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Figure 3-figure supplement 1

ID: ZDB-IMAGE-200511-26
Publication: Volpatti et al., 2020 - Identification of drug modifiers for RYR1 related myopathy using a multi-species discovery pipeline
All Figures: Figures for Volpatti et al., 2020

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Figure 3-figure supplement 1 Phenotypes of zebrafish <italic>ryr1</italic> mutants considered for chemical screening.

(A) ryr1a;ryr1b double mutants exhibit complete lack of movement as shown by optovin-6b8 induced swimming (n = 24 per genotype; Student’s T-test: ***p<0.001). They do not respond to touch stimuli either data not shown; independently shown by Chagovetz et al. (2019). (B) The ryr1a;ryr1b double mutants exhibit early larval lethality with a median survival of 5 days and maximum survival of 7 days (n = 58 per group; Mantel-Cox: ***p<0.001). (C) The ryr1b mutants show significantly reduced movement speed (n = 48; 5.42 ± 0.1327 mm/s) at four dpf by optovin-6b8 induced swimming versus WT siblings (n = 47; 8.598 ± 0.2728 mm/s) (Student’s t-test: ***p<0.001). However, there is sufficient overlap with WT siblings to preclude large-scale screening.

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Phenotype details

Acknowledgments

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