Development of mortality prediction models for infants with isolated, left‐sided congenital diaphragmatic hernia before and after birth

Yoneda, Kota; Amari, Shoichiro; Mikami, Masashi; Uchida, Keiichi; Yokoi, Akiko; Okawada, Manabu; Furukawa, Taiji; Toyoshima, Katsuaki; Inamura, Noboru; Okazaki, Tadaharu; Yamoto, Masaya; Masumoto, Kouji; Terui, Keita; Okuyama, Hiroomi; Hayakawa, Masahiro; Taguchi, Tomoaki; Usui, Noriaki; Isayama, Tetsuya

doi:10.1002/ppul.26172

Cited by 3 publications

(1 citation statement)

References 37 publications

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“…The most widely used variable to stratify lung hypoplasia is prenatal measurement o/eLHR combined with liver position [ 19 ]. As one of the independent predictors of CDH death, more recent studies from large CDH treatment centers have confirmed that o/e LHR had good predictive efficacy and stable expression during pregnancy [ 19 – 22 ]. Specifically, the mean AUC of o/eLHR < 25% for predicting mortality in neonates with left-sided CDH was 0.73, with a specificity of 0.95 and a relatively low sensitivity of 0.45 [ 4 ].…”

Section: Discussionmentioning

confidence: 99%

Development of a prediction nomogram for 1-month mortality in neonates with congenital diaphragmatic hernia

Feng,

Wei,

Wang

et al. 2024

BMC Surg

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Objectives Although many prognostic factors in neonates with congenital diaphragmatic hernia (CDH) have been described, no consensus thus far has been reached on which and how many factors are involved. The aim of this study is to analyze the association of multiple prenatal and postnatal factors with 1-month mortality of neonates with CDH and to construct a nomogram prediction model based on significant factors. Methods A retrospective analysis of neonates with CDH at our center from 2013 to 2022 was conducted. The primary outcome was 1-month mortality. All study variables were obtained either prenatally or on the first day of life. Risk for 1-month mortality of CDH was quantified by odds ratio (OR) with 95% confidence interval (CI) in multivariable logistic regression models. Results After graded multivariable adjustment, six factors were found to be independently and consistently associated with the significant risk of 1-month mortality in neonates with CDH, including gestational age of prenatal diagnosis (OR, 95% CI, P value: 0.845, 0.772 to 0.925, < 0.001), observed-to-expected lung-to-head ratio (0.907, 0.873 to 0.943, < 0.001), liver herniation (3.226, 1.361 to 7.648, 0.008), severity of pulmonary hypertension (6.170, 2.678 to 14.217, < 0.001), diameter of defect (1.560, 1.084 to 2.245, 0.017), and oxygen index (6.298, 3.383 to 11.724, < 0.001). Based on six significant factors identified, a nomogram model was constructed to predict the risk for 1-month mortality in neonates with CDH, and this model had decent prediction accuracy as reflected by the C-index of 94.42%. Conclusions Our findings provide evidence for the association of six preoperational and intraoperative factors with the risk of 1-month mortality in neonates with CDH, and this association was reinforced in a nomogram model.

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Section: Discussionmentioning

confidence: 99%

Development of a prediction nomogram for 1-month mortality in neonates with congenital diaphragmatic hernia

Feng,

Wei,

Wang

et al. 2024

BMC Surg

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Predicting neonatal mortality prior to discharge from hospital in prenatally diagnosed left congenital diaphragmatic hernia

Shinar,

Otvodenko,

Kajal

et al. 2024

Ultrasound in Obstet & Gyne

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ObjectivesTo evaluate the association of standardized prenatal imaging parameters and immediate neonatal variables with mortality prior to discharge in infants with isolated left congenital diaphragmatic hernia (LCDH), and to compare the performance of ultrasound‐ and magnetic resonance imaging (MRI)‐based severity grading for the prediction of neonatal mortality.MethodsThis was a retrospective study of infants with prenatally diagnosed isolated LCDH referred to a single tertiary center between 2008 and 2020. Fetuses with right or bilateral congenital diaphragmatic hernia, additional major structural anomaly or known genetic condition, as well as cases that underwent fetal intervention or declined postnatal intervention, were excluded. Ultrasound and MRI images were reviewed retrospectively. Univariable and multivariable analyses were performed, incorporating prenatal and immediate neonatal factors to analyze the association with neonatal mortality prior to discharge, and a prediction calculator was generated. The performance of ultrasound and that of MRI for the prediction of neonatal mortality were compared.ResultsOf 253 pregnancies with fetal CDH, 104 met the inclusion criteria, of whom 77 (74%) neonates survived to discharge. Seventy‐five fetuses underwent both prenatal ultrasound and MRI. On multivariable analysis, observed/expected (o/e) lung‐to‐head ratio and o/e total fetal lung volume were associated independently with neonatal death (adjusted odds ratio, 0.89 (95% CI, 0.83–0.95) and 0.90 (95% CI, 0.84–0.97), respectively), whereas liver position was not. There was no significant difference in predictive performance between using ultrasound and MRI together (area under the receiver‐operating‐characteristics curve (AUC), 0.85 (95% CI, 0.76–0.93)) compared with using ultrasound alone (AUC, 0.81 (95% CI, 0.72–0.90); P = 0.19). The addition of neonatal parameters (gestational age at birth and small‐for‐gestational age) did not improve model performance (AUC, 0.87 (95% CI, 0.80–0.95)) compared with the combined ultrasound and MRI model (P = 0.22). There was poor agreement between severity assessment on ultrasound and MRI (Cohen's κ, 0.19). Most discrepancies were seen among cases deemed to be non‐severe on ultrasound and severe on MRI, and outcomes were more consistent with MRI‐based prognostication.ConclusionsIn fetuses with prenatally diagnosed isolated LCDH, mortality prediction using standardized ultrasound and MRI measurements performed reasonably well. In cases classified as non‐severe on ultrasound, MRI is recommended, as it may provide more accurate prognostication and assist in the determination of candidacy for fetal intervention. © 2024 The Author(s). Ultrasound in Obstetrics & Gynecology published by John Wiley & Sons Ltd on behalf of International Society of Ultrasound in Obstetrics and Gynecology.

