Healthcare Costs and Resource Use in Patients With Sjögren's Syndrome (SS): A Systematic Literature Review of Real-World Evidence (RWE) Studies
Author(s)
Amrinder Singh, M.Pharm1, Liliane Martinez, MSc2, Rachel Goldgrub, MSc1.
1ICON plc, Vancouver, BC, Canada, 2ICON plc, Mexico city, Mexico.
1ICON plc, Vancouver, BC, Canada, 2ICON plc, Mexico city, Mexico.
Presentation Documents
OBJECTIVES: This systematic review evaluates the healthcare cost and resource utilization for Sjögren's syndrome (SS) patients in the USA, Canada, and EU-5 countries.
METHODS: Literature was searched in EMBASE, MEDLINE, and EconLit (January 2014 - December 2024). Records were screened by two independent reviewers using predefined eligibility criteria, with disagreements resolved by a third senior reviewer at abstract and full-text level.
RESULTS: From 1,321 unique abstracts, 56 citations underwent full-text screening, yielding 13 studies for final inclusion. Patient populations showed mean ages of 52.5-65.8 years, predominantly female (87%-96%). Mean length of stay (LOS) of 3.9 days in the USA and 5.9 days in Canada. Primary SS (pSS) patients aged 17-49 years showed double the hospitalization rate compared to the general population (RR 2.02; 95% CI 1.40-2.87). In Canada, direct and indirect costs were $2,689.10 and $41,093.85 per patient/year, demonstrating 22% and 132% increase in costs versus the costs for patients with no known SS ($2,203.35 and $17,694.17). Total all-cause medical costs including outpatient, emergency department visits and hospitalizations increased 40% in 12 months post SS diagnosis period (pre index: $14,612.10 vs $20,416.50), with outpatient costs making over 50% of the costs. In EU-5, specialist visit frequency varied, being highest in Spain (11.15 visits) and lowest in the UK (5.78 visits). An Italian study reported a mean LOS of 14.3 days. Analysis of disease trends revealed decreasing pSS-related hospitalizations over two decades (13% in 1996 vs 7% in 2016). Despite the decline in the incidence of pSS from 2012 to 2018, the mean annual costs per patient have increased (2011: €5780 vs 2018: €9710) even surpassing the French national projections of expected costs for chronic diseases.
CONCLUSIONS: Despite declining hospitalization rates, SS patients face substantial and increasing healthcare costs across regions, highlighting the need for optimized strategies and further research into cost-effective management approaches.
METHODS: Literature was searched in EMBASE, MEDLINE, and EconLit (January 2014 - December 2024). Records were screened by two independent reviewers using predefined eligibility criteria, with disagreements resolved by a third senior reviewer at abstract and full-text level.
RESULTS: From 1,321 unique abstracts, 56 citations underwent full-text screening, yielding 13 studies for final inclusion. Patient populations showed mean ages of 52.5-65.8 years, predominantly female (87%-96%). Mean length of stay (LOS) of 3.9 days in the USA and 5.9 days in Canada. Primary SS (pSS) patients aged 17-49 years showed double the hospitalization rate compared to the general population (RR 2.02; 95% CI 1.40-2.87). In Canada, direct and indirect costs were $2,689.10 and $41,093.85 per patient/year, demonstrating 22% and 132% increase in costs versus the costs for patients with no known SS ($2,203.35 and $17,694.17). Total all-cause medical costs including outpatient, emergency department visits and hospitalizations increased 40% in 12 months post SS diagnosis period (pre index: $14,612.10 vs $20,416.50), with outpatient costs making over 50% of the costs. In EU-5, specialist visit frequency varied, being highest in Spain (11.15 visits) and lowest in the UK (5.78 visits). An Italian study reported a mean LOS of 14.3 days. Analysis of disease trends revealed decreasing pSS-related hospitalizations over two decades (13% in 1996 vs 7% in 2016). Despite the decline in the incidence of pSS from 2012 to 2018, the mean annual costs per patient have increased (2011: €5780 vs 2018: €9710) even surpassing the French national projections of expected costs for chronic diseases.
CONCLUSIONS: Despite declining hospitalization rates, SS patients face substantial and increasing healthcare costs across regions, highlighting the need for optimized strategies and further research into cost-effective management approaches.
Conference/Value in Health Info
2025-05, ISPOR 2025, Montréal, Quebec, CA
Value in Health, Volume 28, Issue S1
Code
EE431
Topic
Economic Evaluation
Topic Subcategory
Cost/Cost of Illness/Resource Use Studies
Disease
SDC: Rare & Orphan Diseases