The presence of a hernia sac in isolated congenital diaphragmatic hernia is associated with less disease severity: A retrospective cohort study

Levesque, Matthew; Derraugh, Gabrielle; Schantz, Daryl; Morris, Mélanie; Shawyer, Anna C.; Min, Suyin A. Lum; Keijzer, Richard

doi:10.1016/j.jpedsurg.2019.01.016

Cited by 12 publications

(11 citation statements)

References 12 publications

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“…This is in keeping with the findings of Levesque et al showing in their population of 71 patients no association between presence of a hernia sac and either mortality or oxygen dependence. 20 We found no difference(s) in sex ratios between CDH with sac and CDH without a sac, both showing 2:1 male predilection. Interestingly, the side of the defect in CDH patients with a sac in our series is significantly more symmetric with 41% right-sided defects compared with only 16% right-sided defects in CDH without a sac.…”

Section: Discussioncontrasting

confidence: 53%

“…It is notable that Levesque et al in one of the few series with no significant difference between CDH newborns with or without sac also reported excellent survival in the no sac cohort (95%). 20 Ours is not the first such report to highlight the difficulty in defining the survival rate for infants with CDH with challenges arising due to geographic and institutional variations in clinical practice. 21 This highlights the potential problems with external validity and thus the generalizability of findings particularly "effect size" from one institution or geographic location to another.…”

Section: Discussionmentioning

confidence: 93%

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Does the Presence of a Hernia Sac Improve Survival in Newborns with Congenital Diaphragmatic Hernia? A United Kingdom Single-Center Experience

2020

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Introduction A developing body of literature suggests that the presence of a hernia sac in fetuses with congenital diaphragmatic hernia (CDH) may indicate improved prognosis. By examining a large cohort of CDH newborns admitted to a single United Kingdom specialist center, we aimed to establish if presence of hernia sac is a robust predictor of improved survival. Materials and Methods All CDH patients admitted to a single center were recruited. Postneonatal presentations and Morgagni hernias were excluded. Demographics, defect type, laterality, survival, and hernia recurrence were recorded. Results In this study, 192 CDH newborns were managed from 1997 to 2017; 39 were excluded (10 Morgagni and 29 postneonatal); 22 (14%) neonates had a hernia sac. Survival in patients with a hernia sac was 21/22 (95%) versus 107/124 (86%) in cases without hernia sac (p = 0.2). There was no difference in hernia sac proportion by gender (male:female 15 vs. 13.2%, p = 0.8). Conclusion In contrast to studies showing a survival advantage, albeit with smaller patient numbers, we report a statistical nonsignificant benefit of hernia sac. Better survival outcomes at this specialist center with CDH patients without a hernia sac than reported in other published studies are likely responsible for the lack of statistical significance observed, despite a larger cohort. National and international CDH registries yielding “big data” may provide further answers on the utility of a CDH hernia sac as a new prognostic scoring tool.

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Section: Discussioncontrasting

confidence: 53%

Section: Discussionmentioning

confidence: 93%

Does the Presence of a Hernia Sac Improve Survival in Newborns with Congenital Diaphragmatic Hernia? A United Kingdom Single-Center Experience

2020

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“…This could give us new insights in the type of surgery, type of defect, and functional outcome on the requirement of more ventilation and oxygen. Two previous studies by Levesque et al (2019) and Oliver et al (2019) showed a lower oxygen dependency after 28 respectively 30 days for hernia sac patients [28,29]. However, all of these studies (including this study) are retrospective single center studies; therefore differences in diagnostics and treatment could give a bias (e.g., a difference in ECMO rate).…”

Section: Discussionmentioning

confidence: 63%

Congenital diaphragmatic eventration and hernia sac compared to CDH with true defects: a retrospective cohort study

