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. | Â |
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. | Â |
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. | Â |
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) | Â |
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. | Â |