CNS myelin and sertoli cell tight junction strands are absent in Osp/claudin-11 null mice

Oligodendrocyte-specific protein (OSP)/claudin-11 is a recently identified transmembrane protein found in CNS myelin and testis with unknown function. Herein we demonstrate that Osp null mice exhibit both neurological and reproductive deficits: CNS nerve conduction is slowed, hindlimb weakness is co...

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Bibliographic Details
Published in:Cell 1999-12, Vol.99 (6), p.649-659
Main Authors: Gow, A, Southwood, C M, Li, J S, Pariali, M, Riordan, G P, Brodie, S E, Danias, J, Bronstein, J M, Kachar, B, Lazzarini, R A
Format: Article
Language:English
Subjects:
Animals
Brain - cytology
Brain - metabolism
claudin 11
Claudins
Freeze Fracturing
Gene Expression Regulation, Developmental
Haplorhini
Hindlimb - growth & development
Infertility, Male - metabolism
Infertility, Male - pathology
Male
Membrane Proteins - genetics
Membrane Proteins - metabolism
Mice
Mice, Knockout
Microscopy, Confocal
Microscopy, Electron
Myelin Sheath - metabolism
Nerve Tissue Proteins - genetics
Nerve Tissue Proteins - metabolism
Oligodendrocyte-specific protein
Oligodendroglia - cytology
Oligodendroglia - metabolism
Osp gene
Rats
Sertoli Cells - metabolism
Sertoli Cells - ultrastructure
Testis - pathology
Tight Junctions - metabolism
Tight Junctions - ultrastructure
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Summary:Oligodendrocyte-specific protein (OSP)/claudin-11 is a recently identified transmembrane protein found in CNS myelin and testis with unknown function. Herein we demonstrate that Osp null mice exhibit both neurological and reproductive deficits: CNS nerve conduction is slowed, hindlimb weakness is conspicuous, and males are sterile. Freeze fracture reveals that tight junction intramembranous strands are absent in CNS myelin and between Sertoli cells of mutant mice. Our results demonstrate that OSP is the mediator of parallel-array tight junction strands and distinguishes this protein from other intrinsic membrane proteins in tight junctions. These novel results provide direct evidence of the pivotal role of the claudin family in generating the paracellular physical barrier of tight junctions necessary for spermatogenesis and normal CNS function.
ISSN:0092-8674
DOI:10.1016/s0092-8674(00)81553-6