Table 1 Characteristics of COI studies for IRDs, arranged by chronological order (n = 9)
From: Cost-of-illness studies of inherited retinal diseases: a systematic review
Study (year of costing) | Country | Type of IRD | Data source | Sample size | Study design | Perspective | Mean total costs per year | Mean health costs per year (%) | Mean non-health costs per year (%) |
|---|---|---|---|---|---|---|---|---|---|
Aziz et al., (2021) [19] | United States (US) | Stargardt disease | Insurance claims | 472,428 | Retrospective, prevalence-based | Healthcare system | NR | US$105.58/pt | NR |
Chay et al., (2023) [25] | Singapore | Group of IRDs (63.2% with retinitis pigmentosa; median age of symptom onset was 32 years) | Multiple data sources | 500 | Prospective, prevalence-based | Societal | S$48,810,000 (= US$36,027,137) S$9382/pt (= US$6926.36/pt) | S$6,200,000 (= US$4,577,218) S$1194/pt (= US$881.48/pt) (12.1%) | S$42,590,000 (= US$31,442,536) S$8188/pt (= US$6044.88/pt) (87.9%) |
Frick et al., (2012) [20] | United States | Retinitis pigmentosa | Insurance claims | 2990 | Retrospective, prevalence-based | Healthcare system | NR | US$14,988/pt | NR |
Galvin et al., (2020) [6] | Republic of Ireland (RoI) | Group of 10 IRDs (achromatopsia, best disease, choroideremia, cone dystrophy, cone-rod dystrophy, Leber congenital amaurosis, retinitis pigmentosa, Stargardt disease, Usher syndrome and X-linked retinoschisis) | Multiple data sources | 1521 | Retrospective, prevalence-based | Societal | £42,600,000 (= US$53,937,990) US$24,107-US$40,718/pt | £1,900,000 (= US$2,405,685) (4.5%) | £40,700,000 (= US$51,532,305) (95.5%) |
United Kingdom (UK) | 20,815 | £523,300,000 (= US$6662,576,295) US$21,658-US$36,549/pt | £25,000,000 (= US$31,653,750) (4.8%) | £498,300,000 (= US$630,922,545) (95.2%) | |||||
Gong et al., (2021) [7] | United States | Group of 14 IRDs (achromatopsia, Bardet-Biedl Syndrome, best disease, blue cone monochromacy, choroideremia, cone dystrophy, cone-rod dystrophy, Leber congenital amaurosis, Leber’s hereditary optic neuropathy, retinitis pigmentosa, rod-cone dystrophy, Stargardt disease, Usher syndrome and X-linked retinoschisis) | Multiple data sources | 51,325–122,007 | Retrospective, prevalence-based | Societal | US$13,414mil-US$31,797.4mil US$33,017-US$186,051/pt | US$963.8mil- US$2,216.8mil (7%) | US$12,450.2mil- US$29,580.6mil (93%) |
Canada (CA) | 5,841–23,891 | CAN$1,637.8mil- CAN$6,687.5mil (= US$1,236.0mil- US$5,046.9mil) US$16,470-US$275,045/pt | CAN$37.8mil- CAN$144.3mil (= US$28.5mil- US$108.9mil) (2%) | CAN$1,600mil- CAN$6,543.2mil (= US$1,207.5mil- US$4,938.0mil) (98%) | |||||
Kessel et al., (2022) [23] | Denmark | Group of IRDs (childhood-onset; not specified) | Multiple data sources | 412 | Retrospective, prevalence-based | Healthcare system | NR | (Overall) €1,488/pt (= US$1,619.6/pt) (Age 0–10) €1,145/pt (= US$1,246.3/pt) (Age 11–20) €1,409/pt (= US$1,533.6/pt) (Age 21–30) €1,520/pt (= US$1,654.4/pt) Age (30–48) €1,867/pt (= US$2032.1/pt) | NR |
Schofield et al., (2023) [22] | Australia | Group of IRDs (not specified) | Patient and caregiver survey | 94 (patient) 30 (carer) | Prospective, Markov modelling, prevalence-based | Societal | AUS$5.2mil/pt (= US$3,452,140/pt) (Lifetime) AUS$781mil- AUS$1,560mil (= US$518.5mil- US$1,035.6mil) (For whole society) | AUS$690,725/pt (= US$458,554/pt) (Lifetime, 13%) | AUS$4,509,275/pt (= US$2,993,586/pt) (Lifetime, 87%) |
Dong et al., (2021) [21] | United States | Choroideremia | Insurance claims | 199 | Retrospective, prevalence-based | Healthcare system | NR | US$15,372/pt | NR |
Watanabe et al., (2023) [24] | Japan | Retinitis pigmentosa (mean age of onset 11.2 years) | Patient survey | 122 | Prospective, cross-sectional, prevalence-based | Societal | US$20,833/pt US$1,766,013/pt (Lifetime) | US$2176/pt US$184,501/pt (Lifetime, 10%) | US$18,657/pt US$1,581,554/pt (Lifetime, 90%) |