FIGURE

Fig 2

ID

ZDB-FIG-220227-21

Publication

Berger et al., 2022 - Genetic dissection of novel myopathy models reveals a role of CapZα and Leiomodin 3 during myofibril elongation

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Fig 2

Lmod3-deficiency leads to weakening of the trunk and head musculature.

(A) Peak active force of single-twitch contractions generated by 6-dpf-old individual lmod3^sa13018 larvae was significantly weaker compared to their siblings. Siblings generated a maximal active force of 0.82 ± 0.01 mN, compared to and 0.19 ± 0.03 mN generated by lmod3^sa13018 homozygotes (n = 4). (B) Z-stack projections of ventral views depict the cephalic musculature marked by GFP driven by the Tg(−503unc:GFP) transgene. In contrast to 4-dpf-old siblings, a gap between the two hyohyoideus (hh) muscles was formed in the lower jaw of lmod3^sa13018 homozygotes (asterix). (C) At 7dpf, focus stacks of Alcian Blue cartilage stains revealed a widened jaw within lmod3^sa13018 mutants. (D) The angle formed by the ceratohyal cartilages (dotted lines) was significantly wider in lmod3^sa13018 homozygotes compared to siblings (87 ± 2° and 65 ± 1°, respectively. n = 6). Data are presented as mean ± SEM; *** P < 0.001 calculated by Student’s t test. Scale bar sizes are 200 μm.

Expression Data

Gene:	EGFP
Fish:	pc11Tg lmod3^{sa13018/sa13018}; pc11Tg
Anatomical Term:	hyohyoideus
Stage:	Day 4

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	lmod3^{sa13018/sa13018} (TU) lmod3^{sa13018/sa13018}; pc11Tg
Observed In:	ceratohyal cartilage left side ceratohyal cartilage right side hyohyoideus left side hyohyoideus right side trunk musculature skeletal muscle contraction
Stage Range:	Day 4 to Days 7-13

Phenotype Detail

Acknowledgments

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