Document Detail


Abnormalities of developing white matter in lysosomal storage diseases.
MedLine Citation:
PMID:  10499433     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Inborn metabolic errors causing lysosomal storage have well-recognized effects on neuronal function and morphology. In some classically "neuronal" storage diseases, however, neuroradiologic observations of infants have suggested a delay in central nervous system myelination based on persistently "immature" signal intensities monitored over time. This review summarizes reported neuropathologic evaluations of central white matter in infantile and juvenile patients and in corresponding animal models with lysosomal storage disorders. The observed neuropathology is examined in light of published studies of the biochemistry and microscopic anatomy of normal myelinogenesis. Finally, arguments are advanced that at least part of the deficiency of white matter is attributable to direct effects of the metabolic state on oligodendrocyte maturation and function, in addition to secondary effects on neurons and their axons.
Authors:
R D Folkerth
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Publication Detail:
Type:  Journal Article; Review    
Journal Detail:
Title:  Journal of neuropathology and experimental neurology     Volume:  58     ISSN:  0022-3069     ISO Abbreviation:  J. Neuropathol. Exp. Neurol.     Publication Date:  1999 Sep 
Date Detail:
Created Date:  1999-10-07     Completed Date:  1999-10-07     Revised Date:  2005-11-16    
Medline Journal Info:
Nlm Unique ID:  2985192R     Medline TA:  J Neuropathol Exp Neurol     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  887-902     Citation Subset:  IM    
Affiliation:
Department of Pathology (Neuropathology), Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts 02115, USA.
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MeSH Terms
Descriptor/Qualifier:
Animals
Brain / growth & development*
Cell Aging / physiology
Disease Models, Animal
Humans
Lysosomal Storage Diseases / physiopathology*
Myelin Sheath / physiology
Oligodendroglia / physiology

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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