Socioeconomic burden of hereditary angioedema: results from the hereditary angioedema burden of illness study in Europe

Orphanet Journal of Rare Diseases

Teresa Caballero⁵  Henrik B Boysen⁴  Suzanne Wait¹  Zlatko Sisic⁶  Emily Hautamaki²  Kathleen Beusterien²  Anette Bygum⁷  Emel Aygören-Pürsün³ 

[1] SHW Health Ltd, 40 Lena Gardens, W6 7PZ London, UK;Oxford Outcomes Inc., an ICON plc company, 7315 Wisconsin Ave, Suite 250 W, 20814 Bethesda, MD, USA;Department for Children and Adolescents, Angioedema Centre, University Hospital Frankfurt, Goethe University, Theodor-Stern-Kai 7, 60596 Frankfurt, Germany;HAEi – International Patient Organization for C1 Inhibitor Deficiencies, Soenderholmvej 158, DK-8361 Bering, Hasselager, Denmark;Allergy Department, Hospital La Paz Institute for Health Research (IdiPaz), Biomedical Research Network on Rare Diseases U754 (CIBERER), University Hospital La Paz, Paseo de la Castellana 261, 28046 Madrid, Spain;ViroPharma, Chatsworth House, 29 Broadway, SL6 1LY Maidenhead, UK;HAE Centre Denmark, Department of Dermatology and Allergy Centre, Odense University Hospital, 5000 Odense C, Denmark

关键词: Productivity;    Resource utilization;    Burden of illness;    Hereditary angioedema;   

Others  :  861376
DOI  :  10.1186/1750-1172-9-99

 received in 2014-01-24, accepted in 2014-06-16,  发布年份 2014

【 摘 要 】

Background

Hereditary angioedema (HAE) due to C1 inhibitor deficiency is a rare but serious and potentially life-threatening disease marked by spontaneous, recurrent attacks of swelling. The study objective was to characterize direct and indirect resource utilization associated with HAE from the patient perspective in Europe.

Methods

The study was conducted in Spain, Germany, and Denmark to assess the real-world experience of HAE via a cross-sectional survey of HAE patients, including direct and indirect resource utilization during and between attacks for patients and their caregivers over the past 6 months. A regression model examined predictors of medical resource utilization.

Results

Overall, 164 patients had an attack in the past 6 months and were included in the analysis. The most significant predictor of medical resource utilization was the severity of the last attack (OR 2.6; p < 0.001). Among patients who sought medical care during the last attack (23%), more than half utilized the emergency department. The last attack prevented patients from their normal activities an average of 4–12 hours. Patient and caregiver absenteeism increased with attack severity and frequency. Among patients who were working or in school (n = 120), 72 provided work/school absenteeism data, resulting in an estimated 20 days missing from work/school on average per year; 51% (n = 84) indicated that HAE has hindered their career/educational advancement.

Conclusion

HAE poses a considerable burden on patients and their families in terms of direct medical costs and indirect costs related to lost productivity. This burden is substantial at the time of attacks and in between attacks.

【 授权许可】

   
2014 Aygören-Pürsün et al.; licensee BioMed Central Ltd.

【 图 表 】

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