Tranexamic acid for epistaxis in hereditary hemorrhagic telangiectasia patients: a European cross‐over controlled trial in a rare disease

Gaillard, Ségolène; Dupuis‐Girod, Sophie; Boutitie, Florent; Rivière, Sophie; Morinière, S.; Hatron, Pierre‐Yves; Manfredi, Guido; Kaminsky, P.; Capitaine, Anne-Gaëlle; Roy, Pascal; Gueyffier, François; Plauchu, Henri

doi:10.1111/jth.12654

Cited by 98 publications

(94 citation statements)

References 19 publications

(25 reference statements)

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“…26 Tranexamic acid has been shown to be modestly effective in reducing epistaxis frequency and severity in patients with HHT in randomized controlled trials. 8,33 Thermal and chemical coagulations are effective but less favored because of higher rates of complications, such as scarring and septal perforations. Endoscopic application of potassium titanyl phosphate or neodymium-doped yttrium aluminium garnet laser has been shown to be effective but repeated applications are necessary to maintain benefit.…”

Section: Epistaxismentioning

confidence: 99%

Cerebrovascular Manifestations of Hereditary Hemorrhagic Telangiectasia

Brinjikji

Iyer

Sorenson

et al. 2015

Stroke

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Section: Epistaxismentioning

confidence: 99%

Cerebrovascular Manifestations of Hereditary Hemorrhagic Telangiectasia

Brinjikji

Iyer

Sorenson

et al. 2015

Stroke

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“…Nosebleeds often occur daily, and can be associated with acute hemodynamic disturbances and reduced quality of life . Treatments include surgically based therapies such as cauterization, laser photocoagulation, septal dermoplasty, and Young's procedure, and medical therapies such as antioestrogens, tranexamic acid, and bevacizumab (Avastin) . Therapeutics are generally graded according to the severity of epistaxis, and many patients require more than one modality.…”

Section: Introductionmentioning

confidence: 99%

Can Iron Treatments Aggravate Epistaxis in Some Patients With Hereditary Hemorrhagic Telangiectasia?

et al. 2016

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Objectives/HypothesisTo examine whether there is a rationale for iron treatments precipitating nosebleeds (epistaxis) in a subgroup of patients with hereditary hemorrhagic telangiectasia (HHT).Study DesignSurvey evaluation of HHT patients, and a randomized control trial in healthy volunteers.MethodsNosebleed severity in response to iron treatments and standard investigations were evaluated by unbiased surveys in patients with HHT. Serial blood samples from a randomized controlled trial of 18 healthy volunteers were used to examine responses to a single iron tablet (ferrous sulfate, 200 mg).ResultsIron tablet users were more likely to have daily nosebleeds than non–iron‐users as adults, but there was no difference in the proportions reporting childhood or trauma‐induced nosebleeds. Although iron and blood transfusions were commonly reported to improve nosebleeds, 35 of 732 (4.8%) iron tablet users, in addition to 17 of 261 (6.5%) iron infusion users, reported that their nosebleeds were exacerbated by the respective treatments. These rates were significantly higher than those reported for control investigations. Serum iron rose sharply in four of the volunteers ingesting ferrous sulfate (by 19.3–33.1 μmol/L in 2 hours), but not in 12 dietary controls (2‐hour iron increment ranged from −2.2 to +5.0 μmol/L). High iron absorbers demonstrated greater increments in serum ferritin at 48 hours, but transient rises in circulating endothelial cells, an accepted marker of endothelial damage.ConclusionsIron supplementation is essential to treat or prevent iron deficiency, particularly in patients with pathological hemorrhagic iron losses. However, in a small subgroup of individuals, rapid changes in serum iron may provoke endothelial changes and hemorrhage.Level of Evidence4. Laryngoscope, 126:2468–2474, 2016

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“…Drugs such as tranexamic acid [117][118][119] and aminocaproic acid [120] show some efficacy in reducing epistaxis; however efficacy comes with inherent risk of thromboembolism [121]. Estrogenic and antiestrogenic therapies also demonstrate some efficacy [122][123][124], but treatments are not without side effects.…”

Section: Medical Therapies For Hht: Current Practice and Future Perspmentioning

confidence: 97%

Genetic and Molecular Basis for Hereditary Hemorrhagic Telangiectasia

Roman

Finegold

2014

Curr Genet Med Rep

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Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant disorder that predisposes patients to develop direct connections between arteries and veins, or arteriovenous malformations (AVMs). Although the genes responsible for the majority of HHT cases have been known for nearly 20 years, molecular and cellular mechanisms underlying pathogenesis are poorly understood, and the genetic and/or environmental factors that confer variability to age of onset and expressivity of HHT remain unknown. Herein, we review the genetics and genotype/ phenotype correlations associated with HHT and summarize data from animal and cell culture models that lend insight into disease mechanism. At present, therapies available to HHT patients for treatment of visceral AVMs are primarily surgical, although antiangiogenic agents show some efficacy in treatment of telangiectasias, epistaxis, and liver AVMs. In light of new mechanistic data on disease pathogenesis, we consider possible approaches for development of more targeted therapeutics for HHT patients.

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Tranexamic acid for epistaxis in hereditary hemorrhagic telangiectasia patients: a European cross‐over controlled trial in a rare disease

Cited by 98 publications

References 19 publications

Cerebrovascular Manifestations of Hereditary Hemorrhagic Telangiectasia

Cerebrovascular Manifestations of Hereditary Hemorrhagic Telangiectasia

Can Iron Treatments Aggravate Epistaxis in Some Patients With Hereditary Hemorrhagic Telangiectasia?

Genetic and Molecular Basis for Hereditary Hemorrhagic Telangiectasia

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