Lipid and Myelin Abnormalities of Brain in the Crinkled Mouse

Theriault, Linda L.; Dungan, Donna D.; Simons, Sandra; Keen, Carl L.; Hurley, Lucille S.

doi:10.3181/00379727-155-39848

Cited by 12 publications

(6 citation statements)

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“…We found that sulfatide, CE and most other lipids are at normal levels, however, which suggests that the lipid composition of CNS myelin is normal. The likely explanations for the difference between our results and the earlier ones are that either optic nerve is specifically not affected or that some com partment in whole brain other than white matter has the elevated sulfatide and CE pre viously observed [47]. Our study of peri pheral nerve indicates that there are 10-20% elevations in the cerebrosides and the major phospholipids.…”

Section: Mutants With Minor Lipid Abnormalitiescontrasting

confidence: 54%

“…Morphological analysis of cr/cr showed vacuolation of white matter, gross malformation of some brains, and cytoar chitectonie anomalies in the cerebellum [47]. Analysis of the lipids in whole brain at P21 re vealed that sulfatide is increased by about 65% and that CE is abundant, while the other lipids (cerebroside, PE, PI + SM, PS, PC and CO) are at normal levels [47], By analyzing optic nerve, we expected to localize the elevations of sulfatide and CE to white matter and to detect any additional ab normalities among CNS myelin lipids. We found that sulfatide, CE and most other lipids are at normal levels, however, which suggests that the lipid composition of CNS myelin is normal.…”

Section: Mutants With Minor Lipid Abnormalitiesmentioning

confidence: 99%

Section: Mutants With Minor Lipid Abnormalitiesmentioning

confidence: 99%

“…Rather than a behavioral pheno type of a neuromuscular disease, the cr muta tion mainly affects hair growth [16], When it was found that coat development improved with copper supplementation, the question arose as to whether myelin might also be af fected by the mutation [47], since copper is important for the normal development of myelin. Morphological analysis of cr/cr showed vacuolation of white matter, gross malformation of some brains, and cytoar chitectonie anomalies in the cerebellum [47]. Analysis of the lipids in whole brain at P21 re vealed that sulfatide is increased by about 65% and that CE is abundant, while the other lipids (cerebroside, PE, PI + SM, PS, PC and CO) are at normal levels [47], By analyzing optic nerve, we expected to localize the elevations of sulfatide and CE to white matter and to detect any additional ab normalities among CNS myelin lipids.…”

Section: Mutants With Minor Lipid Abnormalitiesmentioning

confidence: 99%

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A Survey of Neurological Mutant Mice

Ganser

Kerner²,

Brown³

et al. 1988

Dev Neurosci

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The lipids of white matter and peripheral nerve from neurological mutant mice with possible myelin abnormalities were analyzed by thin-layer chromatography and quantitated by densitometry. Eight mutants had major abnormalities in the central nervous system (CNS) and/or peripheral nervous system (PNS) tissues examined (optic nerve, and trigeminal and sciatic nerves). In the optic nerve of ax^J/ax^J, there were increases of 20–30% in the levels of the major phospholipids; peripheral nerve was normal. In bc^3J/bc^3JCNS, the major phospholipids and cholesterol were increased by 25–40%; the PNS was normal. In myd/myd CNS, there were increases of about 20% in the levels of both forms of cerebrosides and in the major phospholipids; in the PNS the lipids were normal, ot/ot CNS had 20–40% reductions of all the glycolipids and minor alterations in some of the phospholipids and cholesterol; the PNS had 20% losses of both forms of cerebrosides. In the PNS of ji/ji, there were decreases of 10–40% among the glycolipids and of 15–25% in three of the major phospholipids; the CNS was virtually normal. In the PNS of dt^J/dt^J, vb/vb and wr/wr, almost all lipids were significantly decreased. The CNS of dt^J/dt^Jand vb/vb were normal; wr/wr had minor reductions of certain glycolipids and phospholipids. Six mutants had relatively minor lipid abnormalities in their myelinated tissues. In cr/cr PNS, there were elevated levels of the cerebrosides and major phospholipids; the CNS was virtually normal. In db/db CNS and PNS, there were reduced levels of the nonhydroxy forms of cerebroside and sulfatide. The major change in htr/htr was the elevation of all the glycolipids in the CNS. In the CNS of Lc/+, nonhydroxy cerebroside was reduced. In shm/shm PNS, nonhydroxy sulfatide was elevated and there were small decreases in some of the phospholipids, wl/wl CNS showed decreases among most of the glycolipids. Mutants homozygous for du, mto, spa and tg had virtually normal lipid levels in both the optic and peripheral nerves.

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Section: Mutants With Minor Lipid Abnormalitiescontrasting

confidence: 54%

Section: Mutants With Minor Lipid Abnormalitiesmentioning

confidence: 99%

Section: Mutants With Minor Lipid Abnormalitiesmentioning

confidence: 99%

Section: Mutants With Minor Lipid Abnormalitiesmentioning

confidence: 99%

See 2 more Smart Citations

A Survey of Neurological Mutant Mice

Ganser

Kerner²,

Brown³

et al. 1988

Dev Neurosci

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“…aneurisms of heart and aorta, underdeveloped lungs (which are symptoms of altered collagen and elastin biosynthesis (Hurley et al 1980, Carnes 1971, malformations of the skeleton and neural lesions caused, particularly, by insufficient myelinization of axons (Harris et al 1980). The frequency of such abnormalities could be considerably decreased if copper salts were added to the diet of pregnant and lactating females (Theriault et al 1977). The role of CP or other possible carriers of copper was not studied.…”

Section: Discussionmentioning

confidence: 99%

Embryotoxicity of silver ions is diminished by ceruloplasmin?further evidence for its role in the transport of copper

et al. 1995

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The effect of alimentary administration of silver salts upon embryogenesis in rats was studied. Feeding of female rats throughout the term on a regular diet supplemented with AgCl did not cause alterations of their physiological functions, despite the fact that enzymatically active copper-containing ceruloplasmin (CP) was eliminated from the blood plasma. However, developmental abnormalities of embryos, their prenatal death or the 100% mortality of the newborns in the first 24 h of life was seen. Copper content in placenta and fetal tissues was strongly diminished. Cu, Zn-superoxide dismutase (SOD) activity decreased in cytoplasm of embryonic cells along with a drop, though less pronounced, in the tissues of the pregnant females. Embryotoxicity of AgCl was seriously diminished by repetitive injections of native CP to the pregnant rats. Such treatment resulted in an increase of SOD activity in placenta and embryonic tissues. The mortality of the newborns also became less. It is suggested that the embryotoxic effect of AgCl is caused by its ability to interfere with copper metabolism, in particular by altering the copper-transporting function of CP.

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