Systematic Review/Meta-analysis
Otolaryngology–
Cost-effectiveness of School Hearing
Screening Programs: A Scoping Review
Michael Yong, MD1,2, Jiahe Liang, MSEd2, Jeromie Ballreich, PhD2,
Jane Lea, MD1, Brian D. Westerberg, MD, MHSc1, and
Susan D. Emmett, MD, MPH3,4,5
Abstract
Objective. School hearing screening is a public health interven-
tion that can improve care for children who experience hear-
ing loss that is not detected on or develops after newborn
screening. However, implementation of school hearing screen-
ing is sporadic and supported by mixed evidence to its eco-
nomic benefit. This scoping review provides a summary of all
published cost-effectiveness studies regarding school hearing
screening programs globally. At the time of this review, there
were no previously published reviews of a similar nature.
Data Sources. A structured search was applied to 4 data-
bases: PubMed (Medline), Embase, CINAHL, and Cochrane
Library.
Review Methods. The database search was carried out by 2
independent researchers, and results were reported in
accordance with the PRISMA-ScR checklist and the JBI
methodology for scoping reviews. Studies that included a
cost analysis of screening programs for school-aged children
in the school environment were eligible for inclusion.
Studies that involved evaluations of only neonatal or pre-
school programs were excluded.
Results. Four of the 5 studies that conducted a cost-effectiveness
analysis reported that school hearing screening was cost-
effective through the calculation of incremental cost-
effectiveness ratios (ICERs) via either quality- or disability-
adjusted life years. One study reported that a new school
hearing screening program dominated the existing pro-
gram; 2 studies reported ICERs ranging from 1079 to 4304
international dollars; and 1 study reported an ICER of
£2445. One study reported that school-entry hearing
screening was not cost-effective versus no screening.
Conclusion. The majority of studies concluded that school
hearing screening was cost-effective. However, significant
differences in methodology and region-specific estimates of
model inputs limit the generalizability of these findings.
Keywords
hearing loss, hearing screening, cost-effectiveness, school
children, pediatric
Received November 18, 2019; accepted February 21, 2020.
Head and Neck Surgery
2020, Vol. 162(6) 826–838
Ó American Academy of
Otolaryngology–Head and Neck
Surgery Foundation 2020
Reprints and permission:
sagepub.com/journalsPermissions.nav
DOI: 10.1177/0194599820913507
http://otojournal.org
H
earing loss affects approximately 1.3 billion people
worldwide, ranking as the second-leading cause of
impairment among conditions resulting in chronic
disease and injury.1 The impact of unaddressed hearing loss
can be measured across the life span. Unaddressed hearing
loss in early childhood results in speech and language delays,
decreased school performance, and increased risk of dropout,
as well as downstream health effects such as restricted
employment opportunities and earlier onset of cognitive
decline.2-4
In addition to the clinical effects of unaddressed hearing
loss, the economic burden is substantial. The World Health
Organization (WHO) estimates that the overall global cost
of unaddressed hearing loss is between 750 and 790 billion
international dollars (Int$) per year.5 From the perspective
of education and health, conservative models projects a
worldwide annual cost to education systems of Int$3.9 bil-
lion, while annual health care costs contribute a minimum
of Int$67.3 billion. Most of these costs to education and
health systems are incurred in low- and middle-income
countries.
Universal newborn hearing screening has had a major
impact on reducing the burden of hearing loss.6,7 Early iden-
tification of hearing loss in infants plays a key role in miti-
gating the impact of hearing loss on developmental milestones
such as speech and language acquisition.6,7 Furthermore,
studies have shown that newborn hearing screening is
1
BC Rotary Hearing and Balance Centre, Division of Otolaryngology–Head
and Neck Surgery, Faculty of Medicine, University of British Columbia,
Vancouver, British Columbia, Canada
2
Bloomberg School of Public Health, Johns Hopkins University, Baltimore,
Maryland, USA
3
Head and Neck Surgery and Communication Sciences, School of Medicine,
Duke University, Durham, North Carolina, USA
4
Duke Global Health Institute, Durham, North Carolina, USA
5
Center for Health Policy and Inequalities Research, Duke University,
Durham, North Carolina, USA
This article was presented at the Coalition for Global Hearing Health;
October 26, 2019; Tempe, Arizona.
Corresponding Author:
