A Systematic Review of Socioeconomic Burden in Hereditary Angioedema


Craig TJ1, Cribbs K2, Czado S3
1The Pennsylvania State University, Hershey, PA, USA, 2Alkemi LLC, Manchester Center, VT, USA, 3KalVista Pharmaceuticals, Inc., Cambridge, MA, USA

Presentation Documents

OBJECTIVES: Over the past decade, several treatment options for hereditary angioedema (HAE) have emerged, however, concerns regarding the financial impact of these therapies on patients, caregivers, and healthcare systems persist. Previous literature indicates that HAE is associated with substantial direct medical costs; however, indirect costs are not as well-characterized. This study aimed to comprehensively evaluate and synthesize the current body of evidence around the socioeconomic burden of HAE on patients and caregivers.

METHODS: We conducted a systematic literature review (SLR) on economic outcomes, including indirect costs, among individuals with HAE in accordance with PRISMA guidelines. We searched multiple databases, including, PubMed, Embase, and Google Scholar. Article inclusion was limited to English peer-review and grey literature published between January 1, 2007 – July 1, 2022. Two independent reviewers assessed literature eligibility and abstracted data. We adjusted all costs for inflation to 2022 USD.

RESULTS: We identified 66 studies. The majority of studies were observational (64%) and peer-reviewed (58%), and the most common study location was the United States (42%). Among these 66 studies, 41% assessed socioeconomic burden in HAE, revealing annual mean indirect costs of up to $57,237. Factors contributing to indirect costs included reduced work productivity, travel, childcare, and caregiver burden. Lower labor market participation accounted for up to 37% of total indirect costs, while caregiver burden accounted for up to 14%. Mean percentage impairments in work productivity and activity, as measured by the Work Productivity and Activity Impairment Questionnaire (WPAI), ranged from 22 – 25% and 21 – 34%, respectively. Days missed per year ranged from 9.0 – 19.9, with up to 28.2 days missed for severe attacks.

CONCLUSIONS: Despite advancements in therapeutic options for HAE, substantial socioeconomic burden for patients and caregivers remains, especially as attack severity increases.

Conference/Value in Health Info

2023-05, ISPOR 2023, Boston, MA, USA

Value in Health, Volume 26, Issue 6, S2 (June 2023)




Economic Evaluation, Patient-Centered Research, Study Approaches

Topic Subcategory

Literature Review & Synthesis, Patient-reported Outcomes & Quality of Life Outcomes, Work & Home Productivity - Indirect Costs


No Additional Disease & Conditions/Specialized Treatment Areas

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