Fetoscopic tracheal occlusion for treatment of non‐isolated congenital diaphragmatic hernia

Seravalli, Viola; Jelin, Eric B.; Miller, Jena L.; Tekes, Aylin; Baschat, Ahmet

doi:10.1002/pd.5139

Cited by 14 publications

(6 citation statements)

References 17 publications

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“…17 The strategy to induce lung growth in CDH through the augmentation of transmural (intrapulmonary) pressures has already been applied experimentally in humans by fetoscopic tracheal occlusion (FETO), a procedure performed at our hospital and other selected fetal care centers worldwide. 4,63 During FETO-induced lung growth in experimental models, a marked proliferation of type I alveolar epithelial cells, potentially involving the FGF10-FGFR2b-Sprouty2 pathway, has been shown. 64,65 FETO does not appear to increase NKX2.1 expression in either normal or CDH fetal lungs in late gestational animal models.…”

Section: Discussionmentioning

confidence: 99%

Human induced pluripotent stem cell-derived lung organoids in an ex vivo model of the congenital diaphragmatic hernia fetal lung

Kunisaki

Jiang

Biancotti

et al. 2020

Stem Cells Translational Medicine

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Three-dimensional lung organoids (LOs) derived from pluripotent stem cells have the potential to enhance our understanding of disease mechanisms and to enable novel therapeutic approaches in neonates with pulmonary disorders. We established a reproducible ex vivo model of lung development using transgene-free human induced pluripotent stem cells generated from fetuses and infants with Bochdalek congenital diaphragmatic hernia (CDH), a polygenic disorder associated with fetal lung compression and pulmonary hypoplasia at birth. Molecular and cellular comparisons of CDH LOs revealed impaired generation of NKX2.1 + progenitors, type II alveolar epithelial cells, and PDGFRα + myofibroblasts. We then subjected these LOs to disease relevant mechanical cues through ex vivo compression and observed significant changes in genes associated with pulmonary progenitors, alveolar epithelial cells, and mesenchymal fibroblasts. Collectively, these data suggest both primary cellintrinsic and secondary mechanical causes of CDH lung hypoplasia and support the use of this stem cell-based approach for disease modeling in CDH.

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Section: Discussionmentioning

confidence: 99%

Human induced pluripotent stem cell-derived lung organoids in an ex vivo model of the congenital diaphragmatic hernia fetal lung

Kunisaki

Jiang

Biancotti

et al. 2020

Stem Cells Translational Medicine

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show abstract

“…In most cases, the earlier the prenatal diagnosis of CDH is, the more severe the disease is. The LHR measured by B-mode ultrasound at different stages is an important indicator of the severity of diaphragmatic hernia [15, 16]. After ventilator treatment, children with low partial oxygen pressure and / or high partial carbon dioxide pressure in blood that is difficult to correct often need manual ventilation.…”

Section: Discussionmentioning

confidence: 99%

A comparative study of thoracoscopic and open surgery of congenital diaphragmatic hernia in neonates

Qin

Ren

2019

J Cardiothorac Surg

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Background An increasing number of hospitals have carried out neonatal thoracoscopic assisted repair of congenital diaphragmatic hernia (CDH). Methods The 26 cases received thoracoscopic-assisted repair (observation group) and 44 cases open repair (control group). General anesthesia was performed with endotracheal intubation using a trachea cannula without cuff. The general preoperative data, intraoperative hemodynamic parameters, intraoperative surgical conditions, postoperative complications, postoperative recovery condition, postoperative survival rate and recurrence rate were recorded. Results The intraoperative mean arterial pressure and heart rate at each time point in observation group were more stable and effective than those in control group (all P < 0.001). The number of manual ventilation, SpO 2 < 90% and hypercapnia cases were significantly lower than those in control group (all P < 0.05). Intraoperative bleeding, incision length and operation duration were significantly lower in observation group compared with control group (all P < 0.001). No significant differences were seen between the two groups in postoperative complications including pulmonary infection, incision infection, pulmonary hypertension, hemorrhage, and scleredema (all P > 0.05). The duration of postoperative mechanical ventilation, antibiotic use and hospitalization in observation group was significantly shorter than those in control group (all P < 0.05). There was no significant difference in postoperative survival rate and recurrence rate between the two groups (both P > 0.05). Conclusion The intraoperative hemodynamic parameters of CDH repair under thoracoscopy were more stable, the duration of postoperative mechanical ventilation, antibiotic use and hospitalization were shortened, and the therapeutic effect was better.

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“…For RCDH, fetal therapy is offered in case of severe hypoplasia (o/e LHR < 45%) because they have a predicted survival rate of 17%. For patients who have a more complex presentation with additional findings, a more individualized approach can be taken, yet in the absence of proof of benefit, this is debatable[ 18 , 19 ].…”

Section: Methodsmentioning

confidence: 99%

Fetoscopic endoluminal tracheal occlusion and reestablishment of fetal airways for congenital diaphragmatic hernia

et al. 2018

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BackgroundCongenital diaphragmatic hernia (CDH) is a congenital anomaly with high mortality and morbidity mainly due to pulmonary hypoplasia and hypertension. Temporary fetal tracheal occlusion to promote prenatal lung growth may improve survival. Entrapment of lung fluid stretches the airways, leading to lung growth.MethodsFetal endoluminal tracheal occlusion (FETO) is performed by percutaneous sono-endoscopic insertion of a balloon developed for interventional radiology. Reversal of the occlusion to induce lung maturation can be performed by fetoscopy, transabdominal puncture, tracheoscopy, or by postnatal removal if all else fails.ResultsFETO and balloon removal have been shown safe in experienced hands. This paper deals with the technical aspects of balloon insertion and removal. While FETO is invasive, it has minimal maternal risks yet can cause preterm birth potentially offsetting its beneficial effects.ConclusionFor left-sided severe and moderate CDH, the procedure is considered investigational and is currently being evaluated in a global randomized clinical trial (https://www.totaltrial.eu/). The procedure can be clinically offered to fetuses with severe right-sided CDH.Electronic supplementary materialThe online version of this article (10.1186/s10397-018-1041-9) contains supplementary material, which is available to authorized users.

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Fetoscopic tracheal occlusion for treatment of non‐isolated congenital diaphragmatic hernia

Cited by 14 publications

References 17 publications

Human induced pluripotent stem cell-derived lung organoids in an ex vivo model of the congenital diaphragmatic hernia fetal lung

Human induced pluripotent stem cell-derived lung organoids in an ex vivo model of the congenital diaphragmatic hernia fetal lung

A comparative study of thoracoscopic and open surgery of congenital diaphragmatic hernia in neonates

Fetoscopic endoluminal tracheal occlusion and reestablishment of fetal airways for congenital diaphragmatic hernia

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