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Ann Emerg Med. 2018 Aug;72(2):194-203.e1. doi: 10.1016/j.annemergmed.2018.01.053. Epub 2018 Mar 2.

Specialist Advice Support for Management of Severe Hereditary Angioedema Attacks: A Multicenter Cluster-Randomized Controlled Trial.

Author information

1
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. Electronic address: nicolas.javaud@aphp.fr.
2
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.
3
AP-HP, URCEco Ile de France, Hôpital de l'Hôtel-Dieu, Université Paris 12, Paris, France.
4
Médecine Interne, Centre de Référence sur les angiœdèmes à kinines, Université de Lille, CHRU de Lille, Lille Cedex, France.
5
Médecine Interne, Centre de Référence sur les angiœdèmes à kinines, CHU de Grenoble, Grenoble, France.
6
Université de Paris-Descartes, AP-HP, HUPC, Unité de Gynécologie Endocrinienne, Hôpital Port Royal, Paris, France.
7
AP-HP, Hôpital Hôtel Dieu, Université Paris 5, Paris, France.
8
AP-HP, Urgences-Samu 93, hôpital Avicenne, Université Paris 13, Inserm U942, Bobigny, France.
9
Hospices Civils de Lyon, Médecine Interne, Lyon Cedex, France; Centre de Référence sur les angiœdèmes à kinines, CHU Edouard Herriot, Lyon Cedex, France.
10
Réanimation, CHU Edouard Herriot, Lyon Cedex, France.
11
Médecine Interne, Centre de Référence sur les angiœdèmes à kinines, CHU de Nancy, Nancy, France.
12
Dermatologie, Centre de Référence sur les angiœdèmes à kinines, Université d'Angers, CHU d'Angers, Angers cedex, France.
13
AP-HP, Unité de Recherche Clinique, hôpital Fernand Widal, Paris, France.
14
AP-HP, Urgences-Samu 93, hôpital Avicenne, Université Paris 13, Inserm U942, Bobigny, France; Inserm U942.

Abstract

STUDY OBJECTIVE:

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.

METHODS:

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.

RESULTS:

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.

CONCLUSION:

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.

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