Cargando…

A pilot study of circulating microRNAs as potential biomarkers of Fabry disease

Patients suffering from Fabry disease (FD), a lysosomal storage disorder, show a broad range of symptoms and the diagnosis followed by the therapeutic decision remains a great challenge. The biomarkers available today have not proven to be useful for predicting the evolution of the disease and for a...

Descripción completa

Detalles Bibliográficos
Autores principales: Cammarata, Giuseppe, Scalia, Simone, Colomba, Paolo, Zizzo, Carmela, Pisani, Antonio, Riccio, Eleonora, Montalbano, Michaela, Alessandro, Riccardo, Giordano, Antonello, Duro, Giovanni
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Impact Journals LLC 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6007950/
https://www.ncbi.nlm.nih.gov/pubmed/29937989
http://dx.doi.org/10.18632/oncotarget.25542
_version_ 1783333119269535744
author Cammarata, Giuseppe
Scalia, Simone
Colomba, Paolo
Zizzo, Carmela
Pisani, Antonio
Riccio, Eleonora
Montalbano, Michaela
Alessandro, Riccardo
Giordano, Antonello
Duro, Giovanni
author_facet Cammarata, Giuseppe
Scalia, Simone
Colomba, Paolo
Zizzo, Carmela
Pisani, Antonio
Riccio, Eleonora
Montalbano, Michaela
Alessandro, Riccardo
Giordano, Antonello
Duro, Giovanni
author_sort Cammarata, Giuseppe
collection PubMed
description Patients suffering from Fabry disease (FD), a lysosomal storage disorder, show a broad range of symptoms and the diagnosis followed by the therapeutic decision remains a great challenge. The biomarkers available today have not proven to be useful for predicting the evolution of the disease and for assessing response to therapy in many patients. Here, we used high-throughput microRNA profiling methodology to identify a specific circulating microRNA profile in FD patients. We discovered a pattern of 10 microRNAs able to identify FD patients when compared to healthy controls. Notably, two of these: the miR199a-5p and the miR-126-3p are able to discriminate FDs from the control subjects with left ventricular hypertrophy, a frequent but non-specific FD symptom. These same microRNAs are also sensitive to enzyme replacement therapy showing variation in the subjects under treatment. Furthermore, two other microRNAs of the profile, the miR-423-5p and the miR-451a, seem useful to highlight cardiac involvement in FD patients. A literature and database search revealed that miR-199a-5p, miR-126-3p, miR-423-5p and miR-451a are known to be linked to pathological states that occur during the FD development. In particular, miR-199a-5p, and miR-126-3p are involved in endothelial dysfunction and miR-423-5p and miR-451a in myocardial remodeling. In conclusion, in this study we identified a common plasma microRNA profile in FD patients, useful not only for the correct classification of Fabry patients regardless of sex and age, but also to evaluate the response to therapy. Furthermore, our observations suggest that some microRNAs of this profile demonstrate prognostic qualities.
format Online
Article
Text
id pubmed-6007950
institution National Center for Biotechnology Information
language English
publishDate 2018
publisher Impact Journals LLC
record_format MEDLINE/PubMed
spelling pubmed-60079502018-06-22 A pilot study of circulating microRNAs as potential biomarkers of Fabry disease Cammarata, Giuseppe Scalia, Simone Colomba, Paolo Zizzo, Carmela Pisani, Antonio Riccio, Eleonora Montalbano, Michaela Alessandro, Riccardo Giordano, Antonello Duro, Giovanni Oncotarget Research Paper: Pathology Patients suffering from Fabry disease (FD), a lysosomal storage disorder, show a broad range of symptoms and the diagnosis followed by the therapeutic decision remains a great challenge. The biomarkers available today have not proven to be useful for predicting the evolution of the disease and for assessing response to therapy in many patients. Here, we used high-throughput microRNA profiling methodology to identify a specific circulating microRNA profile in FD patients. We discovered a pattern of 10 microRNAs able to identify FD patients when compared to healthy controls. Notably, two of these: the miR199a-5p and the miR-126-3p are able to discriminate FDs from the control subjects with left ventricular hypertrophy, a frequent but non-specific FD symptom. These same microRNAs are also sensitive to enzyme replacement therapy showing variation in the subjects under treatment. Furthermore, two other microRNAs of the profile, the miR-423-5p and the miR-451a, seem useful to highlight cardiac involvement in FD patients. A literature and database search revealed that miR-199a-5p, miR-126-3p, miR-423-5p and miR-451a are known to be linked to pathological states that occur during the FD development. In particular, miR-199a-5p, and miR-126-3p are involved in endothelial dysfunction and miR-423-5p and miR-451a in myocardial remodeling. In conclusion, in this study we identified a common plasma microRNA profile in FD patients, useful not only for the correct classification of Fabry patients regardless of sex and age, but also to evaluate the response to therapy. Furthermore, our observations suggest that some microRNAs of this profile demonstrate prognostic qualities. Impact Journals LLC 2018-06-08 /pmc/articles/PMC6007950/ /pubmed/29937989 http://dx.doi.org/10.18632/oncotarget.25542 Text en Copyright: © 2018 Cammarata et al. http://creativecommons.org/licenses/by/3.0/ This article is distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0/) (CC-BY), which permits unrestricted use and redistribution provided that the original author and source are credited.
spellingShingle Research Paper: Pathology
Cammarata, Giuseppe
Scalia, Simone
Colomba, Paolo
Zizzo, Carmela
Pisani, Antonio
Riccio, Eleonora
Montalbano, Michaela
Alessandro, Riccardo
Giordano, Antonello
Duro, Giovanni
A pilot study of circulating microRNAs as potential biomarkers of Fabry disease
title A pilot study of circulating microRNAs as potential biomarkers of Fabry disease
title_full A pilot study of circulating microRNAs as potential biomarkers of Fabry disease
title_fullStr A pilot study of circulating microRNAs as potential biomarkers of Fabry disease
title_full_unstemmed A pilot study of circulating microRNAs as potential biomarkers of Fabry disease
title_short A pilot study of circulating microRNAs as potential biomarkers of Fabry disease
title_sort pilot study of circulating micrornas as potential biomarkers of fabry disease
topic Research Paper: Pathology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6007950/
https://www.ncbi.nlm.nih.gov/pubmed/29937989
http://dx.doi.org/10.18632/oncotarget.25542
work_keys_str_mv AT cammaratagiuseppe apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT scaliasimone apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT colombapaolo apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT zizzocarmela apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT pisaniantonio apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT riccioeleonora apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT montalbanomichaela apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT alessandroriccardo apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT giordanoantonello apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT durogiovanni apilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT cammaratagiuseppe pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT scaliasimone pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT colombapaolo pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT zizzocarmela pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT pisaniantonio pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT riccioeleonora pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT montalbanomichaela pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT alessandroriccardo pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT giordanoantonello pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease
AT durogiovanni pilotstudyofcirculatingmicrornasaspotentialbiomarkersoffabrydisease