FIGURE SUMMARY
Title

A Post-Developmental Genetic Screen for Zebrafish Models of Inherited Liver Disease

Authors
Kim, S.H., Wu, S.Y., Baek, J.I., Choi, S.Y., Su, Y., Flynn, C.R., Gamse, J.T., Ess, K.C., Hardiman, G., Lipschutz, J.H., Abumrad, N.N., Rockey, D.C.
Source
Full text @ PLoS One

Zebrafish mutants exhibit post-developmental liver defects without developmental phenotypes.

(A) A wild type control at 7 dpf. Livers are outlined with yellow line. (B) Control siblings are on the top and homozygous mutants are on the bottom of each panel. (C) Magnified images of livers (outlined with yellow) in homozygous mutants. E = eye, L = liver, G = gut and SB = swim bladder. “mu”, stands for MUSC, followed by a number that designates the unique mutants identified in the screen. n>100 per each mutant.

Whole mount Oil Red O (ORO) staining in representative mutants.

A control, wild type example, at 8 dpf, is shown in top of left panel. A sample of 11 representative mutants with a dark-colored liver and one mutant with clear liver (7048mu103) were subjected to ORO staining. Clear colored livers in control and 7048mu103 are outlined with black. n = 9/9 per each mutant.

Three liver phenotypes were identified in the forward genetic screen.

In all panels, the normal sibling is on top and the mutant is on the bottom. On the left, in (A, D) is shown a representative example of a mutant with hepatomegaly alone (n = 9/9). In (B, E) is shown a representative mutant with steatosis alone (n = 9/9), and (C,F) show a mutant with both hepatomegaly and steatosis (n = 9/9). In (A-C), are depicted representative stereomicroscopic images and in (D-F), whole mount ORO stained specimens. The liver is outlined in yellow.

Histological phenotypes.

On the left panel, H & E (top), DAPI (middle) and oil red oil (ORO, bottom) stained zebrafish livers at 8 dpf are shown. Wild-type control (A,F,K), and mutants (B-E, G-J, L-O) are shown. (B) shows and example of microvesicles in hepatocytes, (C) depicts a liver with swollen hepatocytes, (D) shows a liver with accumulation of large vesicles, and prenecrotic hepatocytes (asterisk), while (E) shows hepatic lysis. The black arrow in (E) points to a nucleus with nuclear membrane and the red arrow points to a condensed nucleus without nuclear membrane. The yellow arrowheads in (J) indicate granulated nuclei. g = gut. Scale bar = 100 µm (A-E) and 50 µm (F-O). n = 9/9 per control and each mutant.

Hyperlipidemia in etfavu463, 7604cmu110 and 7703mu108 mutants with steatosis.

ORO staining in plasma of heart chamber in wild-type control (A), etfavu463 (B), 7604cmu110 (C) and 7703mu108 (D) mutants. A = Atrium, V = Ventricle. Scale bar = 50 µm.

Unillustrated author statements

Acknowledgments
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