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Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs
β-thalassaemia is a rare genetic condition caused by mutations in the β-globin gene that result in severe iron-loading anaemia, maintained by a detrimental state of ineffective erythropoiesis (IE). The role of multiple mechanisms involved in the pathophysiology of the disease has been recently unrav...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2021
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8268821/ https://www.ncbi.nlm.nih.gov/pubmed/34281283 http://dx.doi.org/10.3390/ijms22137229 |
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author | Longo, Filomena Piolatto, Andrea Ferrero, Giovanni Battista Piga, Antonio |
author_facet | Longo, Filomena Piolatto, Andrea Ferrero, Giovanni Battista Piga, Antonio |
author_sort | Longo, Filomena |
collection | PubMed |
description | β-thalassaemia is a rare genetic condition caused by mutations in the β-globin gene that result in severe iron-loading anaemia, maintained by a detrimental state of ineffective erythropoiesis (IE). The role of multiple mechanisms involved in the pathophysiology of the disease has been recently unravelled. The unbalanced production of α-globin is a major source of oxidative stress and membrane damage in red blood cells (RBC). In addition, IE is tightly linked to iron metabolism dysregulation, and the relevance of new players of this pathway, i.e., hepcidin, erythroferrone, matriptase-2, among others, has emerged. Advances have been made in understanding the balance between proliferation and maturation of erythroid precursors and the role of specific factors in this process, such as members of the TGF-β superfamily, and their downstream effectors, or the transcription factor GATA1. The increasing understanding of IE allowed for the development of a broad set of potential therapeutic options beyond the current standard of care. Many candidates of disease-modifying drugs are currently under clinical investigation, targeting the regulation of iron metabolism, the production of foetal haemoglobin, the maturation process, or the energetic balance and membrane stability of RBC. Overall, they provide tools and evidence for multiple and synergistic approaches that are effectively moving clinical research in β-thalassaemia from bench to bedside. |
format | Online Article Text |
id | pubmed-8268821 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-82688212021-07-10 Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs Longo, Filomena Piolatto, Andrea Ferrero, Giovanni Battista Piga, Antonio Int J Mol Sci Review β-thalassaemia is a rare genetic condition caused by mutations in the β-globin gene that result in severe iron-loading anaemia, maintained by a detrimental state of ineffective erythropoiesis (IE). The role of multiple mechanisms involved in the pathophysiology of the disease has been recently unravelled. The unbalanced production of α-globin is a major source of oxidative stress and membrane damage in red blood cells (RBC). In addition, IE is tightly linked to iron metabolism dysregulation, and the relevance of new players of this pathway, i.e., hepcidin, erythroferrone, matriptase-2, among others, has emerged. Advances have been made in understanding the balance between proliferation and maturation of erythroid precursors and the role of specific factors in this process, such as members of the TGF-β superfamily, and their downstream effectors, or the transcription factor GATA1. The increasing understanding of IE allowed for the development of a broad set of potential therapeutic options beyond the current standard of care. Many candidates of disease-modifying drugs are currently under clinical investigation, targeting the regulation of iron metabolism, the production of foetal haemoglobin, the maturation process, or the energetic balance and membrane stability of RBC. Overall, they provide tools and evidence for multiple and synergistic approaches that are effectively moving clinical research in β-thalassaemia from bench to bedside. MDPI 2021-07-05 /pmc/articles/PMC8268821/ /pubmed/34281283 http://dx.doi.org/10.3390/ijms22137229 Text en © 2021 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Review Longo, Filomena Piolatto, Andrea Ferrero, Giovanni Battista Piga, Antonio Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs |
title | Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs |
title_full | Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs |
title_fullStr | Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs |
title_full_unstemmed | Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs |
title_short | Ineffective Erythropoiesis in β-Thalassaemia: Key Steps and Therapeutic Options by Drugs |
title_sort | ineffective erythropoiesis in β-thalassaemia: key steps and therapeutic options by drugs |
topic | Review |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8268821/ https://www.ncbi.nlm.nih.gov/pubmed/34281283 http://dx.doi.org/10.3390/ijms22137229 |
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