Sautter NB, Smith TL. Hereditary hemorrhagic telangiectasia-related epistaxis: innovations in understanding and management. Int Forum Allergy Rhinol. 2012;2:422–31.
Lupa MD, Wise SK. Comprehensive management of hereditary hemorrhagic telangiectasia. Curr Opin Otolaryngol Head Neck Surg. 2017;25:64–8.
Al-Samkari H. Hereditary hemorrhagic telangiectasia: systemic therapies, guidelines, and an evolving standard of care. Blood. 2021;137:888–95.
Article CAS PubMed Google Scholar
Khanwalkar AR, Rathor A, Read AK, Paknezhad H, Ma Y, Hwang PH. Randomized, controlled, double-blinded clinical trial of effect of bevacizumab injection in management of epistaxis in hereditary hemorrhagic telangiectasia patients undergoing surgical cauterization. Int Forum Allergy Rhinol. 2022;12:1034–42.
Peterson AM, Lee JJ, Kallogjeri D, Schneider JS, Chakinala MM, Piccirillo JF. Efficacy of Timolol in a Novel Intranasal Thermosensitive Gel for Hereditary Hemorrhagic Telangiectasia-Associated Epistaxis: A Randomized Clinical Trial. JAMA Otolaryngol Head Neck Surg. 2020;146:1006–14.
Al-Samkari H, Kritharis A, Rodriguez-Lopez JM, Kuter DJ. Systemic bevacizumab for the treatment of chronic bleeding in hereditary haemorrhagic telangiectasia. J Intern Med. 2019;285:223–31.
Article CAS PubMed Google Scholar
Dupuis-Girod S, Pitiot V, Bergerot C, Fargeton A-E, Beaudoin M, Decullier E, et al. Efficacy of TIMOLOL nasal spray as a treatment for epistaxis in hereditary hemorrhagic telangiectasia. A double-blind, randomized, placebo-controlled trial. Sci Rep. 2019;9:11986.
Article PubMed PubMed Central Google Scholar
•• Hsu Y-P, Hsu C-W, Bai C-H, Cheng S-W, Chen C. Medical Treatment for Epistaxis in Hereditary Hemorrhagic Telangiectasia: A Meta-analysis. Otolaryngol Head Neck Surg. 2019;160:22–35. Comment: the previous meta-analysis of treatments in HHT patients.
McWilliams JP, Majumdar S, Kim GH, Lee J, Seals K, Tangchaiburana S, et al. North American study for the treatment of recurrent epistaxis with doxycycline: the NOSTRIL trial. J Thromb Haemost. 2022;20:1115–25.
Article CAS PubMed Google Scholar
•• Whitehead KJ, Sautter NB, McWilliams JP, Chakinala MM, Merlo CA, Johnson MH, et al. Effect of topical intranasal therapy on epistaxis frequency in patients with hereditary hemorrhagic telangiectasia: a randomized clinical trial. JAMA. 2016;316:943–51. Comment: comparisons of three commonly used topical treatments with placebo in HHT patients: bevacizumab, estriol, and tranexamic acid spray.
Article CAS PubMed Google Scholar
Lund VJ, Darby Y, Rimmer J, Amin M, Husain S. Nasal closure for severe hereditary haemorrhagic telangiectasia in 100 patients. The Lund modification of the Young’s procedure: a 22-year experience. Rhinology. 2017;55(2):135–41.
Article CAS PubMed Google Scholar
Harvey RJ, Kanagalingam J, Lund VJ. The impact of septodermoplasty and potassium-titanyl-phosphate (KTP) laser therapy in the treatment of hereditary hemorrhagic telangiectasia-related epistaxis. Am J Rhinol. 2008;22:182–7.
Faughnan ME, Mager JJ, Hetts SW, Palda VA, Lang-Robertson K, Buscarini E, et al. Second international guidelines for the diagnosis and management of hereditary hemorrhagic telangiectasia. Ann Intern Med. 2020;173:989–1001.
Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71.
Article PubMed PubMed Central Google Scholar
Shovlin CL. Hereditary haemorrhagic telangiectasia: Pathophysiology, diagnosis and treatment. Blood Rev. 2010;24(6):203–19.
Article CAS PubMed Google Scholar
Higgins J, Thomas J, Chandler J, Cumpston M, Li T, Page M, et al. Cochrane handbook for systematic reviews of interventions [Internet]. 2021. [accessed 1 May 2023]. Available from: https://training.cochrane.org/handbook
Hoag JB, Terry P, Mitchell S, Reh D, Merlo CA. An epistaxis severity score for hereditary hemorrhagic telangiectasia. Laryngoscope. 2010;120:838–43.
Thorlund K, Walter SD, Johnston BC, Furukawa TA, Guyatt GH. Pooling health-related quality of life outcomes in meta-analysis-a tutorial and review of methods for enhancing interpretability. Res Synth Methods. 2011;2:188–203.
Brignardello-Petersen R, Murad MH, Walter SD, McLeod S, Carrasco-Labra A, Rochwerg B, et al. GRADE approach to rate the certainty from a network meta-analysis: avoiding spurious judgments of imprecision in sparse networks. J Clin Epidemiol. 2019;105:60–7.
Sterne JAC, Savović J, Page MJ, Elbers RG, Blencowe NS, Boutron I, et al. RoB 2: a revised tool for assessing risk of bias in randomised trials. BMJ. 2019;366:l4898.
