In-utero and post-delivery supplementation of motor neuron degeneration mutant mice with polyunsaturated fatty acids does not alter the clinical or pathological course

Neuropediatrics. 1997 Feb;28(1):82-4. doi: 10.1055/s-2007-973678.

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.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Animals
  • Animals, Newborn
  • Brain / pathology
  • Disease Models, Animal
  • Fatty Acids, Unsaturated / administration & dosage*
  • Female
  • Male
  • Mice
  • Mice, Neurologic Mutants
  • Microscopy, Electron
  • Neuronal Ceroid-Lipofuscinoses / diet therapy*
  • Neuronal Ceroid-Lipofuscinoses / genetics
  • Neuronal Ceroid-Lipofuscinoses / pathology
  • Pregnancy

Substances

  • Fatty Acids, Unsaturated