ZFIN ID: ZDB-FIG-191230-1509
Schenk et al., 2019 - Characterizing renal involvement in Hermansky-Pudlak Syndrome in a zebrafish model. Scientific Reports   9:17718 Full text @ Sci. Rep.
ADDITIONAL FIGURES
PHENOTYPE:
Fish:
Knockdown Reagents:
Observed In:
Stage: Day 5

Fig. 5

HPS1KD ATG, HPS3KD ATG, HPS4KD ATG and HPS5KD ATG induce varying degrees of glomerular damage including podocyte effacement and deposition of intracellular debris (zebra bodies). Transmission electron microscopy imaging was used to detect ultrastructural changes in glomeruli after HPS protein knockdown induced by MO injection in Tg(l-fabp:eGFP-DBP) transgene zebrafish embryos at a one to four cell stage. Collection of larvae for imaging was done at 120 hpf. (A) HPS1KD ATG causes loss of glomerular endothelial fenestration and podocyte effacement. HPS3KD ATG induces mild podocyte effacement. HPS4KD ATG causes podocyte effacement without endothelial injury, while HPS5KD ATG leads to reduced endothelial fenestration and podocyte effacement. Zebrafish injected with a control morpholino (CTRL-MO) show no sign of glomerular damage. Scale bar = 2 µm. (B) Induction of HPS protein knockdown of HPS1, -3, -4 and -5 leads to the deposition of intracellular debris primarily in podocytes. Scale bar = 500 nm.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
lri500Tg + MO1-hps1 standard conditions Day 5 pronephric glomerulus damaged, abnormal
Day 5 pronephric glomerulus glomerular endothelium fenestra decreased amount, abnormal
Day 5 pronephric podocyte composition, abnormal
Day 5 pronephric podocyte has extra parts of type pronephric podocyte inclusion body, abnormal
Day 5 pronephric podocyte podocyte foot morphology, abnormal
lri500Tg + MO1-hps4 standard conditions Day 5 pronephric glomerulus damaged, abnormal
Day 5 pronephric podocyte composition, abnormal
Day 5 pronephric podocyte has extra parts of type pronephric podocyte inclusion body, abnormal
Day 5 pronephric podocyte podocyte foot morphology, abnormal
lri500Tg + MO2-hps3 standard conditions Day 5 pronephric glomerulus damaged, abnormal
Day 5 pronephric podocyte composition, abnormal
Day 5 pronephric podocyte has extra parts of type pronephric podocyte inclusion body, abnormal
Day 5 pronephric podocyte podocyte foot morphology, abnormal
lri500Tg + MO2-hps5 standard conditions Day 5 pronephric glomerulus damaged, abnormal
Day 5 pronephric glomerulus glomerular endothelium fenestra decreased amount, abnormal
Day 5 pronephric podocyte composition, abnormal
Day 5 pronephric podocyte has extra parts of type pronephric podocyte inclusion body, abnormal
Day 5 pronephric podocyte podocyte foot morphology, abnormal
Acknowledgments:
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ Sci. Rep.