FIGURE

Fig. 2

ID

ZDB-FIG-240715-62

Publication

Banerjee et al., 2024 - Hypomyelinated vps16 Mutant Zebrafish Exhibit Systemic and Neurodevelopmental Pathologies

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Fig. 2

vps16(-/-) mutants at 7 dpf show retinal pathology including photoceptor degeneration, reactive gliosis of Müller glia (MG), and cell death. (A,B) Immunolocalization of Arrestin (Zpr-1) (green) in double cone photoreceptors. (A) Siblings (n = 15) show normal Zpr-1 immunolabeling in perinuclear domain of red/green double cones. (B) Zpr-1 immunolabeling observed throughout cytoplasm of hypertrophied and degenerating cones in vps16(-/-) mutants (n = 18). (C) Graph representing quantification of Zpr-1 immunolocalization in 7 dpf siblings and vps16(-/-) mutants. *** p = 0.0009. (D,E) Immunolocalization of Zpr-3 (green) in outer segments of rod photoreceptors. (D) Outer segments of healthy rod photoreceptors in siblings (n = 13) show normal length and structure. (E) Truncated outer segments of degenerating rod photoreceptors observed in vps16(-/-) mutants (n = 11). (F) Graph representing quantification of average length of rod outer segments immunolabeled with Zpr-3 in 7 dpf siblings and vps16(-/-) mutants. **** p < 0.0001. (G,H) Immunolocalization of glutamine synthetase (G.S.) (green) in MG. (G) Normal levels of G.S. immunolabeling in outer regions of MG of siblings (n = 14). (H) G.S. immunolabeling in ends of MG in outer retina showing reactive gliosis in vps16(-/-) mutants (n = 18). (I) Graph representing quantification of G.S. immunolocalization in 7 dpf siblings and vps16(-/-) mutants. p = 0.06. (J,K) Immunolocalization of PCNA-positive retinal progenitors (green) in CMZ. (J) PCNA-positive retinal progenitors shown in CMZ of siblings (n = 9). (K) Normal population of PCNA-positive retinal progenitors shown in CMZ of vps16(-/-) mutants (n = 8). (L) Graph representing quantification of PCNA immunolocalization in retinal CMZ of 7 dpf siblings and vps16(-/-) mutants. “N.S.” = not significant. (M–O) TUNEL-positive cells (green) in retinal sections. (M) Minimal to no TUNEL-positive apoptotic cells seen in sibling retina (n = 16). (N) Large number of TUNEL-positive apoptotic cells observed in vps16(-/-) mutant retina (n = 16), specifically in outer nuclear layer. (O) Graph representing quantification of average number of TUNEL-positive apoptotic cells in retinal sections of 7 dpf siblings and vps16(-/-) mutants. **** p < 0.0001. Scale bar in panel A = 50 microns for all images. Blue = TO-PRO-3, a nuclear stain. Error bars indicate SEM.

Expression Data

Antibodies:	Ab2-pcna zpr-1 zpr-3
Fish:	AB vps16^wsu4/wsu4 (AB)
Anatomical Terms:	ciliary marginal zone stem cell long double cone cell long double cone cell cytoplasm Muller cell retinal rod cell rod photoreceptor outer segment short double cone cell short double cone cell cytoplasm
Stage:	Days 7-13

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	vps16^wsu4/wsu4 (AB)
Observed In:	ciliary marginal zone cell population proliferation long double cone cell cytoplasm Muller cell cell population proliferation Muller cell reactive gliosis retina apoptotic process retinal rod cell rod photoreceptor outer segment short double cone cell cytoplasm
Stage:	Days 7-13

Phenotype Detail

Acknowledgments

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