FIGURE

Fig. 1

ID
ZDB-FIG-210602-11
Publication
Wood et al., 2021 - FKRP-dependent glycosylation of fibronectin regulates muscle pathology in muscular dystrophy
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Fig. 1

<italic>fkrp</italic><sup><italic>−/−</italic></sup> fish possess a basement membrane failure phenotype.

ad Muscle fibre-crossing and detachment phenotypes at the muscle basement membrane of fkrp−/ mutant larvae. ab” Z-projected images from the entire mediolateral extent of the myotome centred on the region of the anal pore. Phalloidin conjugated to TRITC (red) stains for f-actin as a marker of skeletal muscle and paxillin (green) visualises vertical myosepta muscle basement membrane integrity. White arrowheads: muscle fibres crossing at defective muscle basement membranes, asterisks: muscle fibre detachment. 3 dpf, scale bar = 100 μm. 5 dpf, scale bar = 75 μm, white boxes in a’, b’ denote the high-magnification views presented in a” and b”. c, d Quantitation of fibre detachment (c) and basement membrane crossing events (d) per myosepta in wild-type (n = 9) or fkrp mutant larvae (n = 9) over five independent experiments, analysed using two-way ANOVA multiple comparisons assuming non-parametric data, the significance of ***(P = < 0.0001), error bars represent SEM. e Physiological analysis of muscle function. The maximum active force (mN) generated over a specific time interval (ms) was measured in individual genotypes of 6 dpf larvae represented in single-twitch recordings of fkrp+/+ sibling (sib) black line (n = 8), fkrp−/ green line (n = 6) and dag1/− purple line (n = 5).

Expression Data

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Observed In:
Stage Range: Protruding-mouth to Day 6

Phenotype Detail
Acknowledgments
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