Does Ventilatory Time Retain Its Validity in Predicting Neurodevelopmental Outcome at Two Years of Age in High-Risk Congenital Diaphragmatic Hernia Survivors?

Abstract

To evaluate if in high-risk non-extracorporeal membrane oxygenation (ECMO)-treated congenital diaphragmatic hernia (CDH) survivors, ventilatory time (VT) is correlated to and can be used as clinical marker of neurodevelopmental delay at 2 years of age. Cohort study was conducted between 2008 and 2012. Mental, motor, and language development were assessed by the Bayley Scales of Infant and Toddler Development III. The correlation between VT and neurodevelopmental outcome (NDO) was analyzed using Pearson's test. Receiver operating characteristic (ROC) analysis was performed to determine the accuracy and best cutoff value of VT to predict the risk of neurodevelopmental delay. Statistical significance was set at < 0.05. A total of 49 patients form the subject of this study. VT during first admission was inversely correlated with cognitive ( = -0.4116; = 0.0033), motor ( = -0.4241; = 0.0024), and language development (= -0.3564; = 0.0119). Using ROC curve analysis, VT was a significant predictor for neurodevelopmental delay in the cognitive (area under the curve [AUC]: 0.864, sensitivity: 100; specificity: 66.67; < 0.0001) and motor (AUC: 0.902; sensitivity: 100; specificity: 73.17; < 0.0001) scales, but not in the language scale. The best cutoff value for both scales was 9 days. Within a population of high-risk non-ECMO-treated CDH survivors, VT appears to retain its validity as a clinical marker of adverse NDO in cognitive and motor domains.

Neurodevelopmental outcomes in children with isolated congenital diaphragmatic hernia: A systematic review and meta‐analysis

“…Finally, 90 studies were included in this review, reporting on 24,930 children with CDH (Table ). This included 83 case series 15,17−21,33‐110 , four cohort studies, 16,111‐113 and three randomized controlled trials 7,114,115 . Nine studies reported only on isolated CDH cases 7,21,52,76,85,91,104,105,109 and in four studies a differentiation was made between isolated and nonisolated cases 39,51,81,94 …”

“…Cognitive deficits were reported in 22 studies. 7,16,17,19,33,37,38,[40][41][42]47,63,74,75,80,83,88,94,109,[111][112][113] In total, 10% (7%-15%) of CDH children scored abnormal. The majority of these studies used BSID and if only BSID results were analyzed, 14% (8%-20%) of children scoring abnormally.…”

“…Multiple studies reported on motoric deficits in CDH children. 7,16,17,19,33,37,38,41,42,45,47,56,58,60,68,74,75,83,85,88,91,93,94,109,113 Numbers range from 0% to 47%. Pooling of the data demonstrated that 14% (10%-19%) of children scored abnormal on motoric tests.…”

“…Language performance was reported in nine studies, all used the BSID 16,17,19,38,47,74,88,109,113 or the ASQ. Abnormal scores were reported in 9% (4%-15%).…”

“…Cognitive deficits were reported in 22 studies 7,16,17,19,33,37,38,40‐42,47,63,74,75,80,83,88,94,109,111‐113 . In total, 10% (7%–15%) of CDH children scored abnormal.…”