Lack of endothelial diaphragms in fenestrae and caveolae of mutant Plvap-deficient mice
Abstract
Plasmalemmal vesicle-associated protein (PLVAP, PV-1) is specifically expressed in endothelial cells in which it localizes to diaphragms of fenestrae, caveolae, and transendothelial channels. To learn about its function, we generated mutant mice that lack PLVAP. In a C57BL/6N genetic background, homozygous Plvap-deficient embryos die before birth and suffer from subcutaneous edema, hemorrhages, and defects in the vascular wall of subcutaneous capillaries. In addition, hearts of Plvap(-/-) embryos show ventricular septal defects and thinner ventricular walls. In wild-type embryos, PLVAP and caveolae with a stomatal diaphragm are present in endothelial cells of subcutaneous capillaries and endocardium, while a diaphragm is missing in caveolae of Plvap(-/-) littermates. Plvap(-/-) mice in a mixed C57BL/6N/FVB-N genetic background are born and survive at the most for 4 weeks. Capillaries of exocrine and endocrine pancreas and of kidney peritubular interstitium were investigated in more detail as examples of fenestrated capillaries. In these vascular beds, Plvap(-/-) mice show a complete absence of diaphragms in fenestrae, caveolae, and transendothelial channels, findings which are associated with a substantial decrease in the number of endothelial fenestrae. The changes in the capillary phenotype correlate with a considerable retardation of postnatal growth and anemia. Plvap(-/-) mice provide an animal model to clarify the specific functional role of endothelial fenestrae and their contribution to passage of water and solutes in different organs.
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Plvap scored highly in Topic 3 from scRNA-seq of embryonic cells and was also identified in adult and aged snRNA-seq profiles.
“Moreover, Plvap, which marks diaphragms of fenestrated endothelial cells (Herrnberger et al., 2012), scored highly in Topic 3 from scRNA-seq of embryonic cells (Figure 6A), and was also identified in adult and aged snRNA-seq profiles (Figure S6C).”
From A cellular and spatial map of the choroid plexus across brain ventricles and ages (2021) · cites this paper as Herrnberger et al., 2012
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A cellular and spatial map of the choroid plexus across brain ventricles and ages
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- Authors
- Herrnberger, L., Seitz, R., Kuespert, S., Bösl, M. R., Fuchshofer, R., & Tamm, E. R.
- Affiliations
- University of Regensburg (DE)
- Max Planck Institute of Neurobiology (DE)
- Corresponding author
- Tamm, Ernst R.
- Venue
- Histochemistry and Cell Biology
- Published
- 2012-07-11
- Volume
- 138
- Issue
- 5
- Pages
- 709-724
- ISSN
- 0948-6143 (ISSN-L)
- 1432-119X
- OpenAlex
- W1966424086
- Cited by
- 76
- References
- 41
- Citations by year
- 2026: 4 · 2025: 6 · 2024: 2 · 2023: 10 · 2022: 10 · 2021: 6 · 2020: 5 · 2019: 8 · 2018: 6 · 2017: 3 · 2016: 5 · 2015: 4 · 2014: 2 · 2013: 2 · 2012: 3
- FWCI
- 1.67
- Type
- journal article
- Language
- en
- Open access
- closed
- Licence
- www.springer.com/tdm (tdm)
- Locations
- Histochemistry and Cell Biology (publishedVersion)
- PubMed (publishedVersion)
- MPG.PuRe (Max Planck Society) (submittedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Caveolin-1 and cellular processes
- Life Sciences › Neuroscience › Neurology › Barrier Structure and Function Studies
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Angiogenesis and VEGF in Cancer
- Keywords
- poliovirus type 1, endothelial fenestrae, caveolae, transendothelial channels, endothelial cells, ventricular septal defect, subcutaneous edema, postnatal growth retardation, anemia, fenestrated capillaries, genetic background effects
- MeSH terms
- Animals, Capillaries, Carrier Proteins, Endocardium, Endothelium, Vascular, Female, Homozygote, Kidney, Male, Membrane Proteins, Mice, Inbred C57BL, Mutation, Pancreas, Caveolae, Models, Animal, Mice
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