Hereditary Hemorrhagic Telangiectasia—Current Therapies and Future Prospects

Hereditary hemorrhagic telangiectasia (HHT), also known as Osler–Weber–Rendu disease, is an autosomal dominant vascular disorder caused most commonly by pathogenic variants in the ENG and ACVRL1/ALK1 genes. It is characterized by mucocutaneous telangiectasias and arteriovenous malformations (AVMs) in various organs, leading to recurrent epistaxis, gastrointestinal bleeding, and iron deficiency anemia. Diagnosis relies on the Curaçao Criteria, which include recurrent nosebleeds, characteristic telangiectasias, visceral AVMs, and family history. This review aims to present current therapeutic approaches and emerging treatment strategies for HHT. Traditional surgical and laser-based methods are increasingly complemented or replaced by targeted pharmacological interventions. Antiangiogenic agents such as bevacizumab and thalidomide have demonstrated efficacy in reducing bleeding frequency and transfusion requirements, although adverse effects may limit long-term use. Novel therapies under investigation target molecular pathways involved in vascular remodeling, including tyrosine kinase inhibitors (sorafenib, nintedanib), anti-ANGPT2 antibodies, and modulators of BMP9/ALK1 signaling (tacrolimus, sirolimus). Preclinical and early clinical studies suggest that these agents may provide disease-modifying benefits. Continued research should focus on optimizing treatment efficacy, reducing toxicity, and developing individualized therapeutic regimens based on genetic and clinical characteristics.

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Publication Details

Journal
Journal of Vascular Diseases
Published
2025-11-11
DOI
https://doi.org/10.3390/jvd4040043
Primary Topic
Vascular Anomalies and Treatments
Type
article
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article

Hereditary Hemorrhagic Telangiectasia—Current Therapies and Future Prospects

Milena Krawczyk, Dominika Żyła, Katarzyna Zych, Julia Nowakowska
Journal of Vascular Diseases
Vascular Anomalies and Treatments
article

Hereditary Hemorrhagic Telangiectasia—Current Therapies and Future Prospects

Milena Krawczyk, Dominika Żyła, Katarzyna Zych, Julia Nowakowska
article en

Abstract

Hereditary hemorrhagic telangiectasia (HHT), also known as Osler–Weber–Rendu disease, is an autosomal dominant vascular disorder caused most commonly by pathogenic variants in the ENG and ACVRL1/ALK1 genes. It is characterized by mucocutaneous telangiectasias and arteriovenous malformations (AVMs) in various organs, leading to recurrent epistaxis, gastrointestinal bleeding, and iron deficiency anemia. Diagnosis relies on the Curaçao Criteria, which include recurrent nosebleeds, characteristic telangiectasias, visceral AVMs, and family history. This review aims to present current therapeutic approaches and emerging treatment strategies for HHT. Traditional surgical and laser-based methods are increasingly complemented or replaced by targeted pharmacological interventions. Antiangiogenic agents such as bevacizumab and thalidomide have demonstrated efficacy in reducing bleeding frequency and transfusion requirements, although adverse effects may limit long-term use. Novel therapies under investigation target molecular pathways involved in vascular remodeling, including tyrosine kinase inhibitors (sorafenib, nintedanib), anti-ANGPT2 antibodies, and modulators of BMP9/ALK1 signaling (tacrolimus, sirolimus). Preclinical and early clinical studies suggest that these agents may provide disease-modifying benefits. Continued research should focus on optimizing treatment efficacy, reducing toxicity, and developing individualized therapeutic regimens based on genetic and clinical characteristics.

Journal of Vascular DiseasesVol. 4(4)
Jan Kochanowski University (PL), Warsaw University of Life Sciences (PL), Franciszek Górski Institute of Plant Physiology (PL), Oldham Council (GB)
Openalex Percentile: Top 53%
Vascular Anomalies and Treatments
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Hereditary Hemorrhagic Telangiectasia—Current Therapies and Future Prospects — Milena Krawczyk, Dominika Żyła, et al. · Journal of Vascular Diseases (2025) | TGRS Research Map | TGRS