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Impact of hernia sac in congenital diaphragmatic hernia: Associations with morbidity and mortality
Krysta M Sutyak1, Kylie I Holden2, Charles Green3
1Department of Pediatric Surgery, McGovern Medical School, The University of Texas Health Science Center at Houston (UTHSC), Children's Memorial Hermann Hospital, Houston, TX, USA; Center for Surgical Trials and Evidence-Based Practice (CSTEP), UTHSC at Houston, Houston, TX, USA. Electronic address: https://twitter.com/ksutyak.
Insights
The presence of a hernia sac in congenital diaphragmatic hernia (CDH) does not impact survival rates. However, patients with a hernia sac experienced improved morbidity outcomes, including reduced need for extracorporeal life support (ECLS).
Area of Science:
- Pediatric Surgery
- Neonatal Care
- Congenital Abnormalities
Background:
- The prognostic significance of a hernia sac (HS+) in congenital diaphragmatic hernia (CDH) remains unclear, especially when considering defect size.
- Previous studies have not adequately assessed the impact of HS+ on CDH outcomes while controlling for defect characteristics.
Purpose of the Study:
- To investigate the association between the presence of a hernia sac (HS+) and patient outcomes in congenital diaphragmatic hernia (CDH).
- To determine if HS+ influences survival, morbidity, and resource utilization in CDH patients, accounting for defect size.
Main Methods:
- A multicenter retrospective analysis of 7,828 CDH patients from the CDH Study Group database (2007-2024).
- Data included demographics, defect characteristics, morbidity, and mortality.
- Statistical analyses involved multilevel regression and Bayesian analysis, with results validated on hold-out data.
Main Results:
- HS+ was observed in 20% of CDH patients, more common in right-sided defects and smaller defect sizes (A and B).
- HS+ was not associated with increased mortality (OR 1.19, Bayesian OR 1.07) or adverse events like pulmonary hypertension.
- HS+ correlated with reduced extracorporeal life support (ECLS) utilization, improved oxygen status at 30 days, and shorter mechanical ventilation and hospital stays.
Conclusions:
- The presence of a hernia sac (HS+) in CDH does not adversely affect survival rates.
- HS+ is associated with improved morbidity outcomes, suggesting a potentially less severe presentation or better response to treatment.
- Further research may explore the biological mechanisms underlying these improved morbidity outcomes in HS+ patients.
Introduction:
The impact of hernia sac (HS+) on outcomes in congenital diaphragmatic hernia (CDH) when accounting for defect size is unknown. In a multicenter analysis, considering defect size, we aim to delineate the association of HS+ and morbidity and mortality.
Methods:
A retrospective analysis of CDH Study Group (2007-2024) data was performed. Demographics, defect characteristics, morbidity, and mortality were abstracted. Multilevel regression and parallel Bayesian analysis were performed on 80 % of the data, tested on 20 % hold-out data, and reported in area under the curve (AUC).
Results:
7828 operative CDH patients were included. HS+ in 20 %; most common in A and B defects (24 %, 25 %), decreasing in C and D defects (17 %, 7 %); present in a greater proportion of right-sided defects (30 % vs 18 %, p < 0.001). On multilevel logistic regression, with random effects for center, controlling for birth weight, major cardiac abnormalities, chromosomal anomaly, utilization of ECLS, side of the defect, CDHSG A-D, HS+ was not associated with survival (OR 1.19, 95 % CI 0.92-1.53). On Bayesian analysis the OR of HS+ on survival was 1.07 (95 % credible interval 0.87-1.36). HS+ was not associated with pulmonary hypertension (OR 0.97, 95 % CI 0.82-1.14) or cardiac dysfunction at birth. HS+ was associated with a reduction in ECLS utilization (OR 0.60, 95 % CI 0.48-0.74), oxygen status at 30 days (OR 0.56, 95 % CI 0.44-0.70), length of mechanical ventilation (IRR 0.84, 95 % CI 0.79-0.89), and length of hospital stay (IRR 0.92, 95 % CI 0.88-0.96).
Conclusion:
HS+ is not associated with increased survival, with a neutral Bayesian probability of benefit. However, morbidity outcomes were improved in HS+ patients.
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