et al. 2020

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Congenital diaphragmatic eventration (CDE) and congenital diaphragmatic hernia (CDH) with or without hernia sac are three different types of congenital diaphragmatic malformations, which this study evaluates. All surgically treated patients with CDE or Bochdalek type CDH between 2000 and 2016 were included in this retrospective analysis. Demographics, CDH-characteristics, treatment, and clinical outcome were evaluated. In total, 200 patients were included. Patients with an eventration or hernia sac had no significant differences and were compared as patients without a true defect to patients with a true defect. The 1-year survival of patients with a true defect was significantly lower than patients with no true defect (76% versus 97%, p = 0.001). CDH with no true defect had significantly better short-term outcomes than CDH with true defect requiring patch repair. However, at 30 days, they more often required oxygen supplementation (46% versus 26%, p = 0.03) and had a higher recurrence rate (8% versus 0%, p = 0.006) (three eventration and two hernia sac patients). Conclusion: Patients without a true defect seem to have a more similar clinical outcome than CDH patients with a true defect, with a better survival. However, the recurrence rate and duration of oxygen supplementation at 30 days are higher than CDH patients with a true defect. What is Known:• Congenital diaphragmatic hernia with or without hernia sac and congenital diaphragmatic eventration (incomplete muscularization) are often treated similarly. • Patients with hernia sac and eventration are thought to have a relatively good outcome, but exact numbers are not described. What is New: • Congenital diaphragmatic eventration and patients with hernia sac seem to have a more similar clinical outcome than Bochdalek type CDH with a true defect. • Patients without a true defect (eventration or hernia sac) have a high recurrence rate.

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“…Numerous studies have shown that low birthweight, prematurity, large defect size, right sided defects, liver herniation, absence of a hernia sac, and severe pulmonary hypertension have a negative impact on neonatal outcomes. 2,3,13,[20][21][22][23][24][25][26][27][28][29] For example, the presence of a hernia sac in CDH has been associated with decreased time spent on ventilation, 30 and the presence of severe pulmonary hypertension has been associated with mortality. 22,31 Despite the previously reported associations with poor neonatal outcomes, we found that only lower birthweights and lower gestational ages were associated with worse respiratory outcomes between 0 and 5 years of age.…”

Section: Discussionmentioning

confidence: 99%

Asthma Medication Use in Congenital Diaphragmatic Hernia Survivors: A Retrospective Population Level Data Analysis

et al. 2019

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Introduction The purpose of this study was to determine if congenital diaphragmatic hernia (CDH) survivors had worse long-term respiratory outcomes compared with age-matched controls, as measured by inhaled bronchodilator use, inhaled steroid use, and asthma-related physician visits. Materials and Methods We performed a retrospective case-control study of infants with isolated CDH from 1991 to 2013. The primary outcome measures were inhaled bronchodilator prescriptions, inhaled steroid prescriptions, and asthma-related physician visits between 0 and 5 years of age and between 5 and 10 years of age. Subgroup analysis compared the same outcomes for CDH patients grouped by: birth weight, gestational age, side of defect, defect size, liver herniation, hernia sac, and pulmonary hypertension. Results Fifty-six patients with CDH and 753 age-matched controls met the inclusion criteria for the 0 to 5 years of age analysis. Between 0 and 5 years of age, more CDH survivors were prescribed an inhaled bronchodilator (odds ratio [OR] = 2.47[1.38–4.48], p = 0.001) and inhaled steroid (OR = 2.03[1.07–3.74], p = 0.03), and had an asthma-related physician visit (OR = 1.92[1.00–3.56], p = 0.04). Thirty-eight cases and 491 controls met the inclusion criteria for the 5 to 10 years of age analysis. Between 5 and 10 years of age, CDH survivors were not more likely to be prescribed inhaled bronchodilators, inhaled steroids, or have an asthma-related physician visit. Among the CDH patients, we did not find a clinical characteristic associated with increased inhaled bronchodilator or steroid prescriptions at any age. Conclusion A history of CDH is associated with higher rates of inhaled bronchodilator prescriptions, inhaled steroid prescriptions, and asthma-related physician visits from 0 to 5 years of age compared with age-matched controls. However, this difference resolves by 5 to 10 years of age.

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The presence of a hernia sac in isolated congenital diaphragmatic hernia is associated with less disease severity: A retrospective cohort study

Cited by 12 publications

References 12 publications

Does the Presence of a Hernia Sac Improve Survival in Newborns with Congenital Diaphragmatic Hernia? A United Kingdom Single-Center Experience

Does the Presence of a Hernia Sac Improve Survival in Newborns with Congenital Diaphragmatic Hernia? A United Kingdom Single-Center Experience

Congenital diaphragmatic eventration and hernia sac compared to CDH with true defects: a retrospective cohort study

Asthma Medication Use in Congenital Diaphragmatic Hernia Survivors: A Retrospective Population Level Data Analysis

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