FIGURE

FIGURE 3

ID

ZDB-FIG-260319-80

Publication

Pistocchi et al., 2026 - Neurotrophic Modulation Restores Motor and Developmental Defects in Zebrafish Models of ints11 Deficiency

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FIGURE 3

ints11 deficiency impairs nervous system development and neuronal marker expression. (A–C) Representative WISH for pax2a and krox20 of 24 hpf (A) controls, (B) ints11‐LoF, and (C) rescue (ints11‐LoF injected with ints11 mRNA) embryos. On the top‐right the number of embryos with similar hybridization signal. Scale bar indicates 100 μm. (D–F) Representative WISH for islet1 of 2.5 dpf (D) controls, (E) ints11‐LoF, and (F) rescue (ints11‐LoF injected with ints11 mRNA) embryos. On the top‐right the number of embryos with similar hybridization signal. Scale bar indicates 200 μm. (G–L) RT‐qPCR expression analyses at 2.5 and 4 dpf for (G) nestin, (H) runx1, (I) gfap, (J) mag, (K) map2, and (L) sox10 in the different categories. Data are presented as mean ± SEM after analyses with the 2^−ddCt method. According to 2^−ddCt method, ctrl is set at mean 1. nestin 2.5 dpf (ints11‐LoF: 1.672 ± 0.2015; rescue: 1.226 ± 0.2294); nestin 4 dpf (ints11‐LoF: 1621 ± 0.1508; rescue: 0.8567 ± 0.2164); runx1 2.5 dpf (ints11‐LoF: 0.4780 ± 0.1324; rescue: 0.7421 ± 0.3036); runx1 4 dpf (ints11‐LoF: 1026 ± 0.0038; rescue: 1493 ± 0.0570); gfap 2.5 dpf (ints11‐LoF: 0.5843 ± 0.1378; rescue: 1.086 ± 0.2703); gfap 4 dpf (ints11‐LoF: 0.8523 ± 0.0028; rescue: 1.358 ± 0.0212); mag 2.5 dpf (ints11‐LoF: 0.2227 ± 0.1005; rescue: 1675 ± 1036); mag 4 dpf (ints11‐LoF: 0.4137 ± 0.0861; rescue: 0.8724 ± 0.0775); map2 2.5 dpf (ints11‐LoF: 0.6524 ± 0.0716; rescue: 1.006 ± 0.1221); map2 4 dpf (ints11‐LoF: 0.6867 ± 0.0316; rescue: 0.9025 ± 0.0126); sox10 2.5 dpf (ints11‐LoF: 0.9946 ± 0.1900; rescue: 0.9902 ± 0.1396); sox10 4 dpf (ints11‐LoF: 0.6712 ± 0.0265; rescue: 0.9346 ± 0.0015). Each dot represents an independent biological batch/experiment and is the mean of three technical replicates. Kruskal–Wallis non‐parametric test with Dunn's post hoc. *p < 0.05, ns: Not significant. Refer to Table S4 for raw statistical data. Cn, cranial nerve; e, Epifisis; fb, forebrain; hb, hindbrain; icl, inner cell layer; mhb, midbrain‐hindbrain boundary; ot, optic tectum; r3, rhombomere 3; r5, rhombomere 5; rgc, retinal ganglion cell; sc, spinal cord.

Expression Data

Genes:	egr2b gfap isl1a mag map2 nes pax2a runx1 sox10
Fish:	AB AB + CRISPR1-ints11 + CRISPR2-ints11 AB + MO2-ints11
Knockdown Reagents:	CRISPR1-ints11 CRISPR2-ints11 MO2-ints11
Anatomical Terms:	cranial nerve V cranial nerve VII cranial nerve X epiphysis forebrain hindbrain midbrain hindbrain boundary optic tectum retinal ganglion cell retinal inner nuclear layer rhombomere 3 rhombomere 5 spinal cord whole organism
Stage Range:	Prim-5 to Day 4

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	AB + CRISPR1-ints11 + CRISPR2-ints11 AB + MO2-ints11
Knockdown Reagents:	CRISPR1-ints11 CRISPR2-ints11 MO2-ints11
Observed In:	cranial nerve V cranial nerve VII cranial nerve X epiphysis forebrain hindbrain midbrain hindbrain boundary optic tectum retinal ganglion cell retinal inner nuclear layer rhombomere 3 rhombomere 5 spinal cord whole organism
Stage Range:	Prim-5 to Day 4

Phenotype Detail

Acknowledgments

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