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Hereditary hemorrhagic telangiectasia (HHT) is characterized by extensive telangiectasias and arteriovenous malformations. The primary clinical manifestation is epistaxis that results in iron-deficiency anemia and reduced health-related quality of life. We conducted a randomized, placebo-controlled trial to evaluate the safety and efficacy of pomalidomide for the treatment of HHT. We randomly assigned patients, in a 2:1 ratio, to receive pomalidomide at a dose of 4 mg daily or matching placebo for 24 weeks. The primary outcome was the change from baseline through week 24 in the Epistaxis Severity Score (a validated bleeding score in HHT; range, 0 to 10, with higher scores indicating worse bleeding). A reduction of 0.71 points or more is considered clinically significant. A key secondary outcome was the HHT-specific quality-of-life score (range, 0 to 16, with higher scores indicating more limitations). The trial was closed to enrollment in June 2023 after a planned interim analysis met a prespecified threshold for efficacy. A total of 144 patients underwent randomization; 95 patients were assigned to receive pomalidomide and 49 to receive placebo. The baseline mean (±SD) Epistaxis Severity Score was 5.0±1.5, a finding consistent with moderate-to-severe epistaxis. At 24 weeks, the mean difference between the pomalidomide group and the placebo group in the change from baseline in the Epistaxis Severity Score was -0.94 points (95% confidence interval [CI], -1.57 to -0.31; P = 0.004). The mean difference in the changes in the HHT-specific quality-of-life score between the groups was -1.4 points (95% CI, -2.6 to -0.3). Adverse events that were more common in the pomalidomide group than in the placebo group included neutropenia, constipation, and rash. Among patients with HHT, pomalidomide treatment resulted in a significant, clinically relevant reduction in epistaxis severity. No unexpected safety signals were identified. (Funded by the National Heart, Lung, and Blood Institute; PATH-HHT Clinicaltrials.gov number, NCT03910244).

Objective: Epistaxis is often treated in the emergency department (ED) and is one of the causes of bleeding that can be successfully managed by physicians. This study aimed to investigate whether there is a relationship between recurrent epistaxis and simple blood parameters (the values of mean platelet volume (MPV) and red cell distribution width (RDW)). Method: This retrospective case-control study was designed at a tier-three ED. The patients were chosen from among those who applied to the ED for epistaxis between 1 January 2018 and 31 December 2018. Demographic data, seasonal application frequency, complete blood count results and clinical findings were retrospectively reviewed on the hospital information record system. The patients were categorized into three groups of recurrent epistaxis, non-recurrent epistaxis and healthy control groups. Results: 143 epistaxis patients in total including recurrent (n=73) and non-recurrent (n=70) patients were enrolled in this study. The healthy control group was composed of 77 patients. The MPV value was significantly higher in the recurrent epistaxis group than in the non-recurrent epistaxis group (8.91±1.16, 8.48±1.06, P = 0.006, respectively). In addition, the mean platelet and platecrit values were significantly higher in the non-recurrent epistaxis group than in the recurrent epistaxis group (P = 0.034, P = 0.043, respectively). The high RDW level was found to be statistically significant both in the recurrent and non-recurrent epistaxis groups than in the control group (P < 0.001). The logistic regression analysis concluded that the high MPV value increased epistaxis 1.367 times [OR: 1.367 (0.952-1.963), P = 0.028]. Conclusion: High MPV levels caused an increase in the bleeding tendency in the patients with recurrent epistaxis.

In patients with hereditary hemorrhagic telangiectasia, treatment with engasertib reduced nosebleed frequency and duration over 12 weeks, with mild reversible rash as the most common drug-related side effect.

