FIGURE

Fig. 6

ID

ZDB-FIG-250813-27

Publication

Halford et al., 2025 - TMEM63A, associated with hypomyelinating leukodystrophies, is an evolutionarily conserved regulator of myelination

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Fig. 6

TMEM63A function in myelination is conserved in zebrafish. (A) Topology map of Danio rerio Tmem63a; region targeted by CRISPR/Cas9 flanks M212 (highlighted in red and indicated by the red arrow on the map). Representative micrographs comparing control Cas9 injected or tmem63a sgRNAs injected in Tg(mbp:egfp-caax) fish at 4 dpf (B) and 5 dpf (C). D, dorsal and V, ventral spinal cord. Cartoon larva illustrates ROI (red box). (Scale bar, 25 µm.) (D) Mean mbp:egfp-caax intensity of dorsal (Left) and ventral (Right) spinal cord from z-stack sum projections of Cas9 and tmem63a F0 fish. (n = 25 to 32 animals per crispant and age, mixed effects analysis of multiple comparison by uncorrected Fisher’s LSD).

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	WT + CRISPR1-tmem63a
Knockdown Reagent:	CRISPR1-tmem63a
Observed In:	spinal cord myelin sheath
Stage Range:	Day 4 to 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 @ Proc. Natl. Acad. Sci. USA