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Título

Review of Pharmacological Strategies with Repurposed Drugs for Hereditary Hemorrhagic Telangiectasia Related Bleeding

AutorAlbiñana, Virginia CSIC; Cuesta, Ángel M. CSIC ORCID ; Rojas-P, Isabel de CSIC; Gallardo-Vara, Eunate CSIC ORCID; Recio-Poveda, Lucía ; Bernabéu, Carmelo CSIC ORCID ; Botella, Luisa María CSIC ORCID
Palabras claveHHT
ALK1
Endoglin
Raloxifene
Bazedoxifene
Tranexamic acid
Propranolol
FK506
Etamsylate
N-acetylcysteine
Fecha de publicación2020
EditorMultidisciplinary Digital Publishing Institute
CitaciónJournal of Clinical Medicine 9(6): 1766 (2020)
ResumenThe diagnosis of hereditary hemorrhagic telangiectasia (HHT) is based on the Curaçao criteria: epistaxis, telangiectases, arteriovenous malformations in internal organs, and family history. Genetically speaking, more than 90% of HHT patients show mutations in ENG or ACVRL1/ALK1 genes, both belonging to the TGF-β/BMP9 signaling pathway. Despite clear knowledge of the symptoms and genes of the disease, we still lack a definite cure for HHT, having just palliative measures and pharmacological trials. Among the former, two strategies are: intervention at “ground zero” to minimize by iron and blood transfusions in order to counteract anemia. Among the later, along the last 15 years, three different strategies have been tested: (1) To favor coagulation with antifibrinolytic agents (tranexamic acid); (2) to increase transcription of ENG and ALK1 with specific estrogen-receptor modulators (bazedoxifene or raloxifene), antioxidants (N-acetylcysteine, resveratrol), or immunosuppressants (tacrolimus); and (3) to impair the abnormal angiogenic process with antibodies (bevacizumab) or blocking drugs like etamsylate, and propranolol. This manuscript reviews the main strategies and sums up the clinical trials developed with drugs alleviating HHT.
Descripción© 2020 by the authors.
Versión del editorhttps://doi.org/10.3390/jcm9061766
URIhttp://hdl.handle.net/10261/215680
DOI10.3390/jcm9061766
E-ISSN2077-0383
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