Psychosine, the cytotoxic sphingolipid that accumulates in globoid cell leukodystrophy, alters membrane architecture

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Aberrant production of tenascin-C in globoid cell leukodystrophy alters psychosine-induced microglial functions.

Globoid cell leukodystrophy (GLD), or Krabbe disease, is a rare and often fatal demyelinating disease caused by mutations in the galactocerebrosidase (galc) gene that result in accumulation of galactosylsphingosine (psychosine). We recently reported that the extracellular matrix (ECM) protease, matrix metalloproteinase-3, is elevated in GLD and that it regulates psychosine-induced microglial ac...

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[Globoid cell leukodystrophy].

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MMP-3 mediates psychosine-induced globoid cell formation: implications for leukodystrophy pathology.

Globoid cell leukodystrophy (GLD) or Krabbe disease, is a fatal demyelinating disease attributed to mutations in the galactocerebrosidase (GALC) gene. Loss of function mutations in GALC result in accumulation of the glycolipid intermediate, galactosylsphingosine (psychosine). Due to the cytotoxicity of psychosine, it has been hypothesized that accumulated psychosine underlie the pathophysiology...

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Globoid cell leukodystrophy (GLD) is a rapidly progress- ing pediatric neurodegenerative disease caused by missing or dysfunctional lysosomal enzyme galactosylceramidase

Journal of Lipid Research Volume 54, 2013 3303 Copyright © 2013 by the American Society for Biochemistry and Molecular Biology, Inc. Globoid cell leukodystrophy (GLD) is a rapidly progressing pediatric neurodegenerative disease caused by missing or dysfunctional lysosomal enzyme galactosylceramidase (GALC). Hallmarks of this disease include macrophage (globoid cell) infi ltration into the brain...

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Psychosine accumulates in membrane microdomains in the brain of krabbe patients, disrupting the raft architecture.

Lipid rafts (LRs) are membrane realms characterized by high concentrations of cholesterol and sphingolipids. Often, they are portrayed as scaffolds on which many different signaling molecules can assemble their cascades. The idea of rafts as scaffolds is garnering significant attention as the consequences of LR disruption have been shown to be manifest in multiple signaling pathways. In this st...

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ژورنال

عنوان ژورنال: Molecular Genetics and Metabolism

سال: 2013

ISSN: 1096-7192

DOI: 10.1016/j.ymgme.2012.11.108