Junctional Adhesion Molecule (JAM)-C Deficient C57BL/6 Mice Develop a Severe Hydrocephalus
Open accessAbstract
The junctional adhesion molecule (JAM)-C is a widely expressed adhesion molecule regulating cell adhesion, cell polarity and inflammation. JAM-C expression and function in the central nervous system (CNS) has been poorly characterized to date. Here we show that JAM-C(-/-) mice backcrossed onto the C57BL/6 genetic background developed a severe hydrocephalus. An in depth immunohistochemical study revealed specific immunostaining for JAM-C in vascular endothelial cells in the CNS parenchyma, the meninges and in the choroid plexus of healthy C57BL/6 mice. Additional JAM-C immunostaining was detected on ependymal cells lining the ventricles and on choroid plexus epithelial cells. Despite the presence of hemorrhages in the brains of JAM-C(-/-) mice, our study demonstrates that development of the hydrocephalus was not due to a vascular function of JAM-C as endothelial re-expression of JAM-C failed to rescue the hydrocephalus phenotype of JAM-C(-/-) C57BL/6 mice. Evaluation of cerebrospinal fluid (CSF) circulation within the ventricular system of JAM-C(-/-) mice excluded occlusion of the cerebral aqueduct as the cause of hydrocephalus development but showed the acquisition of a block or reduction of CSF drainage from the lateral to the 3(rd) ventricle in JAM-C(-/-) C57BL/6 mice. Taken together, our study suggests that JAM-C(-/-) C57BL/6 mice model the important role for JAM-C in brain development and CSF homeostasis as recently observed in humans with a loss-of-function mutation in JAM-C.
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JAM-C-deficient mice develop hemorrhages and hydrocephalus.
“JAM-C-deficient mice develop hemorrhages and hydrocephalus (657).”
From Blood-Brain Barrier: From Physiology to Disease and Back (2019) · cites this paper as [657]
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Paracellular Channel in Human Disease
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Beal, R. W. (2018) · Queen Mary Research Online (Queen Mary University of London)
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α-Catenin phosphorylation is tension-sensitive and supports epithelial barrier functions via afadin
Quinn, J. M., Le, P. M., Weng, A., Flozak, A. S., Folmsbee, S. S., Arroyo-Colon, E., Aillon, R. P., Jain, S. I., Pokutta, S., Weis, W. I., et al. (2026) · The Journal of Cell Biology · doi:10.1083/jcb.202508188
- Authors
- Wyss, L., Schäfer, J., Liebner, S., Mittelbronn, M., Deutsch, U., Enzmann, G., Adams, R. H., Aurrand-Lions, M., Plate, K. H., Imhof, B. A., & Engelhardt, B.
- Affiliations
- University of Bern (CH)
- Goethe University Frankfurt (DE)
- University of Münster (DE)
- University of Geneva (CH)
- Corresponding author
- Engelhardt, Britta
- Venue
- PLoS ONE
- Publisher
- Public Library of Science (PLoS)
- Published
- 2012-09-18
- Volume
- 7
- Issue
- 9
- Pages
- e45619
- ISSN
- 1932-6203 (ISSN-L)
- PMC
- PMC3445510
- OpenAlex
- W2029717436
- Cited by
- 42
- References
- 50
- Citations by year
- 2026: 2 · 2025: 7 · 2024: 2 · 2023: 3 · 2022: 4 · 2021: 2 · 2020: 2 · 2019: 4 · 2018: 4 · 2017: 3 · 2015: 4 · 2014: 2 · 2013: 1 · 2012: 2
- FWCI
- 1.19
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Locations
- PLoS ONE (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- Archive ouverte UNIGE (University of Geneva) (publishedVersion, other-oa)
- BORIS (University Library Bern) (submittedVersion, cc-by)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
- Max Planck Institute for Plasma Physics (submittedVersion)
- Publication Server of Goethe University Frankfurt am Main (Goethe University Frankfurt) (submittedVersion, cc-by)
- Archive ouverte UNIGE (University of Geneva) (submittedVersion, cc-by)
- Open Access CRIS of the University of Bern
- Funders
- National Science Foundation
- European Commission (202213, 241861, 201024)
- Schweizerischer Nationalfonds zur Förderung der Wissenschaftlichen Forschung (118390, 241861)
- Topics
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Cerebrospinal fluid and hydrocephalus
- Life Sciences › Neuroscience › Neurology › Barrier Structure and Function Studies
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Ion Transport and Channel Regulation
- Keywords
- JAM-C, hydrocephalus, cerebrospinal fluid flow, choroid plexus, ependymal cells, meninges, cerebral aqueduct, cerebrospinal fluid drainage, brain development, endothelial cells
- MeSH terms
- Animals, Base Sequence, Fluorescent Antibody Technique, Hydrocephalus, Mice, Inbred C57BL, Mice, Transgenic, DNA Primers, Mice, Junctional Adhesion Molecule C
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