FIGURE

Figure 7

ID

ZDB-FIG-210512-58

Publication

Sofou et al., 2021 - Bi-allelic VPS16 variants limit HOPS/CORVET levels and cause a mucopolysaccharidosis-like disease

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Figure 7

Zebrafish embryos deficient in <italic>vps16</italic> demonstrate abnormalities in LROs and myelination

Schematic representation of the HOPS complex, indicating the high amino acid identity between human and zebrafish for each subunit.

Comparison of control and vps16 embryos at 5 dpf. Compared with control, vps16 embryos have reduced pigmentation in the skin and retinal pigment epithelium and do not develop a swim bladder (arrows). Magnified views show altered melanocyte morphology on the head and pale, translucent eyes.

C, D

Myelination, as reported using the Tg(mbp:gfp‐caax) transgenic, is significantly reduced in vps16 crispants. The yellow box indicates the region assessed in (D). Scale bar 100 μm. Bar graphs represent data as mean ±SEM. Statistical comparisons by Mann–Whitney U‐tests (n = 6). **P < 0.01.

Expression Data

Gene:	EGFP
Fish:	ue2Tg + CRISPR1-vps16
Knockdown Reagent:	CRISPR1-vps16
Anatomical Term:	brain myelin sheath
Stage:	Day 5

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	AB + CRISPR1-vps16 ue2Tg + CRISPR1-vps16
Knockdown Reagent:	CRISPR1-vps16
Observed In:	brain myelin sheath eye head melanocyte hindbrain myelination integument midbrain myelination retinal pigmented epithelium swim bladder
Stage:	Day 5

Phenotype Detail

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 @ EMBO Mol. Med.