Imagiological methods for prediction of fetal pulmonary hypoplasia: a systematic review

Gonçalves, Ana N.; Correia‐Pinto, Jorge; Nogueira‐Silva, Cristina

doi:10.1080/14767058.2019.1636029

Cited by 7 publications

(5 citation statements)

References 48 publications

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“…Neonatal respiratory failure is common in moderate-to-severe cases of congenital diaphragmatic hernia (CDH). CDH is defined by a diaphragmatic defect that allows the herniation of abdominal organs into the thorax and consequently lung hypoplasia, affecting 1 in 3000 live-births [ 15 , 16 ]. These hypoplastic lungs, strictly associated with high rates of mortality and morbidity, demonstrate a reduced number of terminal buds at early developmental stages (embryonic/pseudoglandular); reduced surface areas; and a decrease in distal branching and alveoli (saccular/alveolar) that impaired the efficient gas exchanges [ 17 , 18 ].…”

Section: Introductionmentioning

confidence: 99%

ROBO2 signaling in lung development regulates SOX2/SOX9 balance, branching morphogenesis and is dysregulated in nitrofen-induced congenital diaphragmatic hernia

2020

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Background Characterized by abnormal lung growth or maturation, congenital diaphragmatic hernia (CDH) affects 1:3000 live births. Cellular studies report proximal (SOX2+) and distal (SOX9+) progenitor cells as key modulators of branching morphogenesis and epithelial differentiation, whereas transcriptome studies demonstrate ROBO/SLIT as potential therapeutic targets for diaphragm defect repair in CDH. In this study, we tested the hypothesis that (a) experimental-CDH could changes the expression profile of ROBO1, ROBO2, SOX2 and SOX9; and (b) ROBO1 or ROBO2 receptors are regulators of branching morphogenesis and SOX2/SOX9 balance. Methods The expression profile for receptors and epithelial progenitor markers were assessed by Western blot and immunohistochemistry in a nitrofen-induced CDH rat model. Immunohistochemistry signals by pulmonary structure were also quantified from embryonic-to-saccular stages in normal and hypoplastic lungs. Ex vivo lung explant cultures were harvested at E13.5, cultures during 4 days and treated with increasing doses of recombinant rat ROBO1 or human ROBO2 Fc Chimera proteins for ROBO1 and ROBO2 inhibition, respectively. The lung explants were analyzed morphometrically and ROBO1, ROBO2, SOX2, SOX9, BMP4, and β-Catenin were quantified by Western blot. Results Experimental-CDH induces distinct expression profiles by pulmonary structure and developmental stage for both receptors (ROBO1 and ROBO2) and epithelial progenitor markers (SOX2 and SOX9) that provide evidence of the impairment of proximodistal patterning in experimental-CDH. Ex vivo functional studies showed unchanged branching morphogenesis after ROBO1 inhibition; increased fetal lung growth after ROBO2 inhibition in a mechanism-dependent on SOX2 depletion and overexpression of SOX9, non-phospho β-Catenin, and BMP4. Conclusions These studies provided evidence of receptors and epithelial progenitor cells which are severely affected by CDH-induction from embryonic-to-saccular stages and established the ROBO2 inhibition as promoter of branching morphogenesis through SOX2/SOX9 balance.

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Section: Introductionmentioning

confidence: 99%

ROBO2 signaling in lung development regulates SOX2/SOX9 balance, branching morphogenesis and is dysregulated in nitrofen-induced congenital diaphragmatic hernia

2020

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“…Comparing them will offer a better understanding of the true severity, even though the best approach is to calculate the total lung volume. As shown in other specialized literature, we can still benefit from 2D analysis [ 22 ].…”

Section: Discussionmentioning

confidence: 99%

Thoracic Biometry in Patients with Congenital Diaphragmatic Hernia, a Magnetic Resonance Imaging Study

