Table 1.

Key clinical trials of systemic antiangiogenic therapies for the management of HHT

DrugTrialTypenStatus or major findings
Intravenous bevacizumab     
 NCT04404881 Single-arm trial 33 Recruiting 
 Dupuis-Girod19  RCT 24 ↔ Transfusion and ↑Hgb, QOL 
 Al-Samkari20  Retrospective 238 ↓ESS, transfusion and ↑Hgb 
 Vazquez27  Retrospective 20 ↑Hgb and ↓transfusion 
 Al-Samkari28  Retrospective 13 ↓Transfusion, epistaxis and ↑Hgb 
 Guilhem29  Retrospective 46 Clinical improvement in 74% 
 Iyer30  Retrospective 34 ↓ESS, transfusion 
 Dupuis-Girod21  Single-arm trial 25 ↓Cardiac index, epistaxis, and ↑QOL 
Pazopanib     
 NCT03850964 RCT 70 Recruiting 
 Parambil24  Retrospective 13 ↓Transfusion, ESS and ↑Hgb 
 Faughnan31  Single-arm trial ↓Epistaxis and ↑Hgb, QOL 
Pomalidomide     
 Al-Samkari23  RCT 144 ↓ESS and ↑QOL 
Thalidomide     
 Invernizzi32  Single-arm trial 31 ↓ESS, transfusion and ↑Hgb 
Tacrolimus     
 NCT04646356 Single-arm trial 30 Active not recruiting 
 Alvarez-Hernandez26  Single-arm trial 11 ↓ESS and ↑Hgb 
Nintedanib     
 NCT04976036 RCT 48 Recruiting 
 NCT03954782 RCT 61 Completed 
VAD044     
 NCT05406362 RCT 80 Recruiting 
Sirolimus     
 NCT05269849 Single-arm trial 10 Recruiting 
DrugTrialTypenStatus or major findings
Intravenous bevacizumab     
 NCT04404881 Single-arm trial 33 Recruiting 
 Dupuis-Girod19  RCT 24 ↔ Transfusion and ↑Hgb, QOL 
 Al-Samkari20  Retrospective 238 ↓ESS, transfusion and ↑Hgb 
 Vazquez27  Retrospective 20 ↑Hgb and ↓transfusion 
 Al-Samkari28  Retrospective 13 ↓Transfusion, epistaxis and ↑Hgb 
 Guilhem29  Retrospective 46 Clinical improvement in 74% 
 Iyer30  Retrospective 34 ↓ESS, transfusion 
 Dupuis-Girod21  Single-arm trial 25 ↓Cardiac index, epistaxis, and ↑QOL 
Pazopanib     
 NCT03850964 RCT 70 Recruiting 
 Parambil24  Retrospective 13 ↓Transfusion, ESS and ↑Hgb 
 Faughnan31  Single-arm trial ↓Epistaxis and ↑Hgb, QOL 
Pomalidomide     
 Al-Samkari23  RCT 144 ↓ESS and ↑QOL 
Thalidomide     
 Invernizzi32  Single-arm trial 31 ↓ESS, transfusion and ↑Hgb 
Tacrolimus     
 NCT04646356 Single-arm trial 30 Active not recruiting 
 Alvarez-Hernandez26  Single-arm trial 11 ↓ESS and ↑Hgb 
Nintedanib     
 NCT04976036 RCT 48 Recruiting 
 NCT03954782 RCT 61 Completed 
VAD044     
 NCT05406362 RCT 80 Recruiting 
Sirolimus     
 NCT05269849 Single-arm trial 10 Recruiting 

ESS, epistaxis severity score; Hgb, hemoglobin, transfusion, red blood cell transfusion; QOL, quality of life; RCT, placebo-controlled randomized controlled trial.

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