ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERY

b-thalassemia is a hereditary disorder caused by defective production of b-globin chains of hemoglobin (Hb) that leads to an increased a/b globins ratio with subsequent free a-globins. Alpha globin excess causes oxidative stress, RBCs membrane damage, premature death of late-stage erythroid precurso...

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Main Authors: Irene Motta, Valentina Brancaleoni, Isabella Nava, Paola Delbini, Lorena Duca
Format: Article
Language:English
Published: PAGEPress Publications 2020-10-01
Series:Mediterranean Journal of Hematology and Infectious Diseases
Online Access:http://www.mjhid.org/index.php/mjhid/article/view/4358
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author Irene Motta
Valentina Brancaleoni
Isabella Nava
Paola Delbini
Lorena Duca
author_facet Irene Motta
Valentina Brancaleoni
Isabella Nava
Paola Delbini
Lorena Duca
author_sort Irene Motta
collection DOAJ
description b-thalassemia is a hereditary disorder caused by defective production of b-globin chains of hemoglobin (Hb) that leads to an increased a/b globins ratio with subsequent free a-globins. Alpha globin excess causes oxidative stress, RBCs membrane damage, premature death of late-stage erythroid precursors, resulting in ineffective erythropoiesis. The transforming growth factor b (TGF-b) superfamily signaling acts on biological processes, such as cell quiescence, apoptosis, proliferation, differentiation, and migration, and also plays an important role in the regulation of hematopoiesis. This pathway can lose its physiologic regulation in pathologic conditions, leading to anemia and ineffective erythropoiesis. Activin receptor ligand trap molecules such as Sotatercept and Luspatercept downregulate the TGF-b pathway by inhibiting the Smad2/3 cascade, thus alleviating anemia in patients with b-thalassemia and myelodysplastic syndromes. In this review, we describe in extenso the TGF-b pathway, as well as the molecular and biological basis of activin receptors ligand traps, focusing on their role in various b-thalassemia experimental models. The most recent results from clinical trials on sotatercept and luspatercept will also be reviewed.
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issn 2035-3006
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publishDate 2020-10-01
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series Mediterranean Journal of Hematology and Infectious Diseases
spelling doaj-art-1ee4fa2a1f674da287972c0ce14444ab2025-08-20T02:50:42ZengPAGEPress PublicationsMediterranean Journal of Hematology and Infectious Diseases2035-30062020-10-0112110.4084/mjhid.2020.075ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERYIrene MottaValentina BrancaleoniIsabella NavaPaola DelbiniLorena Ducab-thalassemia is a hereditary disorder caused by defective production of b-globin chains of hemoglobin (Hb) that leads to an increased a/b globins ratio with subsequent free a-globins. Alpha globin excess causes oxidative stress, RBCs membrane damage, premature death of late-stage erythroid precursors, resulting in ineffective erythropoiesis. The transforming growth factor b (TGF-b) superfamily signaling acts on biological processes, such as cell quiescence, apoptosis, proliferation, differentiation, and migration, and also plays an important role in the regulation of hematopoiesis. This pathway can lose its physiologic regulation in pathologic conditions, leading to anemia and ineffective erythropoiesis. Activin receptor ligand trap molecules such as Sotatercept and Luspatercept downregulate the TGF-b pathway by inhibiting the Smad2/3 cascade, thus alleviating anemia in patients with b-thalassemia and myelodysplastic syndromes. In this review, we describe in extenso the TGF-b pathway, as well as the molecular and biological basis of activin receptors ligand traps, focusing on their role in various b-thalassemia experimental models. The most recent results from clinical trials on sotatercept and luspatercept will also be reviewed.http://www.mjhid.org/index.php/mjhid/article/view/4358
spellingShingle Irene Motta
Valentina Brancaleoni
Isabella Nava
Paola Delbini
Lorena Duca
ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERY
Mediterranean Journal of Hematology and Infectious Diseases
title ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERY
title_full ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERY
title_fullStr ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERY
title_full_unstemmed ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERY
title_short ACTIVIN RECEPTOR LIGAND FOR THR TREATMENT OF BETA-THALASSEMIA: a SERENDIPITOUS DISCOVERY
title_sort activin receptor ligand for thr treatment of beta thalassemia a serendipitous discovery
url http://www.mjhid.org/index.php/mjhid/article/view/4358
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AT paoladelbini activinreceptorligandforthrtreatmentofbetathalassemiaaserendipitousdiscovery
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