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J Mol Biol ; 431(9): 1711-1728, 2019 04 19.
Artículo en Inglés | MEDLINE | ID: mdl-30878478

RESUMEN

Myotonic dystrophy type 1 is an autosomal-dominant inherited disorder caused by the expansion of CTG repeats in the 3' untranslated region of the DMPK gene. The RNAs bearing these expanded repeats have a range of toxic effects. Here we provide evidence from a Caenorhabditis elegans myotonic dystrophy type 1 model that the RNA interference (RNAi) machinery plays a key role in causing RNA toxicity and disease phenotypes. We show that the expanded repeats systematically affect a range of endogenous genes bearing short non-pathogenic repeats and that this mechanism is dependent on the small RNA pathway. Conversely, by perturbating the RNA interference machinery, we reversed the RNA toxicity effect and reduced the disease pathogenesis. Our results unveil a role for RNA repeats as templates (based on sequence homology) for moderate but constant gene silencing. Such a silencing effect affects the cell steady state over time, with diverse impacts depending on tissue, developmental stage, and the type of repeat. Importantly, such a mechanism may be common among repeats and similar in human cells with different expanded repeat diseases.


Asunto(s)
Envejecimiento/genética , Caenorhabditis elegans/genética , Distrofia Miotónica/genética , Interferencia de ARN , ARN Bicatenario/genética , Repeticiones de Trinucleótidos , Regiones no Traducidas 3' , Animales , Animales Modificados Genéticamente , Caenorhabditis elegans/crecimiento & desarrollo , Caenorhabditis elegans/metabolismo , Proteínas de Caenorhabditis elegans/genética , Proteínas de Caenorhabditis elegans/metabolismo , Modelos Animales de Enfermedad , Genes Reporteros , Proteínas Fluorescentes Verdes/genética , Proteínas Fluorescentes Verdes/metabolismo , Proteínas HSP70 de Choque Térmico/genética , Proteínas HSP70 de Choque Térmico/metabolismo , Proteínas de Choque Térmico/genética , Proteínas de Choque Térmico/metabolismo , Calor , Humanos , Distrofia Miotónica/metabolismo , Distrofia Miotónica/patología , Unión Proteica , ARN Bicatenario/metabolismo , ARN de Helminto/genética , ARN de Helminto/metabolismo
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