Hereditary Amyloidosis: Insights Into a Fibrinogen A Variant Protein
; Gisonno, Romina Antonela
; Abba, Martín Carlos
; Santana, Marianela
; Rosu, Silvana Antonia
; Nucifora, Elsa Mercedes ; Aguirre, Maria Adela ; Giordani, María C. ; Tricerri, Maria Alejandra
; Ramella, Nahuel 
Otros responsables
Fecha
07/2024
Editorial
Wiley-liss, div John Wiley & Sons Inc.
Revista
Proteins: Structure, Function And Genetics
Título del libro
Nombre del evento
Fecha del evento
Institución organizadora
ISSN
0887-3585
eISSN
ISBN
Idioma
Inglés
Tipo del evento
Resumen
Amyloidosis are a group of diseases in which soluble proteins aggregate and deposit in fibrillar conformation extracellularly intissues. The effectiveness of therapeutic strategies depends on the specific protein involved, being crucial to accurately determineits nature. Moreover, following the diagnosis, the search for the mutation within relatives allows the clinical advice. Here wereport the precise diagnosis and explored the possible reasons of the structural pathogenicity for a renal amyloidosis related toa fibrinogen Aα-chain variant. Whole-exome sequencing and GATK calling pipeline were leveraged to characterize the proteinvariant present in a patient with kidney failure. Bioinformatics strategies were applied to suggest potential explanations of thevariants aggregation. Our pipeline allowed the identification of a single-point variant of fibrinogen Aα-chain, which opened thepossibility of curative transplantation. In silico structural analysis suggested that the pathogenicity of the variant may be attrib-uted to a heightened susceptibility to yield a peptide prone to deposit as an oligomer with a β-sheet structure. Exploiting the com-prehensive coverage of whole-genome sequencing, we managed to fill a vacant stage in the diagnosis of hereditary amyloidosisand to stimulate the advancement in biomedicine.
Colecciones
Aprobación
Revisión
Complementado por
Referenciado por
Licencia Creative Commons
Excepto donde se indique lo contrario, la licencia de este ítem se describe como by-nc-sa
