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Fig. 4

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ZDB-IMAGE-220720-33
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Figures for Berdowski et al., 2022
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Fig. 4

Homozygous missense variants in csf1ra causes myelin and behavioral abnormalities related to leukodystrophy. a Representative images of the midbrain (top) and the hindbrain (bottom) of csf1ra+/+ (green), csf1raV614M/+ (purple) and csf1raV614M/V614M (orange) larvae in a sox10:RFP background, visualizing oligodendrocytes, at 5 dpf. Lower right: schematic of zebrafish embryonic midbrain (mb) and hindbrain (hb). b Quantification of the number of sox10:RFP + oligodendrocytes in the midbrain and hindbrain at 5 dpf in WT (green), csf1raV614M/+ (purple) and csf1raV614M/V614M (orange) larvae. c Representative images of the hindbrain of WT (green), csf1raV614M/+ (purple) and csf1raV614M/V614M (orange) larvae in a mbp:GFP-CAAX background, visualizing myelin sheaths, at 5 dpf. d Quantification of the total mbp:GFP + myelinated area (μm2) in the hindbrain at 5 dpf in WT (green), csf1raV614M/+ (purple) and csf1raV614M/V614M (orange) larvae. e Representative images of in situ hybridization of plp1b (top) and mbpa (bottom) in WT (left) and csf1raV614M/V614M (right) larvae at 5 dpf, showing reduce number of plp1b + mature myelinating oligodendrocytes (11/11 larvae) and reduced mbpa + myelin sheaths and myelinating oligodendrocytes (8/9 larvae) in csf1raV614M/V614M larvae. Dashed lines show the hindbrain (hb). f Representative graph showing the total distance traveled (mm) by larvae per 1 min during the dusk–dawn routine (total time: 3 h 15 min), of csf1ra mutants in a csf1rb-deficient background. Grey shading shows the standard error of the mean (SEM). g Quantification of the total distance moved throughout the experiment excluding the dark period. n = 16 larvae per genotype. Days-post-fertilization (dpf), hindbrain (hb), midbrain (mb). One-way ANOVA test was preformed to test for significance (p < 0.05). Error bars represent SD, unless stated otherwise. *p < 0.05, **p < 0.01 ***p < 0.001. Scale bars equal 100 μm

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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 @ Acta Neuropathol.