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Fig. 5

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ZDB-IMAGE-230205-24
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Figures for Marcogliese et al., 2022
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Fig. 5

Loss of irf2bpl in zebrafish leads to defective locomotor activity and increased Wnt1 signaling.

(A) Distance moved (in millimeters) of mutants (irf2bpl−/−) and wild-type sibling controls (irf2bpl+/+) measured at 7 d.p.f. in a locomotor assay. During the assay, test animals were first habituated for 30 min, followed by five light-dark cycles for a total of 30 min (n = 36 and 47 for irf2bpl+/+and irf2bpl−/− animals, respectively). (B) Representative Western blot and quantification of Wnt1 protein expression in irf2bpl−/− brain compared to irf2bpl+/+ controls (n = 3 per group). (C) Analyses of lef1 transcript expression real-time qPCR in irf2bpl−/− treated with DMSO, SSTC3, or XAV-939, compared to irf2bpl+/+ treated with DMSO (n = 8 per group). (D and E) Treatment with SSTC3 significantly improved the irf2bpl−/− mutants’ bout activity when transitioning from a light to dark condition, compared to mutants treated with DMSO (n = 22 and 24 for DMSO-treated irf2bpl+/+ and irf2bpl−/− animals; 25 and 23 for SSTC3-treated irf2bpl+/+ and irf2bpl−/−, respectively). (F and G) Similar to SSTC3, treatment with XAV-939 significantly improved the mutants’ bout activity compared to DMSO-treated mutants (n = 21 and 19 for DMSO-treated irf2bpl+/+ and irf2bpl−/− animals; 36 and 45 for XAV-939–treated irf2bpl+/+ and irf2bpl−/− animals, respectively). Results are means ± SEM (*P < 0.05, **P < 0.001, and ****P < 0.0001).

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