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Trace Elements in Medicine
International scientific and practical peer-reviewed journal
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THE ROLE OF ENVIRONMENTAL FACTORS IN THE DEVELOPMENT OF THE WILSON'S DISEASE PHENOTYPE IN HETEROZYGOUS CARRIERS OF THE ATP7B GENE

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ABSTRACT. Wilson's disease (WD) is a rare inherited monogenic autosomal recessive disease. It is caused by mutations in the ATP7B gene encoding Cu-transport ATPase, which lead to impaired excretion of Cu from the body and its toxic accumulation in the liver, brain and other tissues. WD studies are mainly carried out with the participation of homozygous carriers of mutations in the ATP7B gene or on Atp7b-/- mice. However, the number of heterozygous carriers of this gene in some regions can reach 3% of the total population. Among them, individuals are often identified who manifest signs of WD and/or Parkinson's disease (PD), which can increase depending on various environmental and genetic factors. We studied the effect of a high-calorie diet (HCD) on the development of metabolic syndrome associated with WD in Atp7b+/- mice. HCD in Atp7b+/- mice does not lead to changes in Cu status indexes and lipid metabolism. In the liver of Atp7b+/- mice, structural disorders characteristic of WD are found, turning into fatty degeneration and fibrosis on the background of HCD. All Atp7b+/- mice treated with HCD showed activation of adipose tissue (AT) similar to brown AT and a decrease in blood glucose levels. The mRNA level of Cu-associated genes decreases in the liver of Atp7b+/- mice treated with HCD and increases in subcutaneous fat cells. The data obtained indicate the need for medical control of heterozygous WD carriers to prevent liver pathologies, metabolic disorders, as well as early diagnosis of PD, which can potentially develop in these patients.

KEYWORDS: copper metabolism, Wilson's ATPase, ATP7B, Wilson's disease, adipose tissue.