FIGURE

Fig. S8

ID

ZDB-FIG-170706-30

Publication

Pei et al., 2016 - Additive reductions in zebrafish PRPS1 activity result in a spectrum of deficiencies modeling several human PRPS1-associated diseases

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Fig. S8

prps1a:prps1b double mutants are sensitive to inhibitions of nucleotide synthesis and ATP production.

(A-C) Morphology of embryos treated with 1 μM of leflunomide. A total of 76 embryos generated from a pairwise incross of adult fish carrying prps1a heterozygotic and prps1b homozygotic mutations were used for leflunomide treatment from 6 hpf until 3 dpf. Phenotype classification was performed at 3 dpf, with 19 embryos displaying a morphology as shown in panel A, 38 embryos as in panel B, and another 19 embryos as in panel C. Genotyping for prps1a mutation was conducted for 19 embryos from each class. The 19 embryos as in panel A included 8 wildtype and 11 heterozygotes. The 19 embryos as in panel B included 4 wild-type, 8 heterozygotes and 7 homozygotes. The 19 embryos as in panel C included 1 wild-type, 7 heterozygotes and 11 homozygotes. (D-E) Morphology of embryos treated with 0.5 ng/ml of antimycin A. A total of 32 embryos obtained as above were used for antimycin A treatment from 24 hpf until 3 dpf. Phenotype classification was performed at 3 dpf, with 24 embryos displaying a morphology as shown in panel D, and 8 embryos as in panel E. Genotyping for prps1a mutation revealed 11/11 embryos as in panel D were wild-type or heterozygotes for prps1a, and 8/8 embryos as in panel E were homozygotes for prps1a.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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