. Author manuscript; available in PMC: 2020 Jun 8.

Published in final edited form as: Appl Health Econ Health Policy. 2019 Jun;17(3):331–357. doi: 10.1007/s40258-018-00456-1

Table 5.

Incremental costs and outcomes

Economic evaluation	Country, currency, price year	ICER Intervention arm	Authors’ conclusions
Prager et al. (1987) [21]	USA, USD, price year not reported	Incremental cost per true positive case detected—compared to ABR: COG: $22,591	ABR more cost effective
Brown (1992)^b [22]	UK, GBP, 1986	Alternative policy (in terms of unit output)—Incremental cost per unit output per screened child a Reference b. £12.47 c. £10.22 Conventional: dominated No screening not considered due to lack of information	Screening for clinical indication at 10 months cost effective
White et al. (1995) [23]	USA, USD, 1993	Incremental cost per true positive case detected: UNHS: $979 Targeted screening: Dominated	UNHS more cost effective
Friedland et al. (1996) [24]	USA, USD 1995	Incremental cost per true positive case detected: Base case model: $7936.83 Government: Dominated	Screening at other hospitals than at Mount Sinai Hospital cost effective
Kemper et al. (2000) [25]	USA, USD, NR	Incremental cost per true positive case detected: $23,930	UNHS more cost effective
Kezirian et al. (2001) [26]	USA, USD, 1999	Incremental cost per true positive case detected: S-ABR/S-ABR: $8112 S-ABR/None: $9470 OAE/OAE: $5113 OAE + S-ABR/None: $7996	2- stage OAE/OAE most cost effective strategy
Boshuizen et al. (2001)^b [27]^pp	Netherlands, USD, Price year not clear	Incremental cost per true positive case detected—compared to OAE-2C(B): OAE-3C(B + U): $1846,429 OAE-2C + H(B + U): $208,841 OAE-2C(B + U): dominated OAE-2H(B + U): $759,315 AABR-2C(B + U): dominated On considering only bilateral and unilateral cases (i.e. after excluding the bilateral only cases) Compared to OAE-3C(B + U): OAE-2C + H(B + U): $208,841 OAE-2C(B + U): dominated OAE-2H(B + U): $759,315 OAE-3C(B + U): dominated	3- stage OAEs cost effective [Inconclusive about home vs. child health clinic screening]
Vohr et al. (2001) [28]	USA, USD, 1998	Incremental cost per true positive case detected: AABR: $1749 AABR + TEOAE: $754	AABR more cost effective
Keren et al. (2002)^b [29]	USA, USD, 2001	Incremental cost per true positive case detected by 6 months: $16,000	UNHS more cost effective
Herrero & Monero-Ternero (2002)^b [30]	Spain, USD, Price year not reported	Incremental cost utility ratio: TNHS1: $177 TNHS2: $165 TNHS3: $174 UNHS1: $670 UNHS2: $818 UNHS3: $730 (Strategy with the lowest ICER recommended)	TNHS more cost effective: If direct costs only considered UNHS more cost effective: if indirect costs also considered
Hessel et al. (2003)^b[31]	Germany, Euro, 1999	Incremental cost per true positive case detected UNHS: €13,395 TS: €6715	UNHS more cost effective
Lin et al. (2005) [32]	Taiwan, USD, 2004	Incremental cost per true positive case detected: AABR + TEOAE: $917	AABR + TEOAE^Ȝ
Grill et al. (2006) [33]	UK, GBP, 2002	Incremental costs per true positive case detected: £2423 Incremental cost per QCM: £25	Hospital and community-based strategies equally cost effective
Uus et al. (2006)^b [34]	UK, GBP, 2003	Incremental cost per true positive case detected: £12,526	UNHS more cost effective
Schnell-Inderst et al. (2006) [35]	Germany, Euro, 2004	Incremental cost per true positive case detected (compared to no screening): Targeted: €5201.05 UNHS: €34,463.42	Targeted screening more cost effective
Schopflocher et al. (2007)^b [36]	Canada, CAD, NR	Incremental cost per true positive case detected Compared to 1-stage AABR 1-stage AOAE: dominated 1-stage vs. 2-stage***: $7575	AABR more cost effective
Merlin et al. (2007)^b [37]	Australia, AUD, 2003	For birth cohort of 4000 infants/year: ICER per true positive case detected Compared to no screening: UNHS (OAE-AABR)^p: $9300 UNHS (OAE-AABR)^c: $10,100 UNHS (AABR)^p: $12,500 UNHS (AABR)^c: ($17,600) Compared to TS UNHS 2- stage (OAE-AABR^p: $8800 UNHS 2- stage (OAE-AABR^c: $9500 UNHS (AABR)^p: $14,600 UNHS (AABR^c: $23,800	Short-term cost effectiveness of UNHS may be misleading. May be cost effective in the long-term
Lin et al. (2007)^b [38]	Taiwan, USD, 2005	Incremental cost per true positive case detected: TEOAE: $61,525 AABR + TEOAE: $531	AABR more cost effective
Porter et al.³ (2009)^b [39]	USA, USD, 2004	No definite results given, Suggests that benefits outweigh costs by the ratio of 25:1 when high benefit and low costs are considered	UNHS more cost effective
Olusanya et al. (2009) [40]	Nigeria, USD, price year not reported	Incremental cost per true positive case detected—compared to community-UNHS: Community-TNHS: $1221 Other strategies dominated If no screening strategy with $0 cost and 0 effect was considered (as reference), the Community-UNHS would have ICER: $26,809	Community-based screening more cost effective
Uilenburg et al. (2009) [41]	Netherlands, USD, price year not reported	Incremental cost per true positive case detected—compared to A: B: $79,688 C: Dominated	Home screening including metabolic diseases (B) more cost effective
Burke et al. (2012)^1b [42]	UK, GBP, 2010	Incremental cost per true positive case detected: £36,181 (Health system) ^Ə−296,857 (societal) (cost-saving)	UNHS more cost effective
Burke et al. (2012)^1b [42]	India, INR, 2010	INR-157,084 per true positive case detected ^ƏSocietal −INR 8418,834 (cost saving)
Burke et al. (2012)^2b [42]	UK, GBP, 2010	Incremental cost per true positive case detected: £120,972	Not explicitly reported
Burke et al. (2012)^2b [42]	India, INR, 2010	INR 926,675 per true positive case detected
Huang et al.^a (2012)^b [43]	China, RMB, USD, 2009	ICER per DALYs averted for UNHS: Ranged from $18,000 for Guangdong to $500,000 for Guangxi TS: Ranged from $4000 for Guangdong to $83,000 for Guangxi	UNHS and TS both demonstrated cost effectiveness in rich provinces; TS in poor provinces
Tobe et al. (2012)^b [44]	China, USD, 2009	Compared to TS (OAE): UNHS OAE: $55,000 OAE + AABR: $43,000 TS OAE + AABR: $127,000	OAE + AABR more cost effective
Fortnum et al. (2016)^b [45]	UK, GBP, 2012–2013	PTS vs. no screening: PTS less effective and more costly HC vs. no screening: HC less effective and more costly HC vs. PTS: PTS more effective and less costly	No screening more cost effective
Chiou et al. (2017)^b [46]	Taiwan, USD, NR	TEOAE vs. no screening: TEOAE less costly more effective AABR vs. no screening: AABR less costly more effective AABR vs. TEOAE: $6723 per QALY gained	AABR more cost effective
Chen et al.^a (2017)^b [47]	China, RMB and USD, 2012	Short-term cost–benefit ratio: 1:2.01 Long-term cost–benefit ratio: 1:7.52	UNHS more cost effective
Heidari et al. (2017) [48]	Iran, USD, NR	AABR vs. OAE: AABR less costly more effective	AABR more cost effective
Rivera et al.^a (2017)^b [49]	Philippines, Pesos, 2015	Incremental cost per DALY gained: PhP 105,376^ȼ	UNHS more cost effective

Calculation of incremental cost per true positive case detected involved the following steps: First, detected cases/screened (Yield) was obtained by dividing cost per screening by cost per cases detected. Then, the incremental cost per screening (incremental cost) was divided by incremental yield (incremental outcome) to obtain the incremental cost per true positive case detected

AABR Automated Auditory Brainstem Response, AOAE Automated Otoacoustic Emissions, AUD Australian dollars, CAD Canadian Dollars, COG Crib-O-Gram, DALY disability-adjusted life year, DPOAE Distortion Product Otoacoustic Emissions, GBP Pound Sterling, HC HearCheck, ICER incremental cost-effectiveness ratio, INR Indian Rupees, NA not applicable, NHS National Health Service, NR not reported, PhP Philippine Pesos, PTS pure tone screening, QALY quality-adjusted life year, QCM quality-weighted detected child months, RMB Renminbi, S-ABR Stacked Auditory Brainstem Response, TEOAE Transient Evoked Otoacoustic Emissions, TNHS Targeted Newborn Hearing Screening, TOAE Transient Otoacoustic Emissions, UNHS Universal Hearing Screening, USD United States Dollars

^Ə

Combined societal costs for Burke et al.¹ and Burke et al.²

^ȼ

Calculated using formula $ICER = \frac{(cost of UNHS - Cost of NS)}{(DALY loss of UNHS - DALY loss of NS)}$ . Authors preferred to carry out sensitivity analysis surrounding the ICER instead of calculating the ICER itself. NS = no screening

Sensitivity of protocol given instead of the number of infants

Total instead of mean costs and outcomes reported

Considered to have included downstream resource use: (i) treatment of hearing loss; (ii) long-term productivity; and (iii) education impacts

Using couplers

Using probe tips

^pp

Acronyms used include: OAE-2C for 2-stage OAE at Clinic, OAE-2C + H: 2-stage OAE at home and clinic; B + U = Bilateral and unilateral; B: Bilateral only

Universal vs. Targeted

1-stage vs. 2-stage

Cost-effective alternative was decided based on the fact that TEOAE + AABR leads to reduced additional diagnostic testing. Total cases considered in the model was 100,000. □See Table 2 for cost types included