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Table 4 Direct costs in TSC patients

From: A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC)

Study N patients total Group Costing year Original cost figure given Cost/year/patient (calculated) Cost in 2018 PPP-$ Significant cost drivers (in regression analysis) Out-of-pocket spending Cost per admission Further measures
Shepherd et al. 2017 [23] 286 TSC and epilepsy 2014 GBP 14,335 / 3y GBP 4778 6643 number of organ systems involved; combination of kidney and urinary/dermatological manifestations n. r. n. r.  
Vekeman et al. 2015 – CKD Stage I [35] 369 TSC and renal manifestations 2012 EUR 1275 EUR 1275 1715 CKD stage V vs. CKD stage I; AML size > = 3,5 cm; comorbid moderate or severe LAM n.r. n. r.  
Vekeman et al. 2015 – CKD Stage V [35] 2012 EUR 30,641 EUR 30,641 41,207
Kingswood et al. 2016a [110] 278b All TSC 2014 GBP 12,681 /3y GBP 4227 5876 number of organ systems involved; kidney and urinary tract manifestations, nervous system manifestations; pairwise: circulatory/kidney and urinary tract, nervous system/psychiatric, dermatological/kidney & urinary tract manifestations n. r. n. r. GP visits (GBP 3433, PPP-$ 4772 vs. GBP 1283, PPP-$ 1784)
inpatient hospitalizations (GBP 7050, PPP-$ 9801 vs. GBP 3298, PPP-$ 4585)
outpatient visits (GBP 2071, PPP-$ 2879 vs. GBP 613, PPP-$ 852)
primary care drugs (GBP 2607, PPP-$ 3624 vs. GBP 479, PPP-$ 666)
Kingswood et al. 2016b [111] 79b TSC and renal manifestations 2014 GBP 15,162 / 3y GBP 5054 7026 number of primary TSC manifestations; nervous system manifestations; pairwise: combination of dermatology/psychiatric manifestations n. r. n. r.  
Skalicky et al. 2018 [119] 609a all TSC 2018 Tests/procedures: USD 5499-20,403
Hospital expenses: USD 1263-5533
Doctor’s visits: USD 1646-4462
ER visits: USD 702–2671
n. r. n. r. n. r. For tests and/or procedures: USD 5499-20,403
Hospital expenses: USD 1263-5533
Doctor’s visits: USD 1646-4462
ER visits: USD 702–2671
n. r.  
Skalicky et al. 2015 [106] 116a TSC and SEGA 2015 USD 80–129 n. r. n. r. n. r. median monthly: USD 80–129 (about 50% of patients) n.r.  
Song X. et al. 2017 [114] 743 TSC and AML 2013 USD 29,240-48,499 USD 29,240 to 48,499 31,605 to 52,422 n. r. n. r. n. r.  
Wilson et al. 2016 [112] 5655 (admissions) all TSC n. r. USD 14,807 (IQR 7319-31,180) n. r. n. r. n. r. n. r. USD 14,807 (IQR 7319-31,180)  
Sun et al. 2015 [107] 47 TSC and SEGA surgery 2010 USD 8543.1 (SD 11,187.6) for presurgical year
USD 85,397 (SD 56,258.9) for postsurgical year
USD 8543 to 85,397 9805 to 98,008 n. r. n. r. n. r. Presurgical year: inpatient USD 3770, PPP-$ 4327
outpatient treatments USD 3473, PPP-$ 3986 Medications USD 1300, PPP-$ 1492
postsurgical year
surgery USD 71,562, PPP-$ 82,130
Outpatient costs USD 11,497 PPP-$ 13,195 medication costs USD 2338, PPP-$ 2683
(costs 1.6 to 4.3 times higher than in the presurgical year [inpatient: 4.3:1, outpatient: 2.5:1, medication: 1.6:1, and total: 3.1:1])
Kristof et al. 2015 [104] 1004 (TSC)
29 (LAM)
TSC and/or LAM 2011 TSC: CAD 513 (SD 5.83)
LAM: CAD 1434 (SD 10.14)
TSC-LAM: CAD 1718 (SD 10.53)
General population control: CAD 281 (SD 4.37)
CAD 513 to 1718 424 to 1421 n. r. n. r. n. r.  
  1. a, b denotes patients originating from same cohort; AML Angiomyolipoma, CAD Canadian dollar, CCHMC Cincinnati Children’s Hospital Medical Center, CKD Chronic kidney disease, ER Emergency room, GBP Great Britain pound, HDI-SF Hamilton depression inventory short form, ICF International classification of functioning, disability, and health, IQR Interquartile range, LAM Lymphangioleiomyomatosis, n. r. Not reported, PedsQL Pediatric quality of life inventory, PPPY Cost per person per year, PPP-$ International Dollar purchasing power parities, SEGA Subependymal giant astrocytoma, SF12 Short form health survey 12-item, version 2, TSC Tuberous sclerosis complex, USD United States dollar, WPAI CIQ-SHP Work Productivity and Activity Impairment plus Classroom Impairment Questions: Special Health Problem (WPAI CIQ-SHP), version 2 questionnaire; for data sources please refer to Table 2