FIGURE

Fig. 4

ID

ZDB-FIG-200414-4

Publication

Ward et al., 2019 - Pharmacological restoration of visual function in a zebrafish model of von-Hippel Lindau disease

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Fig. 4

Sunitinib malate significantly improves visual function and reduces VEGF expression in vhl−/− larvae. A) Gross morphology of sibling and vhl-/- larvae treated with increasing doses of sunitinib malate at 5 dpf. Treatment reduces yolk and cardiac oedema but does not result in swim bladder inflation. B) All concentrations of sunitinib tested increase OKR visual function at 5 dpf with 1 μM sunitinib malate highlighting a statistically significant improvement. n = 3, 12 technical replicates per experiment. C) Correlation between hyaloid vessel number and visual function in 5 dpf wildtype larvae treated with sunitinib malate. n = 3 with 10 technical replicates per experiment. Dotted line denotes vhl−/− visual function and number of primary hyaloid branches at 5 dpf. D) VMR Max ‘ON’ response is significantly increased dose dependently in larvae treated with 0.2 μM and 0.5 μM Sunitinib. n = 3, 12 technical replicates per treatment group per experiment. E) VMR chromatograms illustrating the average Max ON and Max OFF peaks after treatment. F) Expression of pro- and anti-angiogenic vhl−/− larval eyes following sunitinib treatment.

Expression Data

Genes:	kdrl sema3aa sema3ab sema3fa sema3fb vegfaa vegfab vegfc
Fish:	y1Tg vhl^{hu2117/hu2117}; y1Tg
Condition:	chemical treatment by environment: sunitinib
Anatomical Term:	eye
Stage:	Day 5

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	y1Tg vhl^{hu2117/hu2117}; y1Tg
Condition:	chemical treatment by environment: sunitinib
Observed In:	eye hyaloid vessel angiogenesis optomotor response
Stage:	Day 5

Phenotype Detail

Acknowledgments

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Reprinted from Developmental Biology, 457(2), Ward, R., Ali, Z., Slater, K., Reynolds, A.L., Jensen, L.D., Kennedy, B.N., Pharmacological restoration of visual function in a zebrafish model of von-Hippel Lindau disease, 226-234, Copyright (2019) with permission from Elsevier. Full text @ Dev. Biol.