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Fig. 2 Copper-deficient embryos display a pleiotropic phenotype
DIC image of notochord from a control embryo at 28 hpf (A, left) reveals normal development of the central canal (c), floor plate (f), notochord (n), and aorta (a), while a copper-deficient embryo of the same age treated with neocuproine (NeoC) (A, right) shows malformations including distorted floor plate (arrow), undulating notochord, additional tissue structures (arrowhead), and displaced aorta. Alcian blue staining of 96 hpf control (B, left) and copper-deficient (B, right) embryos reveals reduced cartilage formation with compression (arrowhead) and impaired jaw elongation. At 48 hpf, fli1:GFP copper-deficient embryos (C) exhibit disorganization of the intersegmental vasculature with microaneurysms (arrowheads). O-dianisidine staining at 72 hpf (D) reveals a marked decrease in hematopoiesis (arrowheads) in a copper-deficient embryo (top) compared to control (bottom). DIC image at 32 hpf reveals an abnormal midbrain-hindbrain boundary (arrowheads) in copper-deficient (E, right) versus control (E, left) embryos. Antibody staining (Znp-1) reveals loss of primary motor neurons (arrow) and associated axons (arrowhead) in copper-deficient (F, right) versus control (F,left) embryo at 28 hpf.
Reprinted from Cell Metabolism, 4(2), Mendelsohn, B.A., Yin, C., Johnson, S.L., Wilm, T.P., Solnica-Krezel, L., and Gitlin, J.D., Atp7a determines a hierarchy of copper metabolism essential for notochord development, 155-162, Copyright (2006) with permission from Elsevier. Full text @ Cell Metab.