Economic Burden of Acquired Hemophilia- A Systematic Literature Review

OBJECTIVES: Acquired hemophilia (AH) is a rare autoimmune disorder characterized by spontaneous bleeding in individuals with no family history, unlike congenital hemophilia. Globally, the economic burden for AH has not been examined systematically, resulting in a lacuna in this aspect, and the review intended to highlight the same.

METHODS: A systematic literature review was conducted using 11 databases including EMBASE, MEDLINE, CINAHL, Web of Science and Scopus, conference abstracts, HTA databases and trial registries through January 2024 to identify publications reporting costs and healthcare resource utilization (HCRU) in AH. As per the Cochrane Handbook of Systematic Reviews, the CCEMG – EPPI-Centre Cost Converter (v.1.6) was used for adjusting estimates of cost expressed in any currency and price year to USD and 2024 respectively.

RESULTS: 9 studies were identified, reporting economic outcomes from USA, Canada, France, Colombia, and Netherlands. Most patients were older than 60 years, and there was female predominance. The costs were from 2010 – 2020 and were adjusted accordingly. The average hospital admission cost ranged from $111,670 to $1,975,003 with average hospitalization cost per patient ranging from $51,120 to $204,953. Among the interventions, the average total cost for rFVIIa per admission was $380,559, while the average total cost per admission was higher for rpFVIII ($1,062,352). The average total patient cost for immunosuppressant therapy ranged between $162,596 to $411,633. The average hospital stay duration across all the interventions ranged from 6.1 to 73.0 days depending on the type of treatment.

CONCLUSIONS: A high economic burden could be a result of the challenges with diagnosis and lack of approved treatments. The cost of hemostatic agents to treat bleeding appears to be the largest contributor to inpatient cost. Future studies should be endeavored to quantify the HCRU and cost attributable to patients with AH and to bridge the evidence gap.