The Cellular and Molecular Effects of Fetoscopic Endoluminal Tracheal Occlusion in Congenital Diaphragmatic Hernia

Short, Walker D; Gilley, Jamie; Hammond, J. D.; Belfort, Michael A.; Lee, Timothy C.; King, Alice; Espinoza, Jimmy; Joyeux, Luc; Lingappan, Krithika; Gleghorn, Jason P.; Keswani, Sundeep G.

doi:10.3389/fped.2022.925106

Cited by 10 publications

(1 citation statement)

References 111 publications

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“…FETO has been shown to promote lung growth and alveolarization, as well as changes in the molecular composition of the lung [15‒18]. In lamb models, those treated with FETO with or without balloon removal had lung growth with normalization of lung parenchymal architecture and type II cell density; however, surfactant content and composition remained abnormal [19].…”

Section: Discussionmentioning

confidence: 99%

The Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia Treated with Fetoscopic Endoluminal Tracheal Occlusion: Beyond the Balloon

Wild,

Rintoul,

Ades

et al. 2024

Fetal Diagn Ther

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Introduction: Randomized controlled trials found that fetoscopic endoluminal tracheal occlusion (FETO) resulted in increased fetal lung volume and improved survival for infants with isolated, severe left-sided congenital diaphragmatic hernia (CDH). The delivery room resuscitation of these infants is particularly unique and the specific delivery room events are largely unknown. The objective of this study was to compare the delivery room resuscitation of infants treated with FETO compared to standard of care and describe lessons learned. Methods: Retrospective single center cohort study of infants treated with FETO compared to infants who met FETO criteria during the same period, but who received standard of care. Results: FETO infants were more likely to be born prematurely with 8/12 infants born < 35 weeks gestational age compared to 3/35 SOC infants. There were 5 infants who required emergent balloon removal (2 ex utero intrapartum treatment and 3 tracheoscopic removal on placental bypass with delayed cord clamping) and 7 with prenatal balloon removal. Surfactant was administered in 6/12 FETO (50.0%) infants compared to 2/35 (5.7%) in the SOC group. Extracorporeal membrane oxygenation use was lower at 25% and survival higher at 91.7% compared to 60% and 71.4% in the SOC infants, respectively. Conclusion: The delivery room resuscitation of infants treated with FETO requires thoughtful preparation with an experienced multidisciplinary team. Given increased survival, FETO should be offered to infants with severe isolated left-sided CDH, but only in high volume centers with the experience and capability of removing the balloon, emergently if needed. The neonatal clinical team must be skilled in managing the unique postnatal physiology inherent to FETO where effective interdisciplinary teamwork is essential. Empiric and immediate surfactant administration should be considered in all FETO infants to lavage thick airway secretions, particularly those delivered < 48 hours after balloon removal.

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Section: Discussionmentioning

confidence: 99%

The Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia Treated with Fetoscopic Endoluminal Tracheal Occlusion: Beyond the Balloon

Wild,

Rintoul,

Ades

et al. 2024

Fetal Diagn Ther

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Lung Development in a Dish: Models to Interrogate the Cellular Niche and the Role of Mechanical Forces in Development

Chernokal,

Gonyea,

Gleghorn

2023

Advances in Experimental Medicine and Biology

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Over the past decade, emphasis has been placed on recapitulating in vitro the architecture and multicellular interactions found in organs in vivo [1, 2]. Whereas traditional reductionist approaches to in vitro models enable teasing apart the precise signaling pathways, cellular interactions, and response to biochemical and biophysical cues, model systems that incorporate higher complexity are needed to ask questions about physiology and morphogenesis at the tissue scale. Significant advancements have been made in establishing in vitro models of lung development to understand cell-fate specification, gene regulatory networks, sexual dimorphism, three-dimensional organization, and how mechanical forces interact to drive lung organogenesis [3–5]. In this chapter, we highlight recent advances in the rapid development of various lung organoids, organ-on-a-chip models, and whole lung ex vivo explant models currently used to dissect the roles of these cellular signals and mechanical cues in lung development and potential avenues for future investigation (Fig. 3.1).

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The effect of tracheal occlusion in congenital diaphragmatic hernia in the nitrofen rat lung explant model

et al. 2022

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Here, we establish a tracheal occlusion (TO) model with rat lung explants in nitrofen-induced pulmonary hypoplasia in congenital diaphragmatic hernia (CDH). Methods:We extracted lungs from rats on embryonic day 18. We mimicked TO in the lung explants by tying the trachea. We assessed lung weight, morphometry, and abundance of Ki-67, Active caspase-3, and Prosurfactant Protein C (proSP-C) with immuno uorescence. Results:Lung weight was higher in TO+ than TO-on day 1. Abundance of Ki-67 was higher in TO+ than TO-(0.15 vs. 0.32, p=0.009 for day 1, 0.07 vs. 0.17, p=0.004 for day 2, 0.07 vs. 0.12, p=0.044 for day 3), and Active caspase-3 was higher in TO+ than TO-on day 2 and day 3 (0.04 vs. 0.03 p=0.669 for day 1, 0.03 vs. 0.13 p<0.001 for day 2, 0.04 vs. 0.17 p=0.008 for day3). However, proSP-C protein abundance was lower in TO+ than TO-(67.9 vs. 59.1 p=0.033 for day 1, 73.5 vs. 51.6 p=0.038 for day 2, 83.1 vs. 56.4 p=0.009 for day 3). Conclusions:The TO model in lung explants mimics the outcomes of current surgical models of TO and further studies can reveal the cellular and molecular effects of TO in CDH lungs.

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The Cellular and Molecular Effects of Fetoscopic Endoluminal Tracheal Occlusion in Congenital Diaphragmatic Hernia

Cited by 10 publications

References 111 publications

The Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia Treated with Fetoscopic Endoluminal Tracheal Occlusion: Beyond the Balloon

The Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia Treated with Fetoscopic Endoluminal Tracheal Occlusion: Beyond the Balloon

Lung Development in a Dish: Models to Interrogate the Cellular Niche and the Role of Mechanical Forces in Development

The effect of tracheal occlusion in congenital diaphragmatic hernia in the nitrofen rat lung explant model

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