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Fig. 4
Limb defects and gene expression changes in Nipbl-deficient zebrafish and mice. (A) Defects in pectoral fin development are observed in nipbla/b morphants; defects are partially rescued by injection of exogenous nipbla mRNA. Dorsal view of zebrafish larvae at 76 hpf. (B) Plots of fin lengths in morphants and controls at 76 hpf. (C) Expression of shha (ISH) in pectoral limb buds (arrows) is reduced in nipbla/b morphants at three indicated stages of development. (D) Whole-mount ISH for Shh in E10.5 mouse embryos; expression is reduced in Nipbl+/- hindlimb buds compared to wildtype (WT). Arrowheads indicate hindlimbs; arrows indicate forelimbs. Scale bar = 0.5 mm. (E) Quantification of Shh ISH shown in D; *P < 0.05, **P < 0.01. (F) Zebrafish larvae injected with low doses of med12 or nipbla/b MOs display small reductions in fin size compared to controls; defects are enhanced when morphants are treated with combined nipbla/b and med12 MOs. (G) Plots of fin lengths in morphants and controls in the four conditions. (H) Changes in hox gene expression in morphants treated with nipbla/b, med12, and nipbla/b + med12 MOs at 36 hpf. Adapted from [Muto et al., 2014].