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A new glucose-6-phosphate dehydrogenase variant with congenital nonspherocytic hemolytic anemia (G6PD Genova). Biochemical characterization and mosaicism expression in the heterozygote.

Authors :
Gaetani GF
Galiano S
Melani C
Miglino M
Forni GL
Napoli G
Perrone L
Ferraris AM
Source :
Human genetics [Hum Genet] 1990 Mar; Vol. 84 (4), pp. 337-40.
Publication Year :
1990

Abstract

A new deficient variant of glucose-6-phosphate dehydrogenase (G6PD) causing severe congenital nonspherocytic hemolytic anemia (CNSHA) is described. The variant enzyme, characterized by slow electrophoretic mobility, extreme in vivo and in vitro lability, high Km for G6P and strongly acidic pH optimum, appears to be unique, and has been designated G6PD Genova. Investigation of an obligate heterozygote using various cytochemical, biochemical and recombinant-DNA techniques showed G6PD mosaicism in the erythrocytes and leukocytes. Therefore, the presence of a disadvantageous mutation at one Gd locus did not determine selection in favor of the normal allele in the heterozygote's hemopoietic cells.

Details

Language :
English
ISSN :
0340-6717
Volume :
84
Issue :
4
Database :
MEDLINE
Journal :
Human genetics
Publication Type :
Academic Journal
Accession number :
2307454
Full Text :
https://doi.org/10.1007/BF00196229