Table 3.
Indirect costs associated with SMA management (n = 13)
| Study, country (currency, year) | Indirect cost categories with available data [EUR 2019] | |||||||
|---|---|---|---|---|---|---|---|---|
| Lost productivity | Informal care, hours per week | Informal care, loss of earning | Informal care, total costs | Family loss of income | Caregiver/patient unemployment | Patient absenteeism/ presenteeism |
Caregiver absenteeism | |
|
Aranda-Reneo et al. (2020) [69] Multi-national (France, Germany, Spain, and the UK) [NA] |
Mean hours of informal caregiving per patient per day, main caregiver, all activities (SD): Type 1 SMA: 8.3 (3.6) Type 2 SMA: 7.86 (4.8) Type 3 SMA: 3.14 (2.42) All types SMA: 6.89 (4.62) Mean hours of informal caregiving per patient per day, all caregivers, all activities (SD): Type 1 SMA: 14.32 (6.94) Type 2 SMA: 10.48 (6.41) Type 3 SMA: 5.59 (4.73) All types SMA: 10.02 (6.68) |
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|
Chambers et al. (2020) [71] Australia (USD, 2017) |
Mean annual cost per patient for informal care (range): Type 1 SMA: $28,271 ($0–$236,303) [€20,694.10; range €0–€172,971.33] Type 2 SMA: $33,885 ($0–$15,246) [€24,803.41; range €0–€11,159.87] Type 3 SMA: $12,778 ($0–$66,012) [€9,353.45; range €0–€48,320.14] All types SMA: $28,439 (NR) [€20,816.98] Type 2/3 SMA: $28,458 (NR) [€20,831.10] |
Mean annual loss of income per patient (range): Type 1 SMA: $15,829 ($0–$63,315) [€11,586.41; range €0–€46,345.64] Type 2 SMA: $38,245 ($0–$58,943) [€27,994.67; range €0–€43,145.91] Type 3 SMA: $29,988 ($0–$140,415) [€21,951.12; range €0–€102,782.69] All types SMA: $34,707 (NR) [€25,405.08] Type 2/3 SMA: $36,865 (NR) [€26,984.82] |
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|
Farrar et al. (2018) [64] Australia (NA) |
Hours spent caring for a child with SMA per day from n = 7 sample size: 0–4: n = 3 4–8: n = 2 8–16: n = 1 16–24: n = 1 |
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|
Klug et al. (2016) [55] Germany (EUR, 2013) |
Total overall care effort per patient, hours/day: Type 1 SMA: 15 Type 2 SMA: 11 Type 3 SMA: 4 Overall: 7.8 |
Mean annual cost per patient with SMA for informal care (SD): €20,170 (€28,924) [€20,691.69; SD €29,672.11] | ||||||
|
Lopez-Bastida et al. (2017) [58] Spain (EUR, 2014) |
Caregiving time per caregiver (mother/father), daily hours: 4.05 | Mean annual non-healthcare informal costs per patient (SD): €21,127 (€30,253) [€24,671.78; SD €35,328.99] | ||||||
|
Lopez-Bastida et al. (2019) [95] Multi-national (France, Germany and the UK) [EUR 2014] |
Mean annual cost of informal care per patient with SMA (any type): UK: €40,526 [unable to inflate without GBP value] France: €26,619 [€26,997.44] Germany: €39,926 [€40,633.60] |
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|
Marcellusi, (2019) et al. [59] Italy (EUR, NR) |
Mean annual cost of caregiver unemployment per patient Type 1 SMA: €11,310.07 Type 2 SMA: €5769.51 Type 3 SMA: €1381.77 |
Mean annual cost per patient: Patient absenteeism: Type 1 SMA: €0 Type 2 SMA: €280.70 Type 3 SMA: €216.47 Patient presenteeism: Type 1 SMA: €0 Type 2 SMA: €105.96 Type 3 SMA: €28.23 |
Mean annual cost of caregiver absenteeism per patient: Type 1 SMA: €1543.66 Type 2 SMA: €2431.26 Type 3 SMA: €2237.62 |
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|
The Lewin Group, Inc. (2012)a [60] USA (USD, 2010) |
Hours of daily care required, n (%): Early-onset SMA: 1 to < 8 h: 0 (0) 8 to < 16 h: 0 (0) 16–24 h: 15 (81.45) Not available: 2 (18.55) Other SMA: Care not needed: 13 (5.92) 1 to < 8 h: 36 (15.66) 8 to < 16 h: 42 (15.98) 16–24 h: 145 (49.68) Not available: 26 (12.75) |
Mean family loss of income (in the past 12 months at time of survey)b: Early-onset SMA: $17,759 [€14,568.64] Other SMA: $13,252 [€10,871.04] |
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|
Pena-Longobardo et al. (2020) [76] Multi-national (France, Germany and the UK) [EUR, 2014] |
Mean daily number of hours of informal care per patient: UK: 12.5 France: 10.65 Germany: 9.31 |
Mean annual cost of informal care per patient (SD): Main carer: UK: €27,012 (€43,826) [unable to inflate without GBP value] France: €17,468 (€22,550) [€17,716.34; SD €22,870.59] Germany: €27,436 (€30,060) [€27,922.24; SD €30,592.75] Other carers: UK: €13,516 (€25,151) [unable to inflate without GBP value] France: €8151 (€16,583) [€8266.88; SD €16,818.76] Germany: €12,490 (€17,050) [€12,711.36; SD €17,352.17] |
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|
Rowell et al. (2020) [52] UK (NA) |
Mean number of hours of unpaid care per week received by patients with SMA in previous 12 months (SD): Overall: 102.9 (60.4) Patients unable to sit even with support: 112.1 (63.7) Patients able to sit with support: 132.0 (51.9) |
Mean days off from educationc because of sickness over previous 12 months (SD): 15.8 (12.3) Mean days taken off employmentd for reasons relating to SMA in previous 12 months (SD): 6.7 (9.2) [0 days for patients who were ‘able to walk with support’ or ‘able to walk without support for at least 5 steps’] |
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|
Sampson and Garau (2019) [67] Multi-national (NA) |
The average caregiving hours for a patient with Type 1 SMA was around 13 h per day | |||||||
|
SMA UK, (2019) [98] UK (GBP, NR) |
Weekly number of unpaid caregiving hours per patient with SMA by health state: Walks unaided: 66c Stands unaided: 19c Stands/walks with assistance: 57d Sits and crawls with hands and knees: 189c Sits and rolls independently: 138 Sits without support but cannot roll: 97 Cannot sit without support: 117 |
Mean annual cost for loss of productivity per unpaid caregiver (based on reducing their hours by 25 h per week): £14,350 [€14,894.82] | ||||||
|
Zuluaga-Sanchez et al. (2019) [61] Sweden (SEK, 2018) |
Annual costs per patient, lost productivity of caregivers: Early-onset SMA: SEK 547,475 [€44,957.52] Later-onset SMA: SEK 140,290 [€11,520.33] Later-onset SMA with advanced motor milestones: SEK 35,073 [€2880.12] |
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EUR Euro, GBP Great British Pound, NA not available/applicable, NR not reported, SD standard deviation, SEK Swedish Krona, SMA spinal muscular atrophy, USD United States Dollar
aStudy commissioned by the Muscular Dystrophy Association and was based on US 2010 data
bFamily income loss was calculated by first estimating the expected family income given all the predicting factors under the circumstances where no care (due to disease) is needed. Then, the predicted family income loss due to intensive care was estimated by calculating the difference between actual self-reported family income and expected family income when patients do not require any care
cA total of 42/53 (79%) patients aged 18 years or younger were receiving education whilst 11/53 (21%) aged 18 years or younger were not
dA total of 30 (25%) patients were unemployed, 20 (16%) were in paid part-time employment, 13 (11%) were in paid full-time employment, 4 (3%) worked part time voluntarily, 1 (1%) worked full time voluntarily and 6 (5%) had ‘other’ employment status
eAuthors of original study recommend that these data are interpreted with caution because of a low sample size (n ≤ 10)