Michael Yong, MD, Division of Otolaryngology–Head and Neck Surgery,
Faculty of Medicine, University of British Columbia, Fourth Floor, 2775
Laurel St, Vancouver, BC V5Z 1M9, Canada.
Email: m.yong@alumni.ubc.ca
Yong et al
cost-effective, prompting policy makers to push for wide-
spread adoption.8 Universal newborn hearing screening is
now standardized across most high-income countries, and
efforts to expand implementation to other parts of the world
continue.7,9-11
However, even in areas with established neonatal screen-
ing programs, childhood hearing loss still remains an under-
addressed public health concern.12,13 Conditions that are not
covered at the time of initial newborn screening include pro-
gressive congenital conditions, drug-related ototoxicity, infec-
tion, cerumen impaction, noise exposure, and otitis media with
effusion.14-16 Of these, otitis media with effusion remains a
persistent cause of hearing loss in young children, especially in
low- and middle-income countries and rural areas.17-21
School hearing screening could help address the gap in
preventative care occurring after the period of newborn
hearing screening, leveraging access to a large proportion of
the total child population in one physical location while uti-
lizing integrated educational infrastructure, such as school
nurses, special education programs, and school-facilitated
follow-up.5 However, unlike neonatal hearing screening, the
current state of school hearing screening adoption is incon-
sistent across regions, and there remains wide variability in
the protocols used.16,22-24 In addition, the small amount of
evidence that currently exists surrounding the cost-effectiveness
of school hearing screening programs is inconclusive, poten-
tially impeding widespread adoption.5,13,25 These considera-
tions are evident in the WHO’s 2017 recommendations,
which highlight the need for additional country-specific data
on the cost of unaddressed hearing loss and associated
interventions.5
This review provides an up-to-date summary of all peer-
reviewed evidence in the literature pertaining to economic
evaluation of school-based childhood hearing screening pro-
grams. While a preliminary search for reviews revealed an
article by Sharma et al8 evaluating childhood hearing
screening programs including neonates and infants, the cur-
rent study differs in that it offers the first exhaustive sum-
mary of cost analyses associated exclusively with school-
based hearing screening programs.
Methods
Protocol
A documented a priori protocol was constructed in accor-
dance to the Joanna Briggs Institute methodology, as well
as the PRISMA-ScR checklist (Preferred Reporting Items
for Systematic Reviews and Meta-analyses Extension for
Scoping Reviews).26,27 Modifications were made to these
reporting guidelines where appropriate for this review. This
protocol is available in Appendix 1 (available online).
Eligibility Criteria
The search strategy was not limited to any specific study
designs. Included studies involved a population of otherwise
healthy children aged 4 to 19 years who received hearing
screening as part of a school program. A formal economic
827
analysis of any sort on a school hearing screening program
was required for inclusion in this review. Types included
cost descriptions, cost analyses, cost-outcome descriptions, or
full economic evaluations.28 Studies that involved evaluations
of only neonatal or preschool programs were excluded.
Information Sources
A structured search was performed according to the protocol
devised in 4 scientific databases: PubMed (Medline), Embase,
CINAHL, and the Cochrane Library. The search was per-
formed in February 2019 and not limited to any publication
date, language, or geographic region. After full-text review,
the reference lists from all eligible studies were examined for
any additional relevant studies that met inclusion criteria. A
gray literature search via Google and Google Scholar was
also performed (Figure 1).
Search Strategy
The following terms were used in title, abstract, and key-
words searches: ‘‘hearing loss,’’ ‘‘hearing disorders,’’ ‘‘hearing
impairment,’’ ‘‘screening,’’ ‘‘audiometry,’’ ‘‘cost effective-
ness,’’ ‘‘cost-benefit,’’ ‘‘school,’’ and ‘‘child.’’ Full search
algorithms for each database can be found in Appendix 2
(available online). The search strategy was devised by 2
study authors (M. Yong and J. Liang) in consultation with a
professional informationist from Johns Hopkins University.
Study Selection Process
Two study authors (M. Yong and J. Liang) independently
conducted the initial database searches. After duplicate arti-
cles were eliminated, all article titles and abstracts were
