Advances in the Understanding of Skeletal Myopathies from Zebrafish Models

Baxter, Emily Claire; Bryson-Richardson, Robert J.

doi:10.1007/978-981-13-1879-5_9

Cited by 1 publication

(4 citation statements)

References 227 publications

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“…a birefringence assay, which examines changes in the polarity of light as it travels through the highly ordered muscle structure. Zebrafish can also readily absorb drugs added to their water and have been successfully utilized for large-scale drug screens to rescue damage to muscles (Baxter and Bryson-Richardson, 2018). Moreover, zebrafish was proposed as a model for the study of aging over a decade ago (Kishi et al, 2003).…”

Section: Zebrafishmentioning

confidence: 99%

“…By 24 hpf, all segmental blocks of muscle, or myotomes (Box 1), are present, partitioned by tendon-like junctions, enabling the embryo to coil and even respond to touch. The muscle becomes fully differentiated by 48 hpf and the larva is freely swimming by 96 hpf (Baxter and Bryson-Richardson, 2018;Stickney et al, 2000;Maves, 2014).…”

Section: Zebrafishmentioning

confidence: 99%

“…Zebrafish skeletal muscle is segmented into repeated chevronshaped myotomes, composed of slow and fast muscle (Box 1) fibers organized along the head-to-tail axis (Lin, 2012;Baxter and Bryson-Richardson, 2018). The partition between each pair of adjacent somites is termed the vertical myoseptum, and the one between the dorsal and ventral halves is termed the horizontal myoseptum.…”

Section: Zebrafishmentioning

confidence: 99%

“…Steffen et al (2007) searched the zebrafish genome for 29 human muscular dystrophy genes and identified orthologs for 28 of them. In a more recent systematic (Clemen et al, 2010) literature review, Baxter and Bryson-Richardson (2018) noted that zebrafish models currently exist for 75 of 121 (62%) of all known skeletal myopathy-associated genes. Most of these genes also show conserved syntenic relationships with humans, reflecting important functional and regulatory relationships between these orthologous genes (Irimia et al, 2012).…”

Section: High Conservation In Coding Dnamentioning

confidence: 99%

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Zebrafish models of sarcopenia

2020

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Sarcopeniathe accelerated age-related loss of muscle mass and functionis an under-diagnosed condition, and is central to deteriorating mobility, disability and frailty in older age. There is a lack of treatment options for older adults at risk of sarcopenia. Although sarcopenia's pathogenesis is multifactorial, its major phenotypesmuscle mass and muscle strengthare highly heritable. Several genome-wide association studies of musclerelated traits were published recently, providing dozens of candidate genes, many with unknown function. Therefore, animal models are required not only to identify causal mechanisms, but also to clarify the underlying biology and translate this knowledge into new interventions. Over the past several decades, small teleost fishes had emerged as powerful systems for modeling the genetics of human diseases. Owing to their amenability to rapid genetic intervention and the large number of conserved genetic and physiological features, small teleostssuch as zebrafish, medaka and killifishhave become indispensable for skeletal muscle genomic studies. The goal of this Review is to summarize evidence supporting the utility of small fish models for accelerating our understanding of human skeletal muscle in health and disease. We do this by providing a basic foundation of the (zebra)fish skeletal muscle morphology and physiology, and evidence of muscle-related gene homology. We also outline challenges in interpreting zebrafish mutant phenotypes and in translating them to human disease. Finally, we conclude with recommendations on future directions to leverage the large body of tools developed in small fish for the needs of genomic exploration in sarcopenia.

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Section: Zebrafishmentioning

confidence: 99%

Section: Zebrafishmentioning

confidence: 99%

Section: Zebrafishmentioning

confidence: 99%