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Prenatal Predictors of Survival in Isolated Congenital Diaphragmatic Hernia

Tofte,

Aghajani,

Jawwad

et al. 2024

O&G Open

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OBJECTIVE: To evaluate prenatal fetal imaging findings associated with survival to hospital discharge, persistent pulmonary hypertension (PH), and need for extracorporeal membrane oxygenation (ECMO) in fetuses with isolated congenital diaphragmatic hernia (CDH) that are undergoing prenatal expectant management. DATA SOURCES: A systematic search was conducted in MEDLINE through PubMed, EMBASE, Web of Science, and The Cochrane Central, and ClinicalTrials.gov from 2000 up to July 2023. METHODS OF STUDY SELECTION: Studies that reported on prenatal imaging in fetuses with isolated CDH that were undergoing expectant management were included. Primary outcomes were survival to hospital discharge, persistent PH within 28 days of age, and need for ECMO. The quality of studies was assessed using the Newcastle-Ottawa Scale. Meta-analysis was performed when at least two studies reported on the same prenatal imaging evaluation. Subgroup analyses were performed according to the side (left or right) of CDH. TABULATION, INTEGRATION, AND RESULTS: A total of 161 full-text articles were assessed for eligibility, with 48 studies meeting the inclusion criteria: 45 (N=3,977) assessed survival, eight (N=994) assessed persistent PH, and 12 (N=2,085) assessed need for ECMO. The pooled proportion was 2,833 of 3,977 (71.2%, 95% CI, 69.8–72.6%) for survival, 565 of 2,085 (27.1%, 95% CI, 25.2–29.1%) for need for ECMO, and 531 of 994 (53.4%, 95% CI, 50.3–56.6%) for need for persistent PH. Prenatal imaging findings that were significantly associated with survival included: total fetal lung volume (mean difference [MD] 13.42, 95% CI, 11.22–15.62), observed-to-expected (O-E) total fetal lung volume less than 30% (odds ratio [OR] 0.09, 95% CI, 0.05–0.17), O-E total fetal lung volume (MD 14.73, 95% CI, 11.62–17.84, I 2 46%), liver/intrathoracic ratio (MD −9.59, 95% CI, −15.73 to −3.46), O-E lung/head ratio (MD 14.03, 95% CI, 12.69–15.36), O-E lung/head ratio less than 25% (OR 0.07, 95% CI, 0.04–0.13), mediastinal shift angle (MD −6.17, 95% CI, −7.70 to −4.64), stomach position in mid-chest (OR 0.14, 95% CI, 0.06–0.36), and intrathoracic liver (OR 0.23, 95% CI, 0.15–0.35). In subgroup analyses, findings for left-sided CDH remained significant in all the aforementioned findings. The only prenatal imaging finding that was significantly associated with persistent PH was intrathoracic liver (OR 1.96, 95% CI, 1.14–3.37), but this association was no longer significant in subgroup analyses. Prenatal imaging findings that were significantly associated with need for ECMO included: O-E total fetal lung volume (MD −10.08, 95% CI, −13.54 to −6.62), O-E lung/head ratio (MD −9.88, 95% CI, 14.44 to −5.33, I 2 30%), subgroup analysis to the left-sided CDH remained significant, percentage of predicted lung volume (MD −9.81, 95% CI, −13.56 to −6.06, I 2 34%), and intrathoracic liver (OR 2.70, 95% CI, 1.60–4.57, I 2 0%), but this association was no longer significant in left-sided CDH subgroup analysis. CONCLUSION: Several prenatal imaging findings, including lung measurements, intrathoracic liver, and stomach position, were predictive of neonatal survival. Lung measurement was predictive of need for ECMO, and intrathoracic liver was significantly associated with persistent PH and need for ECMO.

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Development of mortality prediction models for infants with isolated, left‐sided congenital diaphragmatic hernia before and after birth

Cited by 3 publications

References 37 publications

Development of a prediction nomogram for 1-month mortality in neonates with congenital diaphragmatic hernia

Development of a prediction nomogram for 1-month mortality in neonates with congenital diaphragmatic hernia

Predicting neonatal mortality prior to discharge from hospital in prenatally diagnosed left congenital diaphragmatic hernia

Prenatal Predictors of Survival in Isolated Congenital Diaphragmatic Hernia

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