Neștianu,

Popescu,

Alexandru

et al. 2024

Diagnostics

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This is a retrospective study investigating biometric measurements using magnetic resonance imaging (MRI) examinations in congenital diaphragmatic hernia (CDH). CDH is one of the more common causes of pulmonary hypoplasia, with grave consequences for the fetus. Inclusion criteria were patients diagnosed with CDH as the only observed anomaly, who underwent MRI examination after the second-trimester morphology ultrasound. The patients came from three university hospitals in Bucharest, Romania. In total, 19 patients were included in the study after applying exclusion criteria. Comparing the observed values of the thoracic transverse diameter, the thoracic anterior–posterior diameter, the thoracic circumference, the thoracic area, and the thoracic volume with values from the literature, we observed a predictive alteration of these parameters, with most showing Gaussian distribution. We observed statistical significance for most of our correlations, except between the observed and expected thoracic anterior–posterior diameters and the observed and expected thoracic volume values. This is very helpful when complex studies that can calculate the pulmonary volume cannot be obtained, as in the case of movement artifacts, and allows the clinicians to better assess the severity of the disease. MRI follow-up in CDH cases is a necessity, as it offers the most accurate thoracic biometry.

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“…The study by Büsing et al [ 16 ] demonstrated the validity and reliability of lung volume measurements by MRI independent of the selected analytical sequences in 40 fetuses with CDH. Other studies have shown the superiority of MRI as a predictor of survival compared with ultrasound [ 3 , 17 , 18 ]. Abbasi et al [ 19 ] evaluated the measurement of the lung-to-head ratio (LHR) in 26 centers in the US and found variability in the ultrasound method; they concluded that this measurement needs to be standardized.…”

Section: Discussionmentioning

confidence: 99%

Fetal Lung Volume Appears to Predict Respiratory Morbidity in Congenital Diaphragmatic Hernia

Cerbelle

Duc

Lejeune

et al. 2023

JCM

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Congenital diaphragmatic hernia (CDH) is associated with pulmonary hypoplasia and respiratory morbidity. To assess whether respiratory morbidity during the first 2 years of life in infants with left-sided CDH is associated with fetal lung volume (FLV) evaluated by the observed-to-expected FLV ratio (o/e FLV) on antenatal magnetic resonance imaging (MRI). In this retrospective study, o/e FLV measures were collected. Respiratory morbidity in the first 2 years of life was studied according to two endpoints: treatment with inhaled corticosteroids for >3 consecutive months and hospitalization for any acute respiratory disease. The primary outcome was a favorable progression defined by the absence of either endpoint. Forty-seven patients were included. The median o/e FLV was 39% (interquartile range, 33–49). Sixteen (34%) infants were treated with inhaled corticosteroids and 13 (28%) were hospitalized. The most efficient threshold for a favorable outcome was an o/e FLV ≥ 44% with a sensitivity of 57%, specificity of 79%, negative predictive value of 56%, and positive predictive value of 80%. An o/e FLV ≥ 44% was associated with a favorable outcome in 80% of cases. These data suggest that lung volume measurement on fetal MRI may help to identify children at lower respiratory risk and improve information during pregnancy, patient characterization, decisions about treatment strategy and research, and personalized follow-up.

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Imagiological methods for prediction of fetal pulmonary hypoplasia: a systematic review

Cited by 7 publications

References 48 publications

ROBO2 signaling in lung development regulates SOX2/SOX9 balance, branching morphogenesis and is dysregulated in nitrofen-induced congenital diaphragmatic hernia

ROBO2 signaling in lung development regulates SOX2/SOX9 balance, branching morphogenesis and is dysregulated in nitrofen-induced congenital diaphragmatic hernia

Thoracic Biometry in Patients with Congenital Diaphragmatic Hernia, a Magnetic Resonance Imaging Study

Fetal Lung Volume Appears to Predict Respiratory Morbidity in Congenital Diaphragmatic Hernia

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