A metabolomic comparison of mouse models of the Neuronal Ceroid Lipofuscinoses View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2011-04

AUTHORS

Reza M. Salek, Michael R. Pears, Jonathan D. Cooper, Hannah M. Mitchison, David A. Pearce, Russell J. Mortishire-Smith, Julian L. Griffin

ABSTRACT

The Neuronal Ceroid Lipofuscinoses (NCL) are a group of fatal inherited neurodegenerative diseases in humans distinguished by a common clinical pathology, characterized by the accumulation of storage body material in cells and gross brain atrophy. In this study, metabolic changes in three NCL mouse models were examined looking for pathways correlated with neurodegeneration. Two mouse models; motor neuron degeneration (mnd) mouse and a variant model of late infantile NCL, termed the neuronal ceroid lipofuscinosis (nclf) mouse were investigated experimentally. Both models exhibit a characteristic accumulation of autofluorescent lipopigment in neuronal and non neuronal cells. The NMR profiles derived from extracts of the cortex and cerebellum from mnd and nclf mice were distinguished according to disease/wildtype status. In particular, a perturbation in glutamine and glutamate metabolism, and a decrease in γ-amino butyric acid (GABA) in the cerebellum and cortices of mnd (adolescent mice) and nclf mice relative to wildtype at all ages were detected. Our results were compared to the Cln3 mouse model of NCL. The metabolism of mnd mice resembled older (6 month) Cln3 mice, where the disease is relatively advanced, while the metabolism of nclf mice was more akin to younger (1-2 months) Cln3 mice, where the disease is in its early stages of progression. Overall, our results allowed the identification of metabolic traits common to all NCL subtypes for the three animal models. More... »

PAGES

175-184

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s10858-011-9491-7

DOI

http://dx.doi.org/10.1007/s10858-011-9491-7

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1043048967

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/21461951


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Download the RDF metadata as:  json-ld nt turtle xml License info

HOW TO GET THIS DATA PROGRAMMATICALLY:

JSON-LD is a popular format for linked data which is fully compatible with JSON.

curl -H 'Accept: application/ld+json' 'https://scigraph.springernature.com/pub.10.1007/s10858-011-9491-7'

N-Triples is a line-based linked data format ideal for batch operations.

curl -H 'Accept: application/n-triples' 'https://scigraph.springernature.com/pub.10.1007/s10858-011-9491-7'

Turtle is a human-readable linked data format.

curl -H 'Accept: text/turtle' 'https://scigraph.springernature.com/pub.10.1007/s10858-011-9491-7'

RDF/XML is a standard XML format for linked data.

curl -H 'Accept: application/rdf+xml' 'https://scigraph.springernature.com/pub.10.1007/s10858-011-9491-7'


 

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291 schema:name Department of Pediatrics, Sanford School of Medicine of the University of South Dakota, 2301 East 60th Street North, 57104-0589, Sioux Falls, SD, USA
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294 schema:name Johnson & Johnson PR & D, Turnhoutsweg 30, 2340, Beerse, Belgium
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297 schema:name Department of Biochemistry and the Cambridge Systems Biology Centre, University of Cambridge, 80 Tennis Court Road, Cambridge, CB2 1GA, UK
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301 schema:name Department of Paediatrics and Child Health, Royal Free and University College Medical School, London, UK
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