ArticleJournal of neuromuscular diseases2025
Limited pre-clinical relevance of the heterozygous RYR1-I4895T/+ mouse model due to its mild phenotype.
Article in Journal of neuromuscular diseases, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
1 citing paper in PubMed.
- Molecular Insights into Central Core Disease: Proteomic Signatures and Potential Therapeutic Biomarkers in RYR1 I4895T Mice.International journal of molecular sciences · 2025Article
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Authors and funding
11 authors.
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Abstract
backgroundAlthough genetically-engineered mouse models are revolutionizing our understanding of numerous human diseases, some of them fail to reproduce or to mimic the human condition or even exhibit distinct disease features depending on the mouse genetic background, on the environment conditions, and/or on unknown parameters.
objectiveExperiments aimed at further characterizing the muscle defects associated with the I-T substitution at position 4898 of the human type 1 ryanodine receptor (RyR1) protein sequence, responsible for central core disease in affected patients, to use this model for therapeutic development. RyR1 is a cationic channel in the sarcoplasmic reticulum membrane that is responsible for the Ca
methodsWe used the corresponding I4895T mouse model, previously shown unviable in the homozygous form, and with heterozygous animals suffering from depressed RyR1-mediated Ca
resultsWe found no significant deficit in the heterozygous animals, from force and activity parameters at the whole organism level, to contraction of isolated muscles and Ca
conclusionsOur results prompt the need for caution when using this model, and point to its potential limited relevance for preclinical studies.
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