AP-HP, Urgences-Samu 93, hôpital Avicenne, Université Paris 13, Inserm U942, Bobigny, France; AP-HP, Urgences, Centre de référence sur les angiœdèmes à kinines, hôpital Louis Mourier, Université Paris 7, Colombes, France.
Assistance Publique - Hôpitaux de Paris (AP-HP), Médecine Interne, DHUi2B, Centre de Référence associé sur les angiœdèmes à kinines, Hôpital Saint-Antoine, Université Paris 6, Paris, France.
Ann Emerg Med. 2018 Aug;72(2):194-203.e1. doi: 10.1016/j.annemergmed.2018.01.053. Epub 2018 Mar 2.
Hereditary angioedema is a rare disease associated with unpredictable, recurrent attacks of potentially life-threatening edema. Management of severe attacks is currently suboptimal because emergency medical teams are often unaware of new specific treatments. The objective of this trial is to test whether a dedicated national telephone care-management strategy would reduce resource use during severe hereditary angioedema attacks.
We conducted a cluster-randomized multicenter prospective trial of patients with a documented diagnosis of hereditary angioedema (type I, II or FXII hereditary angioedema). Participants were enrolled between March 2013 and June 2014 at 8 participating reference centers. The randomized units were the reference centers (clusters). Patients in the intervention arm were given a national free telephone number to call in the event of a severe attack. Emergency physicians in the SOS-hereditary angiœdema (SOS-HAE) call center were trained to advise or prescribe specific treatments. The primary outcome was number of admissions for angioedema attacks. Economic evaluation was also performed.
We included 100 patients in the SOS-HAE group and 100 in the control group. During the 2 years, there were 2,368 hereditary angioedema attacks among 169 patients (85%). Mean number of hospital admissions per patient in the 2-year period was significantly greater in the usual-practice group (mean 0.16 [range 0 to 2] versus 0.03 [range 0 to 1]); patient risk difference was significant: -0.13 (95% confidence interval -0.22 to -0.04; P=.02). Probabilistic sensitivity graphic analysis indicated a trend toward increased quality-adjusted life-years in the SOS-HAE group.
A national dedicated call center for management of severe hereditary angioedema attacks is associated with a decrease in hospital admissions and may be cost-effective if facilities and staff are available to deliver the intervention alongside existing services.
遗传性血管性水肿是一种罕见疾病，与不可预测、反复发作的潜在危及生命的水肿有关。由于紧急医疗团队通常不知道新的特定治疗方法，因此严重发作的治疗目前并不理想。本试验的目的是测试专门的国家电话护理管理策略是否会减少遗传性血管性水肿严重发作期间的资源使用。
我们对有遗传性血管性水肿（I 型、II 型或 XII 型遗传性血管性水肿）确诊的患者进行了一项集群随机多中心前瞻性试验。参与者于 2013 年 3 月至 2014 年 6 月在 8 个参与的参考中心招募。随机单位为参考中心（集群）。干预组的患者在发生严重发作时获得了一个全国免费电话号码。 SOS-遗传性血管性水肿（SOS-HAE）呼叫中心的急诊医生接受了培训，以提供建议或开具体的治疗方案。主要结局是血管性水肿发作的住院人数。还进行了经济评估。
我们将 100 名患者纳入 SOS-HAE 组，100 名患者纳入对照组。在 2 年期间，169 名患者中有 85%发生了 2368 次遗传性血管性水肿发作。在 2 年期间，每位患者的平均住院人数在常规实践组中明显更大（平均值为 0.16[范围 0 至 2] 与 0.03[范围 0 至 1]）；患者风险差异显著：-0.13（95%置信区间-0.22 至-0.04；P=0.02）。概率敏感性图形分析表明，在 SOS-HAE 组中，生活质量调整生命年呈增加趋势。
遗传性血管性水肿严重发作的国家专用呼叫中心与住院人数减少有关，如果在提供现有服务的同时提供设施和人员来实施干预措施，则可能具有成本效益。