reviewed independently for inclusion according to the elig-
ibility criteria. Discrepancies regarding inclusion or exclu-
sion were resolved through discussion. Full-text review was
then performed on the remaining studies in a similar
manner. Neither reviewing author was blinded to the journal
titles, individual study authors, or research institutions.
Data-Charting Process
Data from individual articles were extracted and compiled
with Microsoft Excel. Data collection forms were modified
from the Joanna Briggs Institute’s recommendations for
data charting.26
Data Items/Outcomes
All eligible articles were assessed for the age range of chil-
dren screened in the school hearing screening program,
study region, components of the screening protocol, esti-
mated disease prevalence, type of economic analyses per-
formed, and cost perspective. The primary outcome of
interest was whether the school hearing screening program
assessed in the study was deemed cost-effective, as sup-
ported by a cost-effectiveness ratio or incremental cost-
effectiveness ratio (ICER). Discount rates for monetary
amounts were reported when available. No additional dis-
counting or inflation adjustment to the present year was
performed.
828
Figure 1. Search strategy.
Method of Synthesis of Results
Qualitative analyses of individual study data were per-
formed by comparing the outcomes of interest, where appli-
cable. Quantitative analyses and meta-analyses were not
performed in this review due to the heterogeneity of eligible
studies and paucity of available data on cost-effectiveness.
Quality of Evidence
Each cost-effectiveness analysis included in the review was
assessed against the CHEERS checklist (Consolidated Health
Economic Evaluation Reporting Standards).29 The CHEERS
checklist is a tool developed by the International Society for
Pharmacoeconomics and Outcomes Research consisting of 24
items that set guidelines for the reporting of the title and
abstract, introduction, methods, results, discussion, funding,
and conflicts of interest. The items were scored binarily as
‘‘concerns’’ or ‘‘no concerns,’’ and each study was assigned
an aggregate adherence score. The nature of any methodolo-
gic concerns pertaining to each study was described.
Results
Selection of Sources of Evidence
After removal of duplicates, 552 studies were identified for
title and abstract review. An additional 2 studies from refer-
ence lists and gray literature searches were included. The
exclusion of 545 studies left 9 studies for full-text review.
Two additional articles on full-text review were excluded
due to not meeting the inclusion criterion of performing a
formal economic analysis. A final total of 7 articles were
included in this review.13,25,30-34
Populations
Five studies focused solely on school-aged children, while 2
studies carried out a combined approach including school
Otolaryngology–Head and Neck Surgery 162(6)
children and adults (Table 1). One study in Oman and both
studies in the United Kingdom focused on hearing screening
occurring at the time of primary school entry, with 2 studies
in China and Australia examining school hearing screening
for all school-aged children. The remaining 2 studies in
Africa and Southeast Asia evaluated screening of adults and
children.
Type of Economic Analysis and Models
Five of the 7 studies performed a cost-effectiveness analysis.
The other 2 studies included a cost analysis and cost descrip-
tion studies. The only cost-effectiveness analysis to draw
input data from a prospective trial was by Fortnum et al.13
All articles that performed a cost-effectiveness analysis
constructed a model to integrate within-study data and inputs
from outside sources. Two of these used a decision tree
model. Two studies constructed models based on the WHO-
CHOICE methodology (WHO–Choosing Interventions That
Are Cost-effective), which uses the population model
PopMod.35 The remaining study used a Markov model.
Time horizons for these models varied from 1 year to the
lifetime of the child.
Screening Protocols
Methods of screening interventions were highly variable
among studies and, in some cases, unspecified (Table 2).
Information on the personnel carrying out the screening
was unspecified in 2 studies. The other 5 studies made use
of various combinations of traveling nurses and physicians,
audiologists, primary school teachers, school nurses, and
Indigenous health workers. Except in the United Kingdom—
where school entry screening is carried out by local school
staff, as with Bamford et al33 and Fortnum et al13—specially
trained screening teams were created for the purpose of tra-
veling to schools to administer screening.
Yong et al 829

