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Table 7 Studies on direct and indirect costs in children and adolescents with TSC

From: Direct and indirect costs and cost-driving factors of Tuberous sclerosis complex in children, adolescents, and caregivers: a multicenter cohort study

  Grau et al. Current study Betts et al. 2020 [48] Chu et al. 2020 [47] Skalicky et al. 2018 [40] Song et al. 2017 [32] Shepherd et al 2017 [11]* Kingswood et al. 2016 [13]* Kingswood et al. 2016 [36]* Wilson et al. 2016 [39] Sun et al. 2015 [33] Vekeman et al. [35]
Study design multicenter, r multicenter, r multicenter, r multicenter, p multicenter, r multicenter, r multicenter, r multicenter, r multicenter, r multicenter, r monocenter, r
Costing year 2019 2019 2017 2012 2013 2014 2014 2014 n.r 2010 2012
Country (city) Germany USA Hong Kong USA USA United Kingdom United Kingdom United Kingdom USA USA Netherlands (Utrecht)
Group All TSC TSC and epilepsy All TSC All TSC TSC and AML TSC and epilepsy All TSC TSC and kidneys All TSC TSC and SEGA surgery TSC and kidneys
Number of patients 184 2028 284 179 256 209 286 79 5655 47 369
Study population C C & A C & A C C C & A C & A C & A C & A C & A C & A
Patients with epilepsy 91.8% 100% 71.3% n.r n.r 100% n.r n. r 41.2% 91% n.r
Age in years (median) 0.7–21.8 (9.8) Mean 25.3 0.45–89.9 (27.2) 0–18 (6.0) Mean 9.7, 6.9 Mean 26.8 Mean 31.5 Mean 38.3 Mean 22.3 Mean 11.6 Mean 42.87
Patients with ASDs 83.7% 89.5% n. r n.r n.r 88% 42.7% 68.4% n.r n.r n.r
Patients with mTOR-inhibitors 26.6% 10% 16.5% n.r n.r n.r n.r n.r n.r n.r n.r
  Mean PPPY Mean PPPY Mean PPPY Median PPPY Mean PPPY Mean PPPY Mean PPPY Mean PPPY Median Mean PPPY Mean PPPY
Total direct costs EUR 19,796 n.r n.r n.r USD 35,381–29,2404 GBP 47785 GBP 42275 GBP 50545 n.r USD 8543–85,3976 EUR 1275–31,9168
 Medication EUR 10,632 USD 18,836 n.r USD 18002,3 USD 7445–11,0024 no specific amount GBP 5955 (only primary care) GBP 8695 (only primary care) n.r USD 1300–23386 EUR 429–15088
  ASDs EUR 1040 USD 12,866 n.r n.r n.r n.r n.r n.r n.r n.r n.r
  mTOR inhibitors EUR 9236 USD 4028 n.r n.r n.r n.r n.r n.r n.r n.r n.r
 Hospitalization EUR 4108 USD 2106 USD 58191 USD 16752, 3 USD 10,368–89014 no specific amount GBP 21815 GBP 23505 USD 14,807 USD 3770–71,5626 n.r
 Ancillary therapies EUR 1628 n.r n.r USD 12002,3 n.r n.r n.r n.r n.r n.r n.r
 Outpatient treatment EUR 1384 USD 13,455 USD 14141 USD 4002 USD 17,000–90114 no specific amount GBP 6455 GBP 6905 n.r USD 3473–11,4976 n.r
 ER visists n.r USD 1535 USD 1161 USD 4002 USD 568–3264 n.r n.r n.r n.r n.r n.r
  Mean caregivers PY    Mean caregivers        
Total indirect costs in caregivers EUR 12,736 n. r n. r 42%9 n. r n. r n. r n. r n. r n.r n.r
 Quit work EUR 6352 n.r n.r n.r n.r n.r n.r n.r n.r n.r n.r
 Reduction of working hours EUR 4780 n.r n.r n.r n.r n.r n.r n.r n.r n.r n.r
 Days off due to TSC EUR 1604 n.r n.r n.r n.r n.r n.r n.r n.r n.r n.r
  1. p = prospective; r = retrospective; C = Children; A = Adults; n.r. = not reported; PPPY = per person/per year; PY = per year; ASD = antiseizure drug
  2. 110% of actual expenses, government subsidized more than 90%
  3. 2"out-of-pocket" direct spending
  4. 3Calculated
  5. 4The first amount is from commercial cohort, the second one from medicaid cohort
  6. 5Calculated for one year, original cost figure given for a 3 year period, excluding GP administartion encounters
  7. 6From SEGA pre-surgery to post-surgery period
  8. 7Calculated across all CKD stages
  9. 8The first amount is from CKD stage 1, the second one from CKD stage 5, overall mean PPPY costs for AML: EUR 1451-3243
  10. 9Proportion of parents with time missed or greater productivity loss
  11. *Same study cohort