Article
Targeted disruption of a murine glucuronyl C5-epimerase gene results in heparan sulfate lacking L-iduronic acid and in neonatal lethality.
The Journal of biological chemistry - 1 Aug 2003
Li Jin-Ping, Gong Feng, Hagner-McWhirter Asa, Forsberg Erik, Abrink Magnus, Kisilevsky Robert, Zhang Xiao, Lindahl Ulf
Abstract excerpt
The glycosaminoglycan, heparan sulfate (HS), binds proteins to modulate signaling events in embryogenesis. All identified protein-binding HS epitopes contain l-iduronic acid (IdoA). We report that targeted disruption of the murine d-glucuronyl C5-epimerase gene results in a structurally altered HS lacking IdoA. The corresponding phenotype is lethal, with renal agenesis, lung defects, and skeletal malformations....
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