FIGURE

Fig. 5

ID

ZDB-FIG-161228-2

Publication

Giunta et al., 2016 - Altered RNA metabolism due to a homozygous RBM7 mutation in a patient with spinal motor neuropathy

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Fig. 5

Confocal microscopy analysis of PCs (A,B,E,F) and neuromuscular junctions (C,D,G,H) in zebrafish upon injection of three different MOs. PCs fail to differentiate in rbm7-MO (B), exosc8-MO (E) and exosc3-MO (F) zebrafish compared with ctrl-MO fish (A). The percentage of fish with cerebellar defects varies significantly between morphants, reflecting what observed in patients. Motor axon growth is defective in all three morphants (D,G,H); arrows point at abnormally short motor axons. Interestingly, only in exosc8-MO the motor neuron axon branches in close proximity of the spinal cord instead of more ventrally as in the ctrl-MO fish (G). Scale bar = 100 μm. We show the quantity and respective percentage of fish with cerebellar defects (I). Axonal defects in different morphant and phenotypical classes (only mild and moderate phenotypes were considered for this analysis) (J). The number of defective structures increases with the severity of the phenotype.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	rw0Tg + MO1-exosc3 rw0Tg + MO1-rbm7 rw0Tg + MO2-exosc8
Knockdown Reagents:	MO1-exosc3 MO1-rbm7 MO2-exosc8
Observed In:	axonogenesis cerebellar Purkinje cell layer structural organization cerebellum cerebellum Purkinje cell motor neuron axon
Stage Range:	Long-pec to Day 4

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 @ Hum. Mol. Genet.