Horizon, y
Data on the location of screening were variable. Bamford

Lifetime
Time

1, 6, 11
et al33 gave no information regarding the testing environ-

100
NA

NA
ment other than it occurred in the school. Fortnum et al13

10

4
performed screening in a quiet school room while ensuring
standardized ambient sound levels. Nguyen et al34 used a
PopMod (WHO-CHOICE)

PopMod (WHO-CHOICE)
specially equipped mobile van to visit schools, providing all
screening within the van test environment. The other 4 stud-
ies provided no information on the screening environment.
Model

Markov model
Screening procedures also varied significantly in terms of
Decision tree

Decision tree
tests performed, screening algorithms, audiometric frequen-
cies tested, referral thresholds, definition of hearing loss,
and frequency of screening. Pure tone screening audiometry
NA

NA

was the most frequently used screening test, with tympano-

metry, otoscopy, and subjective questionnaires being used
systematic review and retrospective data
Cost analysis and prospective clinical data

to a lesser extent. Fortnum et al13 also made substantial use

Cost description and prospective clinical

systematic review, retrospective data,

Cost-effectiveness / cost-utility analysis

of the HearCheck screener, a preprogrammed handheld
Cost-effectiveness analysis based on

Cost-effectiveness analysis based on

Cost-effectiveness analysis based on

Cost-effectiveness analysis based on

screening tool for hearing loss. In addition, the frequencies

used for pure tone screening were widely variable, with 4
based on retrospective data
Type of Study

studies not specifying any frequencies that were screened

and with the rest of the studies indicating varying frequency
and prospective data

ranges (0.5-8 kHz). Testing and referral threshold levels

retrospective data

retrospective data

were also variable, with only 3 studies indicating test levels,

Abbreviations: NA, not applicable; WHO-CHOICE, World Health Organization Choosing Interventions That Are Cost-effective.

ranging from 20 to 70 dB, and 3 studies listing threshold

levels for diagnostic referral, ranging from 20 to 30 dB.
Last, the 3 studies that used a definition for hearing impair-
data

ment agreed on a threshold .30 dB.

Rescreening procedures were included by 2 studies and
generally not well described. Aasham et al30 indicated that
children who were referred on initial screening were rescreened
‘‘First-year preparatory pupils’’

approximately 2 weeks later before formal referral for fur-

Adults and schoolchildren

Adults and schoolchildren

ther diagnosis and treatment. Bamford et al33 indicated that

Age Range, y

children were rescreened up to 2 times in the model before

being referred for further care.
4-6 (school entry)

3-6 (school entry)

The proportion of children following up for subsequent

diagnostic testing and treatment after being referred on hear-
combined

combined

ing screening was reported by 3 studies. Aasham et al30

reported that 14.9% of children referred for diagnosis and
5-14

3-18

treatment completed their follow-up appointment. Meanwhile,

Bamford et al33 and Fortnum et al,13 both in the United
Africa and Southeast Asia

Africa and Southeast Asia

Kingdom, reported much higher follow-up rates, from 80% to

85%.
United Kingdom

United Kingdom

Hearing Loss Prevalence and Screening Accuracy

Region

Estimates of hearing loss prevalence varied substantially

Australia

according to region and method of calculation, with many

Oman

China

studies using literature-derived numbers instead of within-

study data. Aasham et al30 in Oman showed that 0.74% of
screened children were eventually referred to an ear-nose-
Table 1. Study Characteristics.

Baltussen and Smith32 (2012)

Baltussen and Smith31 (2009)

throat specialist for hearing impairment. The 2 studies con-

ducted by Baltussen and Smith31,32 in Africa and Southeast
Baltussen et al25 (2009)

Fortnum et al13 (2016)

Bamford et al33 (2007)
Aasham et al30 (2004)

Nguyen et al34 (2015)

Asia quoted prevalence estimates of hearing loss in children

between 1.04% and 4.1% based on previous studies and
WHO Global Burden of Disease estimates.36 The study by
Baltussen et al25 in China estimated a prevalence of hearing
loss at 0.01% based on the number of children referred on
Study

screening. Two studies in the United Kingdom by Bamford

et al33 and Fortnum et al13 used prevalence estimates of
 Table 2. Screening Protocols.

