FIGURE

Fig. 3

ID
ZDB-FIG-240206-71
Publication
Guglielmi et al., 2023 - A novel in-frame deletion in MYOT causes an early adult onset distal myopathy
Other Figures
All Figure Page
Back to All Figure Page
Fig. 3

(A, B) Birefringence analysis of wildtype embryos injected with Tol2 plasmids harboring either human wild-type MYOT full cDNA (wtMYO) or human MYOT c.11_28del (mutMYO) was performed at 48 hpf. In (A), representative lateral images of single non-injected, wtMYO-, and mutMYO-injected embryos. Bar: 50 μm. The bar graph in (B) reports the birefringence phenotypes observed in not injected (n = 200), wtMYO (n = 55), and mutMYO (n = 76) embryos. Data are plotted as total percentage of fish showing wildtype (white closed bars) or myopathic (black closed bars) birefringence phenotypes. (C) Number of spontaneous coiling events (tail flips) performed in 15 s have been recorded at 24 hpf in non-injected (n = 330), wtMYO-injected (n = 80) and mutMYO-injected (n = 85) embryos and plotted in the bar graph. Values are reported as the average of the number of tail flips recorded for single embryos at 24 hpf ± SEM ***p < 0.001. (D) Touch-evoked escape response at 48 hpf was checked in non-injected (n = 342), wtMYO (n = 86), and mutMYO (n = 78). Arbitrary values (0–3) have been assigned to each larva based on the observed response. Data in the bar graph are reported as the average of escape values for single embryos ± SEM *p < 0.05, **p < 0.01, ***p < 0.001.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data

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 @ Clin. Genet.