ZFIN Image: Schaffer et al., 2014, Fig. S3

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Figure Caption

Fig. S3

clp1 Mutant Zebrafish Showed Reduced Lifespan, Motor Neuron Fragmentation, and p53-Mediated Neural Loss, Related to Figure 3

(A) In situ hybridization showed clp1 expressed in developing brain and motor neurons of clp1^+/+ zebrafish at 72 hours postfertilization (hpf) but not clp1^R44X/R44X.

(B) Lifespan of clp1^L35R/L35R mutant zebrafish is reduced to 4.5 days compared to heterozygous or WT clutchmates.

(C) Sv2 immunostaining showed motor neuron fragmentation in clp1^R44X/R44X zebrafish compared to control clutchmates at 72 hpf, concordant with neural degeneration.

(D) In situ hybridization at 72 hpf for otx2 in clp1^+/+ and clp1^R44R/R44X zebrafish injected with tp53 antisense morpholino, or uninjected, showed tp53 knockdown restores otx2 expression in mutant zebrafish to near WT levels. Black scale bar, 100 µm, white scale bar, 50 µm.

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Acknowledgments

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Reprinted from Cell, 157, Schaffer, A.E., Eggens, V.R., Caglayan, A.O., Reuter, M.S., Scott, E., Coufal, N.G., Silhavy, J.L., Xue, Y., Kayserili, H., Yasuno, K., Rosti, R.O., Abdellateef, M., Caglar, C., Kasher, P.R., Cazemier, J.L., Weterman, M.A., Cantagrel, V., Cai, N., Zweier, C., Altunoglu, U., Satkin, N.B., Aktar, F., Tuysuz, B., Yalcinkaya, C., Caksen, H., Bilguvar, K., Fu, X.D., Trotta, C.R., Gabriel, S., Reis, A., Gunel, M., Baas, F., Gleeson, J.G., CLP1 Founder Mutation Links tRNA Splicing and Maturation to Cerebellar Development and Neurodegeneration, 651-63, Copyright (2014) with permission from Elsevier. Full text @ Cell