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Biology subjects

Shahriyari, M.

Publications and source records attributed to Shahriyari, M..

2 recordsLinked to original sources

Dystrophin is a mechanical tension modulator

Duchenne muscular dystrophy (DMD) represents the most common inherited muscular disease, where progressing muscle weakness leads to loss of ambulation and premature death. DMD is caused by mutations in the dystrophin gene, and is known to reduce the contractile capacity of muscle tissue both in vivo, and in reconstituted systems in vitro. However, these observations are based on mechanical studies that focused on stimulated contractions of skeletal muscle tissue. Seemingly paradoxical, upon evaluating bioengineered skeletal muscles produced from DMD patient derived myoblasts we observe an increase in myosin motor mediated homeostatic tissue tension that strongly correlates with decreased stimulated tissue strength, suggesting the involvement of dystrophin in regulating the baseline homeostatic tension level of tissue. This was confirmed by three independent pairs of isogenic cell lines, one of each lacking dystrophin compared to the isogenic dystrophin-expressing control. From this we speculate that the protective function of dystrophin also supports cellular fitness via active participation in the mechanosensation to achieve and sustain an ideal level of tissue tension. Hence, this study reveals fundamental novel insights into skeletal muscle biomechanics and into a new key mechanical aspect of DMD pathogenesis, provided by increased homeostatic tissue tension.

biophysics↗

Human engineered skeletal muscle of hypaxial origin from pluripotent stem cells with advanced function and regenerative capacity

Human pluripotent stem cell derived muscle models show great potential for translational research. Here, we describe developmentally inspired methods for derivation of skeletal muscle cells and their utility in three-dimensional skeletal muscle organoid formation as well as skeletal muscle tissue engineering. Key steps include the directed differentiation of human pluripotent stem cells to embryonic muscle progenitors of hypaxial origin followed by primary and secondary fetal myogenesis into hypaxial muscle with development of a satellite cell pool and evidence for innervation in vitro. Skeletal muscle organoids faithfully recapitulate all steps of embryonic myogenesis in 3D Tissue engineered muscle exhibits organotypic maturation and function, advanced by thyroid hormone. Regenerative competence was demonstrated in a cardiotoxin injury model with evidence of satellite cell activation as underlying mechanism. Collectively, we introduce a hypaxial muscle model with canonical properties of bona fide skeletal muscle in vivo to study muscle development, maturation, disease, and repair.

cell biology↗