FIGURE

Fig. 2

ID

ZDB-FIG-090224-47

Publication

Dowling et al., 2009 - Loss of myotubularin function results in T-tubule disorganization in zebrafish and human myotubular myopathy

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Fig. 2

Abnormal motor function in myotubularin morphants.

(A) Quantitation of spontaneous embryo coiling at 24 hpf (see also Supplemental Videos 1 and 2). On average, CTL morphants coiled 10.2 times in 15 seconds, while MTM morphants coiled only 5.2 times. (B) Quantitation of chorion hatching in 60 hpf morphants. 87.2% of CTL morphants are hatched from their protective chorions by 60 hpf, as opposed to only 35.3% of MTM morphants. (C) Touch-evoked swimming was video captured in 72 hpf morphants. As expected, CTL morphants responded to tactile stimuli with a rapid escape response contraction followed by swimming. Conversely, MTM morphants displayed a weak escape contraction followed by “twittering” movements (example at 33 ms) but never normal swimming. Scale bar = 1 mm.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	WT + MO1-mtm1
Knockdown Reagent:	MO1-mtm1
Observed In:	hatching musculoskeletal movement, spinal reflex action thigmotaxis
Stage Range:	Pec-fin to Protruding-mouth

Phenotype Detail

Acknowledgments

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