IMAGE

Fig 1

ID
ZDB-IMAGE-220227-23
Genes
Antibodies
Source
Figures for Berger et al., 2022
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Figure Caption

Fig 1 Loss of <italic toggle='yes'>lmod3</italic> compromises muscle integrity.

(A) The mutant line lmod3sa13018 harboured a premature stop codon within exon 3 of lmod3, the start codon of which locates at 13’814’978 bp on chromosome 23. Scissors mark single gRNA targets and scale is 1 kbp. (B) The codon GAA encoding for E338 was changed to the nonsense mutation TAA within lmod3sa13018. (C) Western blot analysis with antibodies against human LMOD3 detected Lmod3 epitope (76 kDa predicted) in siblings (left lane), but not in lmod3sa13018 homozygotes (right lane). ß-tubulin was used as loading control. (D) In contrast to siblings, the swimming bladder (arrowhead) of 4-dpf-old lmod3sa13018 homozygotes was not inflated. The bar indicates larval length without the caudal fin. (E) Whereas 4-dpf-old siblings were 3.68 ± 0.03 mm long, the larval length of lmod3sa13018 homozygotes was with 3.51 ± 0.04 mm significantly shorter (n = 10 larvae). (F) Under polarised light, the muscle of lmod3sa13018 homozygotes appeared darker compared to their siblings due to a reduction in birefringence. (G) After rescaling to siblings, the birefringence of lmod3sa13018 homozygotes was significantly reduced to 79 ± 2% compared to their 3-dpf-old siblings. Crosses represent averaged birefringence of clutches with a minimum of 5 larvae per genotype (n = 5 clutches). At 6 dpf, the birefringence of lmod3sa13018 homozygotes remained significantly reduced to 54 ± 2% of their siblings (n = 8 individual larvae). (H) Signs of fibrosis were absent on cross sections of 6-dpf-old lmod3sa13018 siblings and homozygotes stained with hematoxylin and eosin (H&E). (I) lmod3ex3 harboured a deletion of 1385 bp and insertion of 5 bp in exon 3 of lmod3. (J) After rescaling to siblings, the birefringence reduction of lmod3ex3 homozygotes was 83 ± 2% at 3 dpf and 55 ± 2% at 6 dpf. Similarly, lmod3ex3/sa13018 compound heterozygotes had a highly significant reduction in birefringence to 83 ± 2% at 3 dpf and to 58 ± 2% at 6 dpf. At 3 dpf, crosses represent averaged birefringence of clutches with a minimum of 5 larvae per genotype (n = 5 clutches) and individual larvae at 6 dpf (n = 8 larvae). Data are presented as mean ± SEM; ** P < 0.01 and *** P < 0.001 were calculated by Student’s t-test. Scale bar sizes are indicated.

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