ArticleDevelopment (Cambridge, England)2013
Gpr177 regulates pulmonary vasculature development.
Article in Development (Cambridge, England), 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 25 papers, 1 of them a synthesis that pooled it.
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Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
25 citing papers in PubMed, 1 synthesis or guideline pooled it, 39 citations in OpenAlex.
- Associations of variants In the hexokinase 1 and interleukin 18 receptor regions with oxyhemoglobin saturation during sleep.PLoS genetics · 2019Pooled it
- Reconstructing signaling histories of single cells via perturbation screens and transfer learning.Nature methods · 2026Article
- Fzd2 orchestrates canonical and non-canonical Wnt signaling to regulate lung development and fibrosis.Cell communication and signaling : CCS · 2025Article
- A spatiotemporal cell atlas of cardiopulmonary progenitor cell allocation during development.Cell reports · 2025Article
- Wnt signaling regulates ion channel expression to promote smooth muscle and cartilage formation in developing mouse trachea.American journal of physiology. Lung cellular and molecular physiology · 2023Article
- Wnt signaling regulates ion channel expression to promote smooth muscle and cartilage formation in developing mouse trachea.bioRxiv : the preprint server for biology · 2023Article
- Review
- The role of Evi/Wntless in exporting Wnt proteins.Development (Cambridge, England) · 2023Review
- Epithelial Wntless regulates postnatal alveologenesis.Development (Cambridge, England) · 2022Article
- Review
- Dual regulation of planar polarization by secreted Wnts and Vangl2 in the developing mouse cochlea.Development (Cambridge, England) · 2020Article
- Wntless regulates lipogenic gene expression in adipocytes and protects against diet-induced metabolic dysfunction.Molecular metabolism · 2020Article
- Bidirectional Wnt signaling between endoderm and mesoderm confers tracheal identity in mouse and human cells.Nature communications · 2020Article
- Ablation of serum response factor in hepatic stellate cells attenuates liver fibrosis.Journal of molecular medicine (Berlin, Germany) · 2019Article
- Gastrointestinal dysfunction in autism displayed by altered motility and achalasia inProceedings of the National Academy of Sciences of the United States of America · 2019Article
- Serum response factor (SRF) promotes ROS generation and hepatic stellate cell activation by epigenetically stimulating NCF1/2 transcription.Redox biology · 2019Article
- Wnt/Fgf crosstalk is required for the specification of basal cells in the mouse trachea.Development (Cambridge, England) · 2019Article
- Inhibition of Wntless/GPR177 suppresses gastric tumorigenesis.BMB reports · 2018Article
- Unique aspects of the developing lung circulation: structural development and regulation of vasomotor tone.Pulmonary circulation · 2016Review
- Mesodermal lineages in the developing respiratory system.Trends in developmental biology · 2016Article
Corrections and comments
- Erratum issued
Authors and funding
6 authors at 2 institutions in 2 countries.
Funding
Abstract
Establishment of the functional pulmonary vasculature requires intimate interaction between the epithelium and mesenchyme. Previous genetic studies have led to inconsistent conclusions about the contribution of epithelial Wnts to pulmonary vasculature development. This discrepancy is possibly due to the functional redundancy among different Wnts. Here, we use Shh-Cre to conditionally delete Gpr177 (the mouse ortholog of Drosophila Wntless, Wls), a chaperon protein important for the sorting and secretion of Wnt proteins. Deletion of epithelial Gpr177 reduces Wnt signaling activity in both the epithelium and mesenchyme, resulting in severe hemorrhage and abnormal vasculature, accompanied by branching defects and abnormal epithelial differentiation. We then used multiple mouse models to demonstrate that Wnt/β-catenin signaling is not only required for the proliferation and differentiation of mesenchyme, but also is important for the maintenance of smooth muscle cells through the regulation of the transcription factor Kruppel-like factor 2 (Klf2). Together, our studies define a novel mechanism by which epithelial Wnts regulate the normal development and maintenance of pulmonary vasculature. These findings provide insight into the pathobiology of congenital lung diseases, such as alveolar capillary dysplasia (ACD), that have abnormal alveolar development and dysmorphic pulmonary vasculature.
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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.