Library Header Image
LSE Research Online LSE Library Services

Socio-economic costs of rare diseases and the risk of financial hardship: a cross-sectional study

Chung, Claudia C.Y., Ng, Nicole Y.T., Ng, Yvette N.C., Lui, Adrian C.Y., Fung, Jasmine L.F., Chan, Marcus C.Y., Wong, Wilfred H.S., Lee, So Lun, Knapp, Martin ORCID: 0000-0003-1427-0215 and Chung, Brian H.Y. (2023) Socio-economic costs of rare diseases and the risk of financial hardship: a cross-sectional study. The Lancet Regional Health - Western Pacific, 34. ISSN 2666-6065

[img] Text (Knapp_socio-economic-costs-of-rare-diseases--published) - Published Version
Available under License Creative Commons Attribution Non-commercial No Derivatives.

Download (619kB)

Identification Number: 10.1016/j.lanwpc.2023.100711


Background: To achieve universal healthcare coverage (UHC), the rare disease (RD) population must also receive quality healthcare without financial hardship. This study evaluates the impact of RDs in Hong Kong (HK) by estimating cost from a societal perspective and investigating related risk of financial hardship. Methods: A total of 284 RD patients and caregivers covering 106 RDs were recruited through HK's largest RD patient group, Rare Disease Hong Kong, in 2020. Resource use data were collected using the Client Service Receipt Inventory for Rare disease population (CSRI-Ra). Costs were estimated using a prevalence-based, bottom-up approach. Risk of financial hardship was estimated using catastrophic health expenditure (CHE) and impoverishing health expenditure (IHE) indicators. Multivariate regression was performed to identify potential determinants. Findings: Annual total RD costs in HK were estimated at HK$484,256/patient (United States (US) $62,084). Direct non-healthcare cost (HK$193,555/US$24,814) was the highest cost type, followed by direct healthcare (HK$187,166/US$23,995), and indirect (HK$103,535/US$13,273) costs. CHE at the 10% threshold was estimated at 36.3% and IHE at the $3.1 poverty line was 8.8%, both significantly higher than global estimates. Pediatric patients reported higher costs than adult patients (p < 0.001). Longer years since genetic diagnosis was the only factor significantly associated with both total costs (p = 0.026) and CHE (p = 0.003). Interpretation: This study serves as the first in the Asia Pacific region to simultaneously assess the societal costs and financial hardship related to RDs and highlights the importance of an early genetic diagnosis. These results contribute to existing evidence on the globally ubiquitous high costs of RDs, warranting collaboration between different stakeholders to include RD population in UHC planning. Funding: Health and Medical Research Fund, and the Society for the Relief of Disabled Children.

Item Type: Article
Official URL:
Additional Information: © 2023 The Authors
Divisions: Health Policy
Subjects: R Medicine > RA Public aspects of medicine
H Social Sciences > HB Economic Theory
Date Deposited: 14 Mar 2023 17:48
Last Modified: 23 Feb 2024 23:21

Actions (login required)

View Item View Item


Downloads per month over past year

View more statistics