[Mice with mottled mutation–a model for defective copper metabolism in humans]

SHORT COMMUNICATION

[Mice with mottled mutation–a model for defective copper metabolism in humans]

M Lenartowicz 1

1. Zakład Genetyki, Ewolucjonizmu Instytutu Zoologii Uniwersytetu Jagiellońskiego,Krakowie.

Published:

GICID: 01.3001.0000.3190

Available language versions: en pl

Issue: Postepy Hig Med Dosw 1998; 52 (5)

Abstract

The group of X-linked mottled (Atp7aMo) mutations in mice is described. A normal gene encodes a copper-binding P-type ATPase. Mutant animals have the disturbance in copper metabolism, hemizygous males (Mo/y) die between 14-18 days of life, heterozygous females (Mo/+) are normal and fertile. This kind of copper metabolic defect is observed also in other animal and in human. In human Menkes disease caused by X-linked Atp7a mutant gene leads to death in early childhood. Because of is 89% of homology between Atp7aMo gene and Atp7a locus in human, mottled mutations are an excellent model for Menkes disease.

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