Brignardello-Petersen R, Florez ID, Izcovich A, Santesso N, Hazlewood G, Alhazanni W, et al. GRADE approach to drawing conclusions from a network meta-analysis using a minimally contextualised framework. BMJ. 2020;371:m3900.
Santesso N, Glenton C, Dahm P, Garner P, Akl EA, Alper B, et al. GRADE guidelines 26: informative statements to communicate the findings of systematic reviews of interventions. J Clin Epidemiol. 2020;119:126–35.
Pyne JM, Murray S, Kelly BC, Song JS, Rosvall BR, Côté DWJ. Surgiflo® hemostatic matrix versus NasoPore® nasal packing following postassium titanyl phosphate laser surgery for hereditary hemorrhagic telangiectasia: A randomized controlled trial. Laryngoscope Investig Otolaryngol. 2023;8:328–34.
Article PubMed PubMed Central Google Scholar
Thompson KP, Sykes J, Chandakkar P, Marambaud P, Vozoris NT, Marchuk DA, et al. Randomized, double-blind, placebo-controlled, crossover trial of oral doxycycline for epistaxis in hereditary hemorrhagic telangiectasia. Orphanet J Rare Dis. 2022;17:405.
Article CAS PubMed PubMed Central Google Scholar
Andorfer KEC, Zeman F, Koller M, Zeller J, Fischer R, Seebauer CT, et al. TIMolol nasal spray as a treatment for epistaxis in hereditary hemorrhagic telangiectasia (TIM-HHT)—a prospective, randomized, double-blind, controlled, cross-over trial. Pharmaceutics. 2022;14(11):2335.
Article CAS PubMed PubMed Central Google Scholar
Dupuis-Girod S, Fargeton A-E, Grobost V, Rivière S, Beaudoin M, Decullier E, et al. Efficacy and safety of a 0.1% tacrolimus nasal ointment as a treatment for epistaxis in hereditary hemorrhagic telangiectasia: a double-blind, randomized, placebo-controlled, multicenter trial. J Clin Med. 2020;9(5):1262.
Article CAS PubMed PubMed Central Google Scholar
• Mei-Zahav M, Gendler Y, Bruckheimer E, Prais D, Birk E, Watad M, et al. Topical propranolol improves epistaxis control in hereditary hemorrhagic telangiectasia (hht): a randomized double-blind placebo-controlled trial. J Clin Med. 2020;9(10):3130. Comment: the trial demonstrated effect of propranolol which is the most effective treatment in this meta-analysis for ESS reduction.
Article CAS PubMed PubMed Central Google Scholar
Dupuis-Girod S, Ambrun A, Decullier E, Fargeton A-E, Roux A, Bréant V, et al. Effect of bevacizumab nasal spray on epistaxis duration in hereditary hemorrhagic telangectasia: a randomized clinical trial. JAMA. 2016;316:934–42.
Article CAS PubMed Google Scholar
Riss D, Burian M, Wolf A, Kranebitter V, Kaider A, Arnoldner C. Intranasal submucosal bevacizumab for epistaxis in hereditary hemorrhagic telangiectasia: a double-blind, randomized, placebo-controlled trial. Head Neck. 2015;37(6):783–7.
Boyer H, Fernandes P, Le C, Yueh B. Prospective randomized trial of sclerotherapy vs standard treatment for epistaxis due to hereditary hemorrhagic telangiectasia. Int Forum Allergy Rhinol. 2015;5:435–40.
Article PubMed PubMed Central Google Scholar
Geisthoff UW, Seyfert UT, Kübler M, Bieg B, Plinkert PK, König J. Treatment of epistaxis in hereditary hemorrhagic telangiectasia with tranexamic acid - a double-blind placebo-controlled cross-over phase IIIB study. Thromb Res. 2014;134(3):565–71.
Article CAS PubMed Google Scholar
Gaillard S, Dupuis-Girod S, Boutitie F, Rivière S, Morinière S, Hatron P-Y, et al. Tranexamic acid for epistaxis in hereditary hemorrhagic telangiectasia patients: a European cross-over controlled trial in a rare disease. J Thromb Haemost. 2014;12(9):1494–502.
Article CAS PubMed Google Scholar
Dupuis-Girod S, Ambrun A, Decullier E, Samson G, Roux A, Fargeton A-E, et al. ELLIPSE Study: a Phase 1 study evaluating the tolerance of bevacizumab nasal spray in the treatment of epistaxis in hereditary hemorrhagic telangiectasia. MAbs. 2014;6(3):794–9.
Luk L, Mace JC, Bhandarkar ND, Sautter NB. Comparison of electrosurgical plasma coagulation and potassium-titanyl-phosphate laser photocoagulation for treatment of hereditary hemorrhagic telangiectasia-related epistaxis. Int Forum Allergy Rhinol. 2014;4:640–5.
Yaniv E, Preis M, Hadar T, Shvero J, Haddad M. Antiestrogen therapy for hereditary hemorrhagic telangiectasia: a double-blind placebo-controlled clinical trial. Laryngoscope. 2009;119:284–8.
Article CAS PubMed Google Scholar
Bergler W, Sadick H, Gotte K, Riedel F, Hörmann K. Topical estrogens combined with argon plasma coagulation in the management of epistaxis in hereditary hemorrhagic telangiectasia. Ann Otol Rhinol Laryngol. 2002;111:222–8.
Vase P, Lorentzen M. Histological findings following oestrogen treatment of hereditary haemorrhagic telangiectasia. A controlled double-blind investigation. J Laryngol Otol. 1983;97:427–9.
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