Metamorphism in TDP-43 prion-like domain determines chaperone recognition

The RNA binding protein TDP-43 forms cytoplasmic inclusions via its C-terminal prion-like domain in several neurodegenerative diseases. Aberrant TDP-43 aggregation arises upon phase de-mixing and transitions from liquid to solid states, following still unknown structural conversions which are primed...

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Detalles Bibliográficos
Autores: Carrasco, Jaime, Antón, Rosa, Valbuena Jiménez, Alejandro, Pantoja-Uceda, David, Mukhi, Mayur, Hervás, Rubén, Laurents, Douglas V., Gasset, María, Oroz, Javier
Tipo de recurso: artículo
Fecha de publicación:2023
País:España
Institución:Universidad Autónoma de Madrid
Repositorio:Biblos-e Archivo. Repositorio Institucional de la UAM
Idioma:inglés
OAI Identifier:oai:repositorio.uam.es:10486/707019
Acceso en línea:http://hdl.handle.net/10486/707019
https://dx.doi.org/10.1038/s41467-023-36023-z
Access Level:acceso abierto
Palabra clave:TDP-43
RNA
HSP70
HSP90
Biología y Biomedicina / Biología
Descripción
Sumario:The RNA binding protein TDP-43 forms cytoplasmic inclusions via its C-terminal prion-like domain in several neurodegenerative diseases. Aberrant TDP-43 aggregation arises upon phase de-mixing and transitions from liquid to solid states, following still unknown structural conversions which are primed by oxidative stress and chaperone inhibition. Despite the well-established protective roles for molecular chaperones against protein aggregation pathologies, knowledge on the determinants of chaperone recognition in disease-related prions is scarce. Here we show that chaperones and cochaperones primarily recognize the structured elements in TDP-43´s prionlike domain. Significantly, while HSP70 and HSP90 chaperones promote TDP43 phase separation, co-chaperones from the three classes of the large human HSP40 family (namely DNAJA2, DNAJB1, DNAJB4 and DNAJC7) show strikingly different effects on TDP-43 de-mixing. Dismantling of the second helical element in TDP-43 prion-like domain by methionine sulfoxidation impacts phase separation and amyloid formation, abrogates chaperone recognition and alters phosphorylation by casein kinase-1δ. Our results show that metamorphism in the post-translationally modified TDP-43 prion-like domain encodes determinants that command mechanisms with major relevance in disease