determinewhether night-time pulse oximetry, an inexpensive and widely available technology, could beused as a surrogate for polysomnography in identifying obstructive apnea in infants withRobin sequence.METHODSThis is a retrospective cohort study for all patients admitted in the neonatal periodwith a final diagnosis of Robin sequence at the Montreal Children’s Hospital from 2003 to2018. All data was thoroughly review from medical records for infants qualifying as Robinsequence: association of micro-retrognathia, glossoptosis and airway obstruction. We alsoreviewed all polysomnography and oximetry test and extracted the following standard data:Apnea hypopnea index (AHI) and mixed obstructive AHI (MOAHI) from polysomnography andDesaturation index (drops by ≥3%/hour, DI ≥3%; drops by ≥4%/hour, DI ≥4%; and time spent<90%) from the oximetry done at the time of polysomnography. A MOAHI ≤5 events/hourwas used to separate infants with no/mild obstruction from those with moderate/severeobstruction (MOAHI >5 events/hour).Analysis of data for the correlations was obtained through linear regression modelswith prediction equations; agreement was judged using the intraclass correlation coefficientand Bland-Altman plots. Next, generalized linear models and receiver-operator characteristiccurves were used to determine the diagnostic accuracy of the desaturation index and to selecta cut-off value. Statistical significance was set at a p value of <0.05.RESULTSThe cohort of patients was comprised of sixty infants (34 males, 56.7%). With thestepwise approach to management, treatment consisted of: no intervention, n = 10; pronepositioning, n = 26; nasopharyngeal airway, n = 4; low flow oxygen: n = 1; and surgery: tongue5lip-adhesion (TLA), n = 9; mandibular distraction osteogenesis (MDO), n = 9; andtracheostomy, n = 1.We reviewed 57 polysomnographies in 29 infants; many infants with very mildobstruction in the neonatal period and normal evolution did not require a polysomnography.However, of the 97 tests that could have been done according to the institution protocol, only59% were done. The tests done were useful to identify moderate to severe obstruction in38.6% of cases.When we compared oximetry to polysomnography, there was a strong correlationbetween DI ≥3% and MOAHI data (correlation coefficient: 0.721, p<0.001). All infants with aDI ≥3% <10 events/hour had no or mild obstructive apnea (MOAHI <5 events/hour, 100%negative predictive value). In patients with MOAHI >5 events/hour, all had a DI ≥3% >7events/hour (100% sensitivity). Finally, in infants who had serial oximetry in the first 6 monthsof life to follow evolution, significant obstruction at age 10-12 months (moderate or severeobstructive index) was associated with an elevated value of the DI ≥3% at age 5 to 6 months(>10 events/hour).CONCLUSIONIn our cohort of infants with Robin sequence, a cohort having the characteristics ofother larger cohorts, oximetry can identify the infants with no or mild obstructive apneathereby decreasing the demand for polysomnography. Oximetry could be also a solution toserial evaluation of infants in the first several months of life to avoid adverse outcome andorganize rapid intervention
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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 | 0.002 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.000 | 0.000 |
| Bibliometrics | 0.002 | 0.001 |
| Science and technology studies | 0.000 | 0.000 |
| Scholarly communication | 0.001 | 0.001 |
| Open science | 0.000 | 0.001 |
| 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".