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          Institute: MPI für molekulare Biomedizin     Collection: Yearbook 2013     Display Documents



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ID: 648373.0, MPI für molekulare Biomedizin / Yearbook 2013
Junctional adhesion molecule (JAM)-C deficient C57BL/6 mice develop a severe hydrocephalus
Authors:Wyss, L.; Schafer, J.; Liebner, S.; Mittelbronn, M.; Deutsch, U.; Enzmann, G.; Adams, R. H.; Aurrand-Lions, M.; Plate, K. H.; Imhof, B. A.; Engelhardt, B.
Date of Publication (YYYY-MM-DD):2012
Title of Journal:PLoS ONE
Volume:7
Issue / Number:9
Start Page:e45619
Review Status:Internal review
Audience:Not Specified
Abstract / Description: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.
Free Keywords:Animals; Base Sequence; DNA Primers; Fluorescent Antibody Technique; Hydrocephalus/*genetics; Junctional Adhesion Molecule C/genetics/*physiology; Mice; Mice, Inbred C57BL; Mice, Transgenic
External Publication Status:published
Document Type:Article
Communicated by:keuker
Affiliations:MPI für molekulare Biomedizin
External Affiliations:%G eng
Identifiers:ISSN:1932-6203 (Electronic) 1932-6203 (Linking) %R 10.1... [ID No:1]
URL:http://www.ncbi.nlm.nih.gov/pubmed/23029139 [ID No:2]
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