tardbp^Y220X/Y220X zebrafish embryos injected with an ATG tardbpl morpholino (MO) displayed significant morphological defects and reduced survival.

A, Representative images of 2 dpf larvae from the following treatment groups: WT, WT + MO, tardbp^Y220X/+, tardbp^Y220X/Y220X, tardbp^Y220X/Y220X + Control MO (CoMo), tardbp^Y220X/Y220X + MO. Scale bar represents 1 mm, arrow indicates cardiac defect in the tardbp^Y220X/Y220X + MO treated larva. B, Eye diameter and C, body length was determined for each treatment group. tardbp^Y220X/Y220X + MO 2dpf larvae displayed significantly reduced eye diameter compared to all other treatment groups (p < 0.01). WT + MO, tardbp^Y220X/Y220X and tardbp^Y220X/Y220X + CoMo larvae displayed significantly reduced body length compared to WT larvae (p < 0.05) and tardbp^Y220X/Y220X + MO displayed significantly reduced body length compared to all treatment groups (p < 0.01). D, Quantification of gross morphological defects observed in all treatment groups indicating a higher incidence of defects in the tardbp^Y220X/Y220X + MO condition. E, Percentage survival curves of all treatment groups tracked over 11 days. tardbp^Y220X/Y220X + MO-treated larvae displayed reduced survival, with all animals dying by 10 dpf (p < 0.0001). N = 2, n = 50, *p < 0.05; **p < 0.01.

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Motor neuron projections in tardbp^Y220X/Y220X + MO larvae show increased number of orphaned presynaptic and postsynaptic puncta.

A, Representative images of single ventral root projection double-labelled for ZNP-1 (presynaptic marker, i) and sulforhodamine-conjugated αBTX (postsynaptic marker, ii). WT larvae display extensive co-localization of both ZNP-1 and αBTX (merged image, iii). Scale bar in (iii) and insets represent 25 μm and 10 μm respectively, arrowheads indicate orphaned ZNP-1 puncta and arrows indicate orphaned αBTX labelling. B, Quantification of the number synapses formed, quantified as the number of colocalized ZNP-1 and αBTX puncta. WT + MO (p < 0.05), tardbp^Y220X/Y220X (p < 0.01) and tardbp^Y220X/Y220X + MO larvae (p < 0.01) all had a significantly reduced number of synapses at the NMJ compared to WT larvae. C, Quantification of orphaned presynaptic ZNP-1 puncta over total number of ZNP-1 puncta. WT + MO, tardbp^Y220X/Y220X and tardbp^Y220X/Y220X + MO larvae displayed significantly higher proportion of orphaned ZNP-1 puncta compared to WT animals (p < 0.01). D, Quantification of orphaned postsynaptic αBTX staining over total number of αBTX puncta. tardbp^Y220X/Y220X + MO fish displayed significantly higher proportion of orphaned αBTX puncta compared to WT larvae (p < 0.05). Numbers in parentheses represent the number of somites analyzed for each treatment group. Data expressed as mean ± SEM: *p < 0.05; **p < 0.01.