FIGURE

Fig 6

ID

ZDB-FIG-220227-26

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 6

Residual sarcomeres assemble within <italic toggle='yes'>capza1a</italic> and <italic toggle='yes'>capza1b</italic> depleted zebrafish.

(A) At 2 dpf, Lifeact-GFP (green) and mCherry-CaaX (red) highlighted the sarcomere organisation and myofibril striation within muscle fibres of WT siblings. In two out of four analysed capza1b^ex5 homozygotes, thin filament deposits (arrows) were detected at the peripheral ends of some fibres. Although myofibril striation (arrowheads) was detected in capza1b^ex5 homozygotes that were hetero- or homozygous for capza1a^ex5, thin filament deposits (arrows) were frequently located at the peripheral myofiber ends. (B) At 2 dpf, highly organised sarcomeres were found on transmission electron micrographs of siblings and capza1a^ex5;capza1b^ex5 compound homozygotes. However, electron-dense aggregates with a lattice structure (arrowheads) and filament deposits (star) were present close to the myosepta (arrow) in the compound homozygotes. Scale bar sizes are indicated.

Expression Data

Genes:	EGFP mCherry
Fish:	pc21Tg; pc22Tg capza1b^pc56/pc56; pc21Tg; pc22Tg capza1a^pc55/+; capza1b^pc56/pc56; pc21Tg; pc22Tg capza1a^pc55/pc55; capza1b^pc56/pc56; pc21Tg; pc22Tg
Anatomical Terms:	myotome sarcolemma myotome sarcomere myotome striated muscle thin filament
Stage:	Long-pec

Expression Detail

Gene	Fish	Conditions	Stage	Anatomy	Assay
EGFP	capza1a^pc55/+; capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome sarcomere	IFL
EGFP	capza1a^pc55/+; capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome striated muscle thin filament	IFL
EGFP	capza1a^pc55/pc55; capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome sarcomere	IFL
EGFP	capza1a^pc55/pc55; capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome striated muscle thin filament	IFL
EGFP	capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome sarcomere	IFL
EGFP	capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome striated muscle thin filament	IFL
EGFP	pc21Tg; pc22Tg	standard conditions	Long-pec	myotome striated muscle thin filament	IFL
mCherry	capza1a^pc55/+; capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome sarcolemma	IFL
mCherry	capza1a^pc55/pc55; capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome sarcolemma	IFL
mCherry	capza1b^pc56/pc56; pc21Tg; pc22Tg	standard conditions	Long-pec	myotome sarcolemma	IFL

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Antibody Labeling

Phenotype Data

Fish:	capza1b^pc56/pc56; pc21Tg; pc22Tg capza1a^pc55/+; capza1b^pc56/pc56; pc21Tg; pc22Tg capza1a^pc55/pc55; capza1b^pc56/pc56 (TU) capza1a^pc55/pc55; capza1b^pc56/pc56; pc21Tg; pc22Tg
Observed In:	myotome peripheral region myotome protein-containing complex myotome sarcomere myotome striated muscle thin filament
Stage:	Long-pec

Phenotype Detail

Acknowledgments

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