Epistaxis greatly affects patients with hereditary hemorrhagic telangiectasia (HHT). Although few systemic treatment exist, nintedanib, is a good candidate thanks to its anti-angiogenic activity. Our main objective was to evaluate the efficacy of oral nintedanib on epistaxis duration in HHT patients with moderate to severe epistaxis. This multicenter phase 2 randomized, placebo-controlled, double-blind trial was conducted between June 2020 and February 2023. Inclusion criteria were being over 18 years old and having a confirmed HHT diagnosis with an epistaxis severity score greater than 4. Sixty patients were randomized to receive either nintedanib or placebo for 12 weeks with a 12 week follow-up. The primary endpoint was the proportion of patients achieving a reduction of at least 50% in mean monthly epistaxis duration comparing the 8 weeks before treatment to the last 8 weeks of treatment. Main secondary outcomes included monthly duration and frequency of epistaxis and hemoglobin levels. Of the 60 randomized patients, 56 completed the trial. Thirteen patients (43%) in the nintedanib group vs 8 (27%) in the placebo group met the primary endpoint (p = 0.28). We observed a significant decrease in median epistaxis (57% vs 27%, p = 0.013) and a significant increase in median hemoglobin levels (+ 18 vs − 1 g/L, p = 0.02) in the nintedanib vs the placebo group. Although we did not achieve our primary outcome, we observed a significant reduction in epistaxis duration and a significant increase in hemoglobin levels in patients treated with nintedanib. This supports the efficacy of nintedanib, and further studies are needed.

Hereditary hemorrhagic telangiectasia is a rare vascular genetic disease. Epistaxis is the most frequent and disabling manifestation, and timolol appears to be a new therapeutic option as non-selective beta-blockers have in vitro and in vivo anti-angiogenic properties. Our main objective was to evaluate the efficacy of TIMOLOL nasal spray as a treatment for epistaxis in hereditary hemorrhagic telangiectasia. This study is a single-center, randomized, phase 2, double-blind placebo-controlled study with an allocation ratio of 1:1. It was proposed to patients with hereditary hemorrhagic telangiectasia monitored at the French Reference Center, and we included patients aged over 18 years, diagnosed with hereditary hemorrhagic telangiectasia and epistaxis. The treatment was self-administered by the patient with a posology of one spray (50 microL) of timolol 0.5% or placebo in each nostril twice a day for 28 consecutive days. The primary efficacy endpoint was mean monthly epistaxis duration, assessed by monitoring epistaxis grids. A total of 58 patients were randomized and treated. The baseline characteristics were similar in the 2 groups. Mean monthly epistaxis duration measured at 3 months was not significantly different in the 26 patients receiving the drug in comparison with the placebo group (p = 0.54). Toxicity was low and no severe adverse events were reported. One limitation is that we included all HHT patients with nosebleeds and did not take into account history of nasal surgery or nasal crusts. Timolol, administered by nasal spray at a dose of 0.25 mg in each nostril twice a day for 28 consecutive days, did not improve epistaxis in patients with hereditary hemorrhagic telangiectasia at 4 months after the beginning of the treatment.

Hereditary hemorrhagic telangiectasia (HHT) is a rare angiogenic disorder causing chronic gastrointestinal bleeding, epistaxis, and severe anemia. Pazopanib is an oral multi-kinase angiogenesis inhibitor with promise to treat bleeding in HHT. We analyzed outcomes of HHT patients with the most severe bleeding causing RBC transfusion dependence treated on a predefined institutional pazopanib treatment pathway (with data collected retrospectively). The primary endpoint was achievement of transfusion independence. Secondary endpoints included hemoglobin, epistaxis severity score, RBC transfusion and iron infusion requirements, number of local hemostatic procedures, ferritin and transferrin saturation, compared using paired and repeated measures mean tests. Thirteen transfusion-dependent HHT patients received pazopanib [median (range) dose 150 (25–300) mg daily)] for a median of 22 months. All patients achieved transfusion independence. Compared with pretreatment, pazopanib increased mean hemoglobin by 4.8 (95% CI, 3.6–5.9) g/dL (7.8 vs. 12.7 g/dL, P < 0.0001) and decreased mean epistaxis severity score by 4.77 (3.11–6.44) points (7.20 vs. 2.43 points, P < 0.0001) after 12 months of treatment. Compared with 3 months of pretreatment, RBC transfusions decreased by 93% (median of 16.0 vs. 0.0 units, P < 0.0001) and elemental iron infusion decreased by 92% (median of 4500 vs. 0 mg, P = 0.005) during the first 3 months of treatment; improvements were maintained over time. Pazopanib was well-tolerated: hypertension, lymphocytopenia, and fatigue were the most common TEAEs. In conclusion, pazopanib was safe and effective to manage severe bleeding in HHT, liberating all patients from transfusion dependence and normalizing hematologic parameters at doses lower than used to treat malignancies. These findings require confirmation in a randomized trial.

Initial management of epistaxis involves digital compression of the lower third of the nose for 15 to 20 minutes. Anterior bleeding not controlled with compression can usually be controlled with topical vasoconstrictors, tranexamic acid, cautery, or anterior nasal packing. If epistaxis continues, posterior packing, arterial ligation, or embolization may be used.

The purpose of this study is to (1) to determine if treatment of underlying allergic rhinitis (AR) in children will affect epistaxis outcome, (2) to compare efficacy of three outpatient AR treatment regimens in epistaxis outcomes, and (3) to investigate potential factors in the pathogenesis of epistaxis with underlying AR. A single-blind randomized-controlled study was conducted in the Otolaryngology clinic in KK Women’s and Children’s Hospital. Sixty children aged below 18 years with underlying untreated AR, with first presentation of epistaxis, were randomized to three different AR treatments: treatment 1, antihistamine (20 patients); treatment 2, nasal steroid spray (20 patients); and treatment 3, both antihistamine and nasal steroid spray (20 patients). Epistaxis severity and frequency were assessed. Pre-treatment, 95% of patients within each of the three treatment groups described epistaxis symptoms. Post-treatment, there was improvement in epistaxis outcome (resolution of epistaxis) with 20% (4/20), 40% (8/20), and 60% (12/20) of patients in treatment groups 1 (antihistamine), 2 (nasal steroid spray), and 3 (combined therapy) respectively, who reported resolution of epistaxis. Treatment regimens containing nasal steroid spray resulted in greater improvement of epistaxis severity and frequency. Combined therapy (treatment 3) resulted in the best epistaxis outcome at 1-month follow-up. Majority (90%) reported nose-picking/rubbing behavior. Conclusions: Intranasal corticosteroids are superior to oral antihistamines in relieving itch or rhinorrhea in AR. Intranasal corticosteroids may be important in treating epistaxis with underlying AR, because digital trauma from itch/rhinorrhea-related nose-picking/rubbing frequently leads to epistaxis. Results from this study will be important to primary and emergency physicians, community pediatricians, and pediatric allergists and otolaryngologists. What is Known: •  Childhood epistaxis commonly co-exists with allergic rhinitis (AR), causing significant symptoms and distress to patients. •  There are currently no studies reporti ng on epistaxis outcome aft er treatment of underlying AR. What is New: • This is a single-blind randomized-controlled study of 60 children aged below 18 years with underlying untreated AR, with first presentation of epistaxis to a children’s hospital in Singapore Patients were randomized to three different regimens to treat AR: treatment 1, antihistamine; treatment 2, nasal steroid spray; and treatment 3, both antihistamine and nasal steroid spray. • Treatment regimens containing nasal steroid spray improved epistaxis outcomes, with combined therapy of antihistamine and nasal steroid spray resulting in the best outcome for resolution of epistaxis among the three treatment regimens.

Nasotracheal intubation is mainly performed to provide a safe airway during maxillofacial surgeries. Several guiding devices are suggested to facilitate nasotracheal intubation and reduce complications. We attempted to compare intubation conditions during nasotracheal intubation using a nasogastric tube and a suction catheter, which are readily available in operating rooms. In this study, 114 patients undergoing maxillofacial surgery were randomly divided into the nasogastric tube guidance group (NG group) and the suction catheter guidance group (SC group). The primary outcome was the total intubation time. Moreover, the incidence and degree of epistaxis, the position of the tube in the nasal cavity after intubation, and the number of manipulations during intubation in the nasal cavity were investigated. The insertion time from the nostril to the oral cavity and the total intubation time were significantly shorter in the SC group than in the NG group (p < 0.001). The incidence of epistaxis was lower at 35.1% in the NG group and 43.9% in the SC group than the previously reported 60–80%, but there was no statistical difference between the two groups. The use of a suction catheter aid during nasotracheal intubation can be used effectively because it shortens the intubation time and does not increase complications.