| Literature DB >> 9151333 |
M J Bennett1, R L Boriack, D G Birch.
Abstract
We have studied the effects of polyunsaturated fatty acid (PUFA) supplementation in utero and throughout life in mnd mutant mice, a proposed model for juvenile neuronal ceroid lipofuscinosis (CLN-3). Unlike our earlier in-vitro studies in humans with CLN-3, and in-vitro studies in CLN-3 lymphoblasts, we saw no beneficial effects in electroretinographic, electron microscopic or clinical studies in the mnd mice. Electron microscopy of brain revealed a pattern which was not consistent with the characteristic ceroid patterns in CLN-3. Our data suggest that the mnd mouse is not responsive to PUFA supplementation and may not be an appropriate animal model for CLN-3.Entities:
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Year: 1997 PMID: 9151333 DOI: 10.1055/s-2007-973678
Source DB: PubMed Journal: Neuropediatrics ISSN: 0174-304X Impact factor: 1.947