FIGURE

Fig. S8

ID
ZDB-FIG-170428-2
Publication
Wiessner et al., 2017 - Mutations in INPP5K, Encoding a Phosphoinositide 5-Phosphatase, Cause Congenital Muscular Dystrophy with Cataracts and Mild Cognitive Impairment
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Fig. S8

Neuromuscular junction morphology in inpp5ka+inpp5kb morphants.

For whole-mount immunofluorescence staining, 48-hpf zebrafish embryos were fixed in 4% paraformaldehyde in PBS at 4°C overnight and blocked for 1 hr at RT in 5% horse serum in PBS, 0.1% Tween-20. Presynaptic motor nerve endings were visualized by incubation with mouse anti-synaptic vesicle protein 2 antibody (SV2, DSHB; 1:200) overnight at 4°C followed by Alexa Fluor 488-conjugated goat anti-mouse IgG antibody for 1 hr at RT. Acetylcholine receptors (AChR) were labelled with Alexa Fluor 594-conjugated α- bungarotoxin (Thermo Fisher Scientific; 1:1,000). Images were captured with a Nikon A1R confocal microscope (Nikon). Images for inpp5ka+inpp5kb morphants represent mild, moderate and severe phenotypes (from left to right) as staged macroscopically. Both in controls and in inpp5ka+inpp5kb morphants, motor axons had made branches into somites where they formed contacts with AChR clusters. Scale bars = 50 μm.

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Knockdown Reagents:
Observed In:
Stage: Long-pec

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 @ Am. J. Hum. Genet.