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Quantitative isolation of α1AT mutant Z protein polymers from human and mouse livers and the effect of heat

✍ Scribed by Jae-Koo An; Keith Blomenkamp; Douglas Lindblad; Jeffrey H. Teckman


Book ID
102240783
Publisher
John Wiley and Sons
Year
2005
Tongue
English
Weight
443 KB
Volume
41
Category
Article
ISSN
0270-9139

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✦ Synopsis


Alpha-1-antitrypsin (alpha1AT) deficiency in its most common form is caused by homozygosity for the alpha1AT mutant Z gene. This gene encodes a mutant Z secretory protein, primarily synthesized in the liver, that assumes an abnormal conformation and accumulates within hepatocytes causing liver cell injury. Studies have shown that mutant alpha1ATZ protein molecules form unique protein polymers. These Z protein polymers have been hypothesized to play a critical role in the pathophysiology of liver injury in this disease, although a lack of quantitative methods to isolate the polymers from whole liver has hampered further analysis. In this study, we demonstrate a quantitative alpha1ATZ polymer isolation technique from whole liver and show that the hepatocellular periodic acid-Schiff-positive globular inclusions that are the histopathological hallmark of this disease are composed almost entirely of the polymerized alpha1ATZ protein. Furthermore, we examine the previously proposed but untested hypothesis that induction of alpha1ATZ polymerization by the heat of physiological fever is part of the mechanism of hepatic alpha1ATZ protein accumulation. The results, however, show that fever-range temperature elevations have no detectable effect on steady-state levels of intrahepatic Z protein polymer in a model in vivo system. In conclusion, methods to separate insoluble protein aggregates from liver can be used for quantitative isolation of alpha1ATZ protein polymers, and the effect of heat from physiological fever may be different in vivo compared with in vitro systems.


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