ArticleDevelopment (Cambridge, England)2013
Cftr controls lumen expansion and function of Kupffer's vesicle in zebrafish.
Article in Development (Cambridge, England), 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 75 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
75 citing papers in PubMed, 112 citations in OpenAlex.
- Biophysics of lumen morphogenesis.Development (Cambridge, England) · 2026Review
- Article
- Cystic fibrosis: insights from zebrafish models.Disease models & mechanisms · 2026Review
- Transcriptional divergence of the zebrafishbioRxiv : the preprint server for biology · 2026Article
- Specific mitotic events drive left-right organizer development.Development (Cambridge, England) · 2025Article
- The dynamics of tubulogenesis in development and disease.Development (Cambridge, England) · 2025Review
- Bioelectric stimulation controls tissue shape and size.Nature communications · 2024Article
- Fluid extraction from the left-right organizer uncovers mechanical properties needed for symmetry breaking.eLife · 2023Article
- Multicellular rosettes link mesenchymal-epithelial transition to radial intercalation in the mouse axial mesoderm.Developmental cell · 2023Article
- Rab8, Rab11, and Rab35 coordinate lumen and cilia formation during zebrafish left-right organizer development.PLoS genetics · 2023Article
- Polarized transport of membrane and secreted proteins during lumen morphogenesis.Seminars in cell & developmental biology · 2023Review
- Activation of a transient progenitor state in the epicardium is required for zebrafish heart regeneration.Nature communications · 2022Article
- Human epiblast lumenogenesis: From a cell aggregate to a lumenal cyst.Seminars in cell & developmental biology · 2022Review
- Hydrostatic pressure as a driver of cell and tissue morphogenesis.Seminars in cell & developmental biology · 2022Review
- PHLPP1 regulates CFTR activity and lumen expansion through AMPK.Development (Cambridge, England) · 2022Article
- Morphogenetic Roles of Hydrostatic Pressure in Animal Development.Annual review of cell and developmental biology · 2022Review
- Rab11 endosomes and Pericentrin coordinate centrosome movement during pre-abscission in vivo.Life science alliance · 2022Article
- Cystic Fibrosis and the Cells of the Airway Epithelium: What Are Ionocytes and What Do They Do?Annual review of pathology · 2022Review
- Understanding laterality disorders and the left-right organizer: Insights from zebrafish.Frontiers in cell and developmental biology · 2022Review
- Stress ball morphogenesis: How the lizard builds its lung.Science advances · 2021Article
15 more citing papers are in PubMed but not listed here.
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
3 authors at 3 institutions in 1 country.
Funding
Abstract
Regulated fluid secretion is crucial for the function of most organs. In vertebrates, the chloride channel cystic fibrosis transmembrane conductance regulator (CFTR) is a master regulator of fluid secretion. Although the biophysical properties of CFTR have been well characterized in vitro, little is known about its in vivo role during development. Here, we investigated the function of Cftr during zebrafish development by generating several cftr mutant alleles using TAL effector nucleases. We found that loss of cftr function leads to organ laterality defects. In zebrafish, left-right (LR) asymmetry requires cilia-driven fluid flow within the lumen of Kupffer's vesicle (KV). Using live imaging we found that KV morphogenesis is disrupted in cftr mutants. Loss of Cftr-mediated fluid secretion impairs KV lumen expansion leading to defects in organ laterality. Using bacterial artificial chromosome recombineering, we generated transgenic fish expressing functional Cftr fusion proteins with fluorescent tags under the control of the cftr promoter. The transgenes completely rescued the cftr mutant phenotype. Live imaging of these transgenic lines showed that Cftr is localized to the apical membrane of the epithelial cells in KV during lumen formation. Pharmacological stimulation of Cftr-dependent fluid secretion led to an expansion of the KV lumen. Conversely, inhibition of ion gradient formation impaired KV lumen inflation. Interestingly, cilia formation and motility in KV were not affected, suggesting that fluid secretion and flow are independently controlled in KV. These findings uncover a new role for cftr in KV morphogenesis and function during zebrafish development.
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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.