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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