ArticleDevelopment (Cambridge, England)2021
Defective mesothelium and limited physical space are drivers of dysregulated lung development in a genetic model of congenital diaphragmatic hernia.
Article in Development (Cambridge, England), 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
What it found
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The trial behind it
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Who cites it
20 citing papers in PubMed, 30 citations in OpenAlex.
- Mapping Embryonic Mouse Lung Development Using Enhanced Spatial Transcriptomics.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Organ-Specific and Conserved Regulatory Logic Orchestrates Gene Expression in the Embryonic Mesothelium.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Back to the Future-A 50-Year Dive into Embryo Implantation Research: Cell Biological Paradox, Epithelial Cell Polarity, and EMT.Biomolecules · 2026Review
- Impact of Surgical Diaphragmatic Repair on Central Airway Shape in Neonatal Congenital Diaphragmatic Hernia.Pediatric pulmonology · 2026Article
- Democratizing Organ-On-Chip Technologies With a Modular, Reusable, and Perfusion-Ready Microphysiological System.Advanced healthcare materials · 2026Article
- Modular Parallel Plate Flow Chamber with Tunable Substrate Mechanics and Defined Shear Stress.bioRxiv : the preprint server for biology · 2025Article
- Confocal-Compatible Workflow for Sectioning, Staining, and Imaging Serial Vibratome Sections for 3D Anatomical Reconstruction of the Lymph Node.bioRxiv : the preprint server for biology · 2025Article
- Interplay of ECM organization, ROCK signaling, and cell polarity drives mesothelium formation and lung growth.Nature communications · 2025Article
- Therapeutic nanoparticle safety in pregnancy: Bridging knowledge gaps with environmental insights and a translational roadmap.Journal of controlled release : official journal of the Controlled Release Society · 2025Review
- The nitrofen/bisdiamine murine model of congenital diaphragmatic hernia has a pulmonary hypertension vascular phenotype consistent with human CDH.American journal of physiology. Lung cellular and molecular physiology · 2025Article
- Zonal patterning of extracellular matrix and stromal cell populations along a perfusable cellular microchannel.Lab on a chip · 2024Article
- Mesenchymal Vangl1 and Vangl2 facilitate airway elongation and widening independently of the planar cell polarity complex.Development (Cambridge, England) · 2024Article
- Zonal Patterning of Extracellular Matrix and Stromal Cell Populations Along a Perfusable Cellular Microchannel.bioRxiv : the preprint server for biology · 2024Article
- FGF2 promotes the expansion of parietal mesothelial progenitor pools and inhibits BMP4-mediated smooth muscle cell differentiation.Frontiers in cell and developmental biology · 2024Article
- Connecting clinical, environmental, and genetic factors point to an essential role for vitamin A signaling in the pathogenesis of congenital diaphragmatic hernia.American journal of physiology. Lung cellular and molecular physiology · 2023Review
- Sex-related external factors influence pulmonary vascular angiogenesis in a sex-dependent manner.American journal of physiology. Heart and circulatory physiology · 2023Article
- Congenital diaphragmatic hernia.Nature reviews. Disease primers · 2022Review
- Genetically Modified Mouse Models of Congenital Diaphragmatic Hernia: Opportunities and Limitations for Studying Altered Lung Development.Frontiers in pediatrics · 2022Review
- Cellular, molecular, and metabolic aspects of developing lungs in congenital diaphragmatic hernia.Frontiers in pediatrics · 2022Review
- Cellular Origin(s) of Congenital Diaphragmatic Hernia.Frontiers in pediatrics · 2021Review
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
Congenital diaphragmatic hernia (CDH) is a developmental disorder associated with diaphragm defects and lung hypoplasia. The etiology of CDH is complex and its clinical presentation is variable. We investigated the role of the pulmonary mesothelium in dysregulated lung growth noted in the Wt1 knockout mouse model of CDH. Loss of WT1 leads to intrafetal effusions, altered lung growth, and branching defects prior to normal closure of the diaphragm. We found significant differences in key genes; however, when Wt1 null lungs were cultured ex vivo, growth and branching were indistinguishable from wild-type littermates. Micro-CT imaging of embryos in situ within the uterus revealed a near absence of space in the dorsal chest cavity, but no difference in total chest cavity volume in Wt1 null embryos, indicating a redistribution of pleural space. The altered space and normal ex vivo growth suggest that physical constraints are contributing to the CDH lung phenotype observed in this mouse model. These studies emphasize the importance of examining the mesothelium and chest cavity as a whole, rather than focusing on single organs in isolation to understand early CDH etiology.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.