Protein misfolding diseases: Prospects of pharmacological treatment

Protein misfolding has been linked to numerous inherited diseases. Loss- and gain-of-function mutations (common features of genetic diseases) may cause the destabilization of proteins, leading to alterations in their properties and/or cellular location, resulting in their incorrect functioning. Misf...

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Detalles Bibliográficos
Autores: Gámez, Alejandra, Yuste-Checa, Patricia, Brasil, Sandra, Briso-Montiano, Álvaro, Desviat, Lourdes R., Ugarte, Magdalena, Pérez-Cerdá, Celia, Pérez, Belén
Tipo de recurso: artículo
Estado:Versión aceptada para publicación
Fecha de publicación:2018
País:España
Institución:Consejo Superior de Investigaciones Científicas (CSIC)
Repositorio:DIGITAL.CSIC. Repositorio Institucional del CSIC
OAI Identifier:oai:digital.csic.es:10261/342517
Acceso en línea:http://hdl.handle.net/10261/342517
Access Level:acceso abierto
Palabra clave:Conformational diseases
Congenital disorders of glycosylation
Inborn errors of metabolism
Misfolding diseases
Pharmacological chaperones
Protein folding
Proteostasis regulators
Descripción
Sumario:Protein misfolding has been linked to numerous inherited diseases. Loss- and gain-of-function mutations (common features of genetic diseases) may cause the destabilization of proteins, leading to alterations in their properties and/or cellular location, resulting in their incorrect functioning. Misfolded proteins can, however, be rescued via the use of proteostasis regulators and/or pharmacological chaperones, suggesting that treatments with small molecules might be developed for a range of genetic diseases. This work describes the potential of these small molecules in this respect, including for the treatment of congenital disorder of glycosylation (CDG) due to phosphomannomutase 2 deficiency (PMM2-CDG).