Table 3.
Included studies evaluating hearing screening.
| Reference | Model Type | Setting, Population, HL Type | Screening Strategies | Screening tests included | Time Horizon, Perspective | Main Outcome | Main Cost-Effectiveness Findings* | Quality Score |
|---|---|---|---|---|---|---|---|---|
| Neonatal Screening | ||||||||
| Beswick et al. 2019 | Decision Tree | Australia/NZ, Children, Unclear | A. Newborn CMV screening | A. Salivary CMV PCR | 18 years, Modified Societal | Infants detected | HA treatment cost $60.00/QALY gained compared to no treatment, while HA + audiologic rehabilitation cost only $31.91/QALY gained compared to no treatment. | 26 |
| Chiou et al. 2017 | Markov | Asia, Children with SNHL | A. No screening B. Universal screening |
A. TEOAE B. AABR |
Lifetime, Societal | QALY | At willingness to pay of $20,000, aABR had a 90% probability of being cost-effective against TEOAE. | 94 |
| Heidari et al. 2017 | Decision Tree | Middle East, Children, SNHL and CHL | A. Universal screening | A. AABR B. OAE C. Clinical ABR |
1 year, Payer | Infants diagnosed | Over 1 year, the AABR device cost $103,400 less than the OAE device, and detected 800 more cases than the OAE device. | 87 |
| Prusa et al. 2017 | Decision Tree | UK/Europe, Children, Unclear | A. No screening B. Bimonthly toxoplasmosis screening |
A. Toxoplasma antibody or PCR tests B. Amniocentesis + PCR |
Lifetime, Societal | NA | The model calculated total lifetime costs of €103 per birth under prenatal screening as carried out in Austria, saving €323 per birth compared with No-Screening. Without screening and treatment, lifetime societal costs for all affected children would have been €35 million per year. | 100 |
| Rivera et al. 2017 | Markov | Asia, Children, Unclear | A. No screening B. Universal Screening |
A. OAE B. ABR |
Lifetime, Payer and Societal | DALY | Community-based universal newborn hearing screening was found to be cost saving. | 72 |
| Wong et al. 2017 | Decision Tree | South America, Children with b/l SNHL | A. Universal screening B. Screening at the regional health center (RHC) C. Targeted screening D. Screening at the RHC plus targeted screening |
A. OAE | 10 years, Health System | DALY | OAE screening was cost-effective without treatment (CER/GDP=0.06–2.00) and with treatment (CER/GDP-0.58–2.52). | 49 |
| Gantt et al. 2016 | Decision Tree | United States, Children with SNHL | A. No screening B. Targeted cCMV screening C. Universal cCMV screening |
NA | Lifetime, Payer | CMV-related hearing loss identified, CI prevented | The cost of identifying 1 case of hearing loss due to cCMV ranged from $27,460 - $90,038 for universal screening, and $975 - $3916 for targeted screening. | 90 |
| Vallejo-Torres et al. 2015 | Decision Tree | UK/Europe, Children, Unclear | A. No screening B. Biotinidase deficiency screening |
NA | Lifetime, Payer | QALY | Newborn biotinidase deficiency screening was CE at $24,677/QALY in Spain. | 100 |
| Williams et al. 2015 | Decision Tree | UK/Europe, Children with SNHL | A. No screening B. Targeted cCMV screening |
cCMB salivary antigen test | NR, Health System | Case identified, CMV-related SNHL improved | The cost per case of cCMV-related SNHL identified was £668. The cost per case of cCMV-related SNHL improved was £14,202. | 93 |
| Tobe et al. 2013 | Decision Tree | Asia, Children with SNHL | A. No screening B. Targeted screening C. Universal screening |
A. OAE B. AABR |
Lifetime, Modified Societal | DALY | OAE was the most cost-effective strategy at an average cost-effectiveness ratio of I$13,100 (95% CI: 8400–17,200) per DALY averted. | 72 |
| Burke et al. 2012 | Decision Tree | Asia, UK/Europe Children with SNHL | A. Targeted (high-risk) screening B. Universal screening C. One-stage screen D. Two-stage screen |
A. TEOAE B. AABR |
Lifetime, Health System and Societal | Case detected | Universal screening vs. selective screening had an ICER per case detected of £36,181 ($58,497), and INR 157,084 ($9863) for the UK and India, respectively. One-stage vs. two-stage universal screening had an ICER per case detected of £120,972 ($195,586), and INR 926,675 ($58,183) for the UK and India, respectively. | 100 |
| Huang et al. 2012 | Decision Tree | Asia, Children with SNHL | A. Targeted screening B. Universal screening |
NA | Lifetime, Modified Societal | DALY | Targeted strategy tended to be cost-effective in Guangxi, Jiangxi, Henan, Guangdong, Zhejiang, Hebei, Shandong, and Beijing from the level of 9%, 9%, 8%, 4%, 3%, 7%, 5%, and 2%, respectively; while universal strategy trended to be cost-effective in those provinces from the level of 70%, 70%, 48%, 10%, 8%, 28%, 15%, 4%, respectively. | 82 |
| Langer et al. 2012 | Decision Tree | UK/Europe, Children with SNHL | A. Newborn screening alone B. Newborn screening + tracking program |
A. TEOAE B. AABR C. OAE |
Unclear, Payer | Case detected | The ICER of tracking vs. no tracking was €1697 per additional case of bilateral hearing impairment detected. | 93 |
| Schopflocher et al. 2007 | Decision Tree | Canada, Children with SNHL and CHL | A. One-stage screening B. Two-stage screening |
A. AOAE B. AABR |
Other, Societal | Proportion of newborns whose hearing status is correctly identified | 1-stage AABR was more cost-effective than 1-stage AOAE. 2-stage (AOAE followed by AABR) cost $7574.78 additional to correctly identify one additional infant. | 100 |
| Merlin et al. 2007 | Decision Tree | Australia/NZ, Children, Unclear | A. No screening B. Targeted screening C. Universal screening D. One-stage screening E. Two-stage screening |
A. OAE B. AABR |
Immediate, Societal | Infant identified | In the short term, the decision analytic model presented in this report predicted that implementing a two-stage AABR universal neonatal hearing screening (UNHS) program for a cohort of 250,000 newborns would identify an extra 607 infants with unilateral or bilateral hearing impairment by the age of 6 months compared to no formal screening program, at an incremental cost of $6–$11 million. Where a targeted screening program is already in place, expanding to a universal screening program would identify 319 more infants, at an incremental cost of $4–$8 million. | 100 |
| Grill et al. 2006 | Markov | UK/Europe, Children with SNHL | A. Community-based screening B. Hospital-based screening |
NA | 120 months, Health System | Quality weighted detected child months | Both hospital and community programs yielded 794 quality weighted detected child months (QCM) at the age of 6 months with total costs of £3690,000 per 100,000 screened children in the hospital and £3340,000 in the community. | 89 |
| Hessel et al. 2003 | Markov | UK/Europe, Children with SNHL | A. No screening B. Risk screening C. Universal Screening |
A. Two-step TEOAE | 10 years, Health System | Case detected | Cost per case detected: Universal screening = €13,395 Risk screening = €6715 No screening = €4125 |
94 |
| Keren et al. 2002 | Decision Tree | UK/Europe, Children with SNHL | A. No screening B. Selective screening C. Universal screening |
A. TEOAE followed by AABR | Lifetime, Societal | Cost per infant diagnosed by 6 months, cost per deaf child with normal language | The ICER for selective screening vs. no screening was $16,400 per additional infant whose deafness was diagnosed by 6 months of age. The ICER for universal screening vs. selective screening was $44,000 per additional infant whose deafness was diagnosed by 6 months of age. | 100 |
| Boshuizen et al. 2001 | Decision Tree | UK/Europe, Children with Mixed Hearing Loss | A. Home screening B. Child health clinic screening C. Unilateral screening D. Bilateral screening E. Two-stage screening F. Three-stage screening |
A. AABR B. OAE |
NR, Payer | Child detected | Costs of a three-stage screening process in child health clinics were €39.0 (95% confidence interval 20.0 to 57.0) per child detected with automated auditory brainstem response compared with €25.0 (14.4 to 35.6) per child detected with otoacoustic emissions. |
78 |
| Kezirian et al. 2001 | Decision Tree | United States, Children with SNHL | A. One-stage screening B. Two-stage screening |
A. Short-ABR B. OAE |
NR, Provider | Infant identified | Cost per infant with HL identified: 1- S-ABR/S-ABR = $8112 2- S-ABR/None = $9470 3- OAE/OAE= $5113 4- OAE then S-ABR/None= $7996. |
94 |
| Kemper and Downs 2000 | Decision Tree | United States, Children with b/l SNHL | A. Targeted screening B. Universal screening |
A. TEOAE followed by ABR | NR, Health System | Case detected | For every 100,000 newborns screened, universal screening detected 86 of 110 cases of congenital hearing loss, at a cost of $11,650 per case identified. Targeted screening identified 51 of 110 cases, at $3120 per case identified. | 63 |
| Brown 1992 | Decision Tree | UK/Europe, Children, Unclear | A. No screening B. Conventional screening (1st screen at 8–9 months by health visitor, 2nd screen at 10 months by medical officer) C. Screening at 10 months only if clinical indication/concern |
NA | NR, Modified Societal | Unit output | Cost per unit output: - £20.57 for the conventional screening - £11.23 for Alternative policy 1 - £11.23 for Alternative policy 2 - £11.13 for Alternative policy 3 - £11.27 for No screening |
60 |
| Turner 1992 | Decision Tree | United States, Children, Unclear | A. No screening B. Universal screening |
A. High risk register test B. ABR |
NR, Payer | Infant identified | NA | 30 |
| Prager et al. 1987 | Decision Tree | United States, Children | –-NA | A. ABR B. Crib-O-Gram |
Episode of care, Payer | Hearing loss case detected | Crib-O-Gram cost $14,310 per case detected. | 68 |
| Child Screening | ||||||||
| Fortnum et al. 2016 | Decision Tree | UK/Europe, Children with CHL and b/l SNHL | A. No Screening B. School Entry Screening |
A. Pure-tone screen B. HearCheck screen |
Lifetime, Health System | QALY | No screening was dominant over screening. Screening using pure-tone screening was dominant over screening using the HearCheck screener. | 100 |
| Nguyen et al. 2015 | Markov | Australia/NZ, Children with Mixed HL | A. Deadly Ears Program (outreach ENT surgical service and screening program) B. Deadly Ears Program + with mobile telemedicine-enabled screening and surveillance service |
NA | Ages 3–18 to age 50, Societal | QALY | The ICER of MTESS (mobile telemedicine-enabled screening and surveillance) + Deadly Ears vs Deadly ears alone was AUD $656/QALY gained | 94 |
| Baltussen et al. 2012 | Dynamic | Africa, Asia, Children and Adults with Mixed HL | A. Passive Child Screening B. Annual Primary School Screening C. Annual Secondary School Screening D. Annual Primary+Secondary School Screening |
NA | Lifetime, Modified Societal | DALY | The cost per DALY averted was < I$285 for all hearing loss interventions. | 100 |
| Baltussen et al. 2009 | Dynamic | Africa, Asia, Children and Adults, Unclear | A. Passive screening B. Primary School screening C. Secondary school screening D. Primary and secondary school screening E. Adult screening q5 years F. Adult screening q10 years |
Pure tone audiometry | Lifetime, Societal | DALY | Findings showed that in both regions, screening strategies for hearing impairment and delivery of hearing aids cost between I$1000/DALY and I$1600/DALY, with passive screening being the most efficient intervention. Active screening at schools and in the community were somewhat less cost-effective. In the treatment of chronic otitis media, aural toilet in combination with topical antibiotics costs was more efficient than aural toilet alone, and cost between I$11 and I$59/DALY in both regions. The treatment of meningitis with ceftriaxone cost between I$55 and I$217/DALY at low coverage levels, in both regions. | 94 |
| Rob et al. 2009 | Decision Tree | Asia, Children and Adults, Unclear | A. Passive screening and fitting at tertiary care center B. Active screening and fitting at secondary care level |
NA |
5 years, Payer and Modified Societal |
DALY | The cost per DALY averted was around Rs 42,200 (US$900) at secondary care level and Rs 33,900 (US$720) at tertiary care level. | 86 |
| Bamford et al. 2007 | Decision Tree | UK/Europe, Children with Mixed Hearing Loss | A. No Screening B. Targeted School Entry Screening C. Universal School Entry Screening |
A. Pure tone sweep audiometry B. Parental questionnaire C. Tympanometry D. Spoken word test |
1-year, Societal | QALY | Universal school entry screening based on pure-tone sweep tests was associated with higher costs and slightly higher QALYs compared with no screen and other screen alternatives; ICER = £2500/QALY gained. The range of expected costs, QALYs, and net benefits was broad. | 92 |
| Brown, 1992 | Decision Tree | UK/Europe Children, Unclear | A. No Screening B. Conventional screening (1st screen at 8–9 months by health visitor, 2nd screen at 10 months by medical officer) C. Screening at 10 months only if clinical indication/concern |
NA | NR, Modified Societal | Other | Cost per unit output: - £20.57 for the conventional screening - £11.23 for Alternative policy 1 - £11.23 for Alternative policy 2 - £11.13 for Alternative policy 3 - £11.27 for No screening |
60 |
| Adult Screening | ||||||||
| Garcia et al. 2018 | Markov | United States Adults, Unclear | A. No screening B. Annual screening |
NA | 20 years, Payer | Hearing loss avoided | The ICER of a hearing conservation program was $10,657 per case of hearing loss prevented. | 89 |
| Linssen et al. 2015 | Markov | UK/Europe Adult, NR | A. Age at first screening (50, 55, 60, 65, or 70 years) B. Number of repeated screenings (up to five repetitions) C. Time interval between repeated screenings (5 or 10 years). |
A. No screening B. Telephone screening C. Internet screening D. Handheld device (HearCheck) E. Audiometric screening |
Lifetime, Health System | QALY | Incremental costs of the screening strategies compared with no screening ranged from €4 to €59. Incremental QALYs ranged from 0.0003 to 0.0104. The ICERs of all the screening strategies compared with the current practice were below €20,000/QALY gained. | 94 |
| Morris et al. 2013 | Markov | UK/Europe Adults with b/l SNHL | A. One-stage audiometric screen (60–70 y/o adults) B. Two-stage screen (postal questionnaire + audiometric) C. GP Referral |
NA | Lifetime, Health System | QALY | The ICER of one-stage screening for 35 dB HL from 60 years vs. GP referrals was £1461. Two-stage screening was eliminated by extended dominance. | 100 |
| Baltussen et al. 2012 | Dynamic | Africa, Asia, Children and Adults with Mixed HL | A. Passive Adult Screening B. Adult Screening q5 years C. Adult Screening q10 years |
NA | Lifetime, Modified Societal | DALY | The cost per DALY averted was < I$285 for all hearing loss interventions. | 100 |
| Liu et al. 2011 | Other/Unclear | United States Adults with SNHL | NA | A. No Screening B. Tone-emitting otoscope C. Self-administered questionnaire D. Otoscope + questionnaire |
1 year, Payer | Probability of hearing aid use after one year | The tone-emitting otoscope was the most cost-effective strategy, with a significant increase in hearing aid use 1 year after screening (2.8%) and an ICER of $1439.00 per additional hearing aid user compared with the control group. | 52 |
| Morris 2011 | Markov | UK/Europe Adults, Other NR | Twelve screening scenarios that vary according to 1. age at first screen (55, 60 or 65 years), 2. target hearing loss (better ear average ≥30 dB HL or ≥35 dB HL) and 3. one- or two-stage screening program |
NA | Lifetime, Health System | QALY | The ICER of screening compared to GP referral service ranged from £1266 to £2185. | 55 |
| Baltussen et al. 2009 | Dynamic | Africa, Asia, Adults and Children, Unclear | A. Passive screening B. Primary School screening C. Secondary school screening D. Primary and secondary school screening E. Adult screening q5 years F. Adult screening q10 years |
Pure tone audiometry | Lifetime, Societal | DALY | Findings showed that in both regions, screening strategies for hearing impairment and delivery of hearing aids cost between I$1000/DALY and I$1600/DALY, with passive screening being the most efficient intervention. Active screening at schools and in the community were somewhat less cost-effective. In the treatment of chronic otitis media, aural toilet in combination with topical antibiotics costs was more efficient than aural toilet alone, and cost between I$11 and I$59/DALY in both regions. The treatment of meningitis with ceftriaxone cost between I$55 and I$217/DALY at low coverage levels, in both regions. | 94 |
| Rob et al. 2009 | Decision Tree | Asia, Adults and Children, Unclear | A. Active Screening and Fitting at Secondary Care Level B. Passive Screening and Fitting at Tertiary Care Level |
NA | 5 years, Payer and Modified Societal | DALY | The cost per DALY averted was around Rs 42,200 (US$900) at secondary care level and Rs 33,900 (US$720) at tertiary care level. | 86 |
*Main Cost-Effectiveness findings cost-effectiveness ratios and costs are presented in the published currency and year.
Abbreviations: AABR – automated auditory brainstem response, ABR – auditory brain response, b/l: bilateral, AOAE – automatic otoacoustic emissions, cCMV – congenital cytomegalovirus, CE – cost-effectiveness, CHL – conductive hearing loss, CI – cochlear implant, COG – Crib-O-Gram, DALY – disability-adjusted life years, GP- general practitioner, HA – hearing aid, HL – hearing loss, (I)CER – (incremental) cost-effectiveness ratio, NA – not applicable, NR – not reported, OAE - Otoacoustic emissions, PCR – polymerase chain reaction, RHC – regional hearing center, SNHL – sensorineural hearing loss, UK – United Kingdom, US – United States.
Currencies: € – Euro, I$ – International Dollars, INR/Rs – Indian Rupee, £ – Pound.