Why the study?
The nematode model Caenorhabditis elegans has frequently been used to evaluate the molecular and physiological consequences of human MYH7 mutations on muscle function.
The C. elegans model is inappropriate for studying MYH7 mutations when UNC-54 protein is expressed above endogenous levels, cautioning against its use in certain experimental designs.
C. elegans unc-54 model unsuitable for MYH7 expression studies; leaves open need for validated alternatives in myosin myopathy research.
Mutations in the human MYH7 gene, encoding a slow skeletal muscle/β-cardiac myosin heavy chain, cause different types of myopathies. The nematode model Caenorhabditis elegans has frequently been employed to study the molecular and physiological consequences of MYH7 mutations in muscle function by introducing mutations into the unc-54 gene, the worm MYH7 ortholog. We report here that the C. elegans model is not appropriate for such studies if they involve expression of the UNC-54 protein (wild-type or fused to green fluorescent protein) above endogenous levels.
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Gil‐Gálvez et al. (2020) studied this question.
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