830
Hearing Loss
Study Administering Personnel Testing/Environment Screening Procedures Follow-up procedures Prevalence Screening Accuracy

Aasham et al30 Traveling nurses and 10 Equipment: calibrated Procedure: special team visits Rescreen: First failure: repeat 0.74%. Source: Unspecified
(2004) physicians who were battery-operated micro- schools, conducts screening, test in 2 wk. Second failure: study data on
trained by an ENT audiometer, cuffed refers pupils for follow-up care refer. Procedure: parents total number of
specialist headphones, otoscope. after. Screening: 1, 2, 4 kHz at contacted by school, advised to children referred
Tests: audiometry, 25 and 35 dB. Refer threshold: consult with ENT specialist (for
otoscopy. Environment: unspecified. Hearing study purposes, compliance on
unspecified impairment: unspecified. referral at 2 mo was reviewed).
Frequency: 1 time Follow-up rate: 14.9%
Baltussen and Trained primary health Equipment: unspecified. Procedure: annual screening of Rescreen: unspecified. Children: 1.04%- True- to false-positive
Smith32 (2012) workers (assumption) Tests: unspecified. schoolchildren. Screening: Procedure: children are 2.15%. Adults: ratio: 1:3.6. Source:
Environment: unspecified. Refer threshold: referred to secondary-level 1.47%-3.73%. previous study by
unspecified unspecified. Hearing hospital for further Total: 3.62%- Limburg et al (1995)
impairment: unspecified. consultation with audiologist. 4.77%. Source:
Frequency: annually Follow-up rate: unspecified WHO Global
Burden of
Disease (2004)
Baltussen and Trained primary health Equipment: unspecified. Procedure: screening team Rescreen: unspecified. Children: 3.7%- 1 false positive per 1 true
Smith31 (2009) workers (assumption) Tests: unspecified. drives to schools and delivers Procedure: unspecified. 4.1%. Adults: positive. Source:
Environment: screening. Screening: Follow-up rate: unspecified 5.7%-32%. assumption
unspecified unspecified. Refer threshold: Source: previous
unspecified. Hearing studies by Smith
impairment: .30 dB in better- and Mathers
hearing ear. Frequency: annual (2006) and
Mathers et al
(2005)
Baltussen et al25 Audiologists and trained Equipment: unspecified. Procedure: initial screening Rescreen: unspecified. 0.01%. Source: Unspecified
(2009) hearing health care Tests: unspecified. questionnaire, then flagged Procedure: children with study data on
workers (primary Environment: children selected for further potential hearing loss flagged total number of
school teachers who unspecified in-person screening at schools. at school and referred to children referred
underwent a 3-wk Screening: unspecified. Refer consultation rehabilitation
training program on threshold: .30 dB. Hearing centers for further testing and
basic hearing health impairment: .30 dB in possible hearing aid fitting.
care, audiometry, ear frequency range of 0.5-4 kHz Comparisons were made
mould impressions, and in better-hearing ear. between referral centers (1
hearing aid fitting) Frequency: 1 time. Otherwise primary care center vs 2
unspecified tertiary care centers).
Follow-up rate: unspecified

(continued)
 Table 2. (continued)
Hearing Loss
Study Administering Personnel Testing/Environment Screening Procedures Follow-up procedures Prevalence Screening Accuracy

Bamford et al33 Trained school nurses Equipment: unspecified. Procedure: children are Rescreen: children rescreened 0.35%. Source: Sensitivity: audiometry:
(2007) Tests: Pure tone sweep screened in the first year of up to 2 times in the model study data 86%-100%. Parental
audiometry (PTS), school by school nurses. before being referred. questionnaire: 34%-71%.
otoscopy, Screening: 0.25-8 kHz at 25 Procedure: children are Spoken words: 51%-
tympanometry, parental dB. Refer threshold: .20 dB. referred on to the community 100%. Otoscopy: 23%-
questionnaire. Hearing impairment: variable. GP or ENT specialist. Follow- 89%. Tympanometry:
Environment: Frequency: upon school entry up rate: 80% 40%-90%. Specificity:
unspecified audiometry: 65%-100%.
Parental questionnaire:
52%-95%. Spoken
words: 93%-97%.
Otoscopy: 60%-93%.
Tympanometry: 57%-
97%. Source: systematic
review of previous
studies
Fortnum et al13 Trained school nurses Equipment: PTS by Procedure: screening is carried Rescreen: failure on initial 0.46%. Source: Sensitivity: PTS: 95.9%.
(2016) Amplivox (Eynsham, out by school staff. Screening: screen led to direct referral study estimates HC: 88.7%. Specificity:
UK) and HC screener PTS: 0.5, 1, 2, 4 kHz. HC: 1 for diagnostic PTA. Procedure: from Nottingham, PTS: 79.8%. HC: 83.8%.
(Siemens, Frimely, UK). kHz at 20, 35, 55 dB; 3 kHz at coordinated between school UK Source: study data with
Tests: audiometry. 35, 55, 75 dB. Referral nurse screeners and local PTA as reference
Environment: quiet threshold: PTS: .20 dB. HC: audiology establishments for standard
school room with responses to \6 tones. further diagnostic testing and
ambient sound levels Hearing impairment: .30 dB evaluation by ENT specialists.
measured at any 1 of 4 frequencies. Follow-up rate: 85%
Frequency: upon school entry
Nguyen et al34 Indigenous health worker Equipment: audiometer, Procedure: van is driven to local Rescreen: unspecified. Indigenous: 7%-8%. Specificity: 90%. Source:
(2015) with advanced hearing- video-otoscope, community centers, primary Procedure: screening reviewed Non-Indigenous: expert opinion
health training tympanometer. Tests: schools, and high schools to by the specialist ENT team, 3%. Source:
audiometry, service Indigenous children. and it provides a diagnosis and Australian Bureau
tympanometry, Screening: unspecified. Refer treatment plan. Surgical of Statistics
otoscopy. Environment: threshold: unspecified. Hearing treatment is referred to the
mobile testing van impairment: unspecified. surgical outreach clinic, and
Frequency: biannual medical treatment is referred
to the local community GPs.
Follow-up rate: unspecified
Abbreviations: ENT, ear-nose-throat; GP, general practitioner; HC, HearCheck; PTS, pure tone screen.

831
832
0.35% and 0.46%, respectively, based on regional study
data. Finally, Nguyen et al34 reported that in Australia, the
prevalence of childhood hearing loss in Indigenous children
was as high as 7% to 8%.
Screening test sensitivity and specificity were explicitly
reported by the 2 studies in the United Kingdom. Bamford
et al33 attributed the highest sensitivity to pure tone screen-
ing at 86% to 100%. This estimate was based on several
previous literature studies. Fortnum et al13 conducted a pro-
spective trial and found a sensitivity of 88.7% to 95.9% and
a specificity of 79.8% to 83.8% depending on whether pure
tone screening or HearCheck screening was used. Nguyen
et al34 reported using a presumed screening test specificity
of 90% and an unspecified sensitivity, with both estimates
based on expert opinion. The 2 studies in Africa and
Southeast Asia by Baltussen and Smith31,32 estimated true-
to false-positive ratios based on previous literature and
assumptions. The remaining 2 studies did not report sensi-
tivity or specificity of the screening tests.
Study Perspective
The 2 studies by Aashem et al30 and Nguyen et al34 adopted
a strictly health care perspective, measuring costs incurred
by the health care system as a result of screening and subse-
quent treatment. The study by Aasham et al, however,
included direct health care costs, without considering subse-
quent costs for diagnostic and treatment care.
Three other studies reported using a societal perspective.
Specifically, Baltussen and Smith,31,32 in Africa and
Southeast Asia, followed a previous cost approach devel-
oped by WHO-CHOICE that included fixed and variable
costs incurred by patients and health care systems.35
Bamford et al33 described using an approximation of a soci-
etal perspective but did not include indirect or intangible
costs, such as lost income for families or child care costs
during or after treatment.
Two studies considered mixed perspectives. Baltussen
et al25 in China used health care and societal perspectives,
which included indirect costs. Fortnum et al13 used health care
and patient perspectives to evaluate costs; however, patient-
related costs were limited to transportation expenditures.
Health Utilities
Of the 5 studies that conducted a formal cost-effectiveness
analysis, 2 chose to measure utility with the number of
disability-adjusted life years (DALYs) averted. The remain-
ing 3 studies used quality-adjusted life years (QALYs)
gained. These data are shown in Table 3.
None of the studies carried out evaluations that directly
measured utility values from children. Instead, all the stud-
ies derived utility inputs from previous validated literature,
such as the Health Utilities Index and EQ-5D, expert opin-
ion, and modeling assumptions. Additionally, where utility
estimates were based on previous literature, most utility
weights were derived from hearing-impaired adult popula-
tions, often estimated from single studies.
Otolaryngology–Head and Neck Surgery 162(6)
The raw amount of QALYs gained or DALYs averted in
each study was affected by variations in time scales, such as
measurements per year up to measurements per 10 years, as
well as differing assumptions, including screening coverage
and treatment compliance proportions. These inherent dif-
ferences in methodology made QALY and DALY compar-
ability difficult.
Discounting was usually applied to utility values at a rate
from 3% to 5%. The only study to omit reporting on dis-
counting of utility measures was Bamford et al.33
Costs
Costs were reported in different local currencies, except
where the study design followed WHO-recommended meth-
odology and the values were reported in Int$. Discounting
to the study date of publication was performed in all cost-
effectiveness analyses at a rate from 3% to 5%.
Two studies, Baltussen et al25 in China and Aasham
et al30 in Oman, examined costs without health utilities and
reported the average cost of screening per child, as well as
the cost per referred or treated child.
The 5 cost-effectiveness studies reported substantially
different cost estimates that were largely dependent on the
unit division of costs and the periods over which costs were
calculated. For example, Baltussen and Smith31 in Africa
and Southeast Asia displayed costs per 10 years of screen-
ing as a total cost listed in millions of Int$. Meanwhile,
Bamford et al,33 Fortnum et al,13 and Nguyen et al34 each
displayed costs per child screened, although even these
values were substantially different.
The sources for cost estimates generally came from
national statistical databases and local purchasing data, as
well as previous literature sources and expert opinion as
needed.
Individual Study Conclusions
Overall, 4 of the 5 cost-effectiveness studies concluded that
school hearing screening was cost-effective. This was gener-
ally based on either a standard regional willingness-to-pay
(WTP) threshold or a comparison of the ICER with a multi-
plier of average income per capita. However, the methodology
of screening that qualified as cost-effective was inconsistent
among studies.
Baltussen and Smith32 measured various combinations of
hearing screening among children and adults in Africa and
Southeast Asia at differing screening coverage percentages. In
Africa, passive screening of all children and adults at a pre-
sumed screening coverage of 80% dominated other forms of
hearing screening. In Southeast Asia, annual screening of pri-
mary and secondary school students, combined with screening
of adults every 5 years at presumed screening coverages of
80% and 95%, dominated all other combinations of screening
strategies. Most notably, any screening programs that involved
screening schoolchildren alone were dominated by the com-
bined screening approaches of children and adults described
earlier, and probabilistic sensitivity analysis showed that these
 Table 3. Economic Analyses.
Study Health Utilities Costs Cost-effectiveness Ratios Sensitivity Analysis Author Conclusions

Aasham et al30 NA Screening cost per child: US$5. NA NA Screening does not seem cost-
(2004) Overall cost per referred effective due to low
child: US$4700. Discount: prevalence of ear problems,
None and feasibility of using primary
health center staff is low.
Baltussen and Measurement: DALYs. Factors Annual cost per capita. Africa: Passive screening of all children Type: Probabilistic uncertainty Passive screening of children and
Smith32 (2012) considered: Geographic Passive screening: Int$1.28. 1° and adults: Africa ICER: analysis (MCLeague software). adults, as well as annual
coverage of interventions, school only: Int$1.71. 2° Int$766. SE Asia ICER: Variations: Costs and effects screening of schoolchildren
attendance for screening school only: Int$ 0.81. 1° and Int$892. (Given 80% screening 625% of baseline. Results: and screening of adults every 5
(assumed 70%), compliance 2° school: Int$2.28. SE Asia: coverage) Annual screening of Remained either very cost- y is ‘‘very cost-effective’’ under
with treatment including Passive screening: Int$1.04. 1° 1° and 2° school and adults effective or cost-effective the WHO guidelines of less
hearing aids (assumed 70%). school only: Int$ 0.62. 2° every 5 y: Africa ICER: than average income per capita
Annual DALYs averted (per school only: Int$ 0.40. 1° and Int$1089. SE Asia ICER:
million). Africa: Passive 2° school: Int$ 0.89. Discount Int$1063. (Given 80%
screening: 1719. 1° school rate: 3% screening coverage) Annual
only: 1400. 2° school only: screening of 1° and 2° school
731. 1° and 2° school: 2091. and adults every 5 y: Africa
SE Asia: Passive screening: ICER: Int$3639. SE Asia ICER:
1207. 1° school only: 501. 2° Int$4340. (Given 95%
school only: 372. 1° and 2° screening coverage) All other
school: 850. Discount rate: active school screening
3%. Source: WHO-CHOICE strategies were dominated.
methodology