Key result
The accordion mutation in the atp2a1 gene (SERCA1) causes significantly slower muscle relaxation and cytosolic Ca2+ decay in zebrafish embryos, serving as a model for Brody disease.
Why the study?
Does the accordion mutation in zebrafish cause a muscle relaxation defect due to impaired Ca2+ re-uptake?
Does the accordion mutation in zebrafish cause a muscle relaxation defect due to impaired Ca2+ re-uptake?
Zebrafish accordion mutants with a SERCA1 mutation exhibit impaired muscle relaxation and Ca2+ re-uptake, serving as a potential animal model for Brody disease.
Answers are grounded in this paper and cite their sources.
Offers zebrafish SERCA1 model for Brody disease; leaves open human translation and therapeutic implications.
When wild-type zebrafish embryos are touched at 24 hours post-fertilization (hpf), they typically perform two rapid alternating coils of the tail. By contrast, accordion (acc) mutants fail to coil their tails normally but contract the bilateral trunk muscles simultaneously to shorten the trunk, resulting in a pronounced dorsal bend. Electrophysiological recordings from muscles showed that the output from the central nervous system is normal in mutants, suggesting a defect in muscles is responsible. In fact, relaxation in acc muscle is significantly slower than normal. In vivo imaging of muscle Ca2+ transients revealed that cytosolic Ca2+ decay was significantly slower in acc muscle. Thus, it appears that the mutant behavior is caused by a muscle relaxation defect due to the impairment of Ca2+ re-uptake. Indeed, acc mutants carry a mutation in atp2a1 gene that encodes the sarco(endo)plasmic reticulum Ca2+-ATPase 1 (SERCA1), a Ca2+ pump found in the muscle sarcoplasmic reticulum (SR) that is responsible for pumping Ca2+ from the cytosol back to the SR. As SERCA1 mutations in humans lead to Brody disease, an exercise-induced muscle relaxation disorder, zebrafish accordion mutants could be a useful animal model for this condition.
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Hirata et al. (2004) studied Brody disease (animal model). accordion (acc) mutation in atp2a1 gene vs. wild-type zebrafish embryos was evaluated on muscle relaxation and cytosolic Ca2+ decay. The accordion mutation in the atp2a1 gene (SERCA1) causes significantly slower muscle relaxation and cytosolic Ca2+ decay in zebrafish embryos, serving as a model for Brody disease.
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