FIGURE SUMMARY
Title

WNT5A mutations in patients with autosomal dominant Robinow syndrome

Authors
Person, A.D., Beiraghi, S., Sieben, C.M., Hermanson, S., Neumann, A.N., Robu, M.E., Schleiffarth, J.R., Billington, C.J. Jr, van Bokhoven, H., Hoogeboom, J.M., Mazzeu, J.F., Petryk, A., Schimmenti, L.A., Brunner, H.G., Ekker, S.C., and Lohr, J.L.
Source
Full text @ Dev. Dyn.

Cysteine mutations in WNT5A reduce the ability of this protein to activate noncanonical Wnt signaling in zebrafish embryos. A,B: Uninjected brightfield (A) and fluorescent images of insulin-enhanced green fluorescent protein (eGFP) transgenic zebrafish embryos (B) at 30 hours postfertilization (hpf) showing insulin-positive cells have coalesced to form an islet. C,D: Insulin-eGFP-positive cells in embryos injected with WNT5A mRNA do not coalesce properly, providing an in vivo assay for noncanonical Wnt5a activity. E: In situ hybridization showing insulin-expressing cells coalescing to form the zebrafish islet at 30 hpf. F-H,K: Injections with WNT5A mRNA (F),WNT5AC182R mRNA (G,H), or WNT5AC83R mRNA (not shown) show scattered insulin-expressing cells at 30 hpf, but the efficacy of this effect is reduced in WNT5AC182R (32%±3% n = 93; P = 0.00005) and WNT5AC83R (55% ± 9% n = 152; P = 0.002) mRNA injected embryos compared with WNT5A mRNA injected embryos (100% ± 6% n = 260) (K). I: Uninjected zebrafish embryo at 26 hpf. J: Zebrafish embryo at 26 hpf injected with dnWNT5A mRNA (25 pg) showing a bent tail phenotype similar to pipetail (Wnt5) mutants.

EXPRESSION / LABELING:
Genes:
Fish:
Anatomical Term:
Stage: Prim-15
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 @ Dev. Dyn.