A model of glucose-6-phosphate dehydrogenase deficiency in the zebrafish.

ABSTRACT: Glucose-6-phosphate dehydrogenase (G6PD) deficiency is the most common genetic defect and enzymopathy worldwide, affecting approximately 400 million people and causing acute hemolysis in persons exposed to prooxidant compounds such as menthol, naphthalene, antimalarial drugs, and fava beans. Mouse models have not been useful because of a lack of significant response to oxidative challenge. We turned to zebrafish (Danio rerio) embryos, which develop ex utero and are transparent, allowing visualization of hemolysis. We designed morpholinos to zebrafish g6pd that were effective in reducing gene expression as shown by Western blot and G6PD enzyme activity, resulting in a brisk hemolysis and pericardial edema secondary to anemia. Titration of the g6pd knockdown allowed us to generate embryos th

SUBMITTER: Patrinostro X 

PROVIDER: S-EPMC3856909 | biostudies-literature | 2013 Aug

REPOSITORIES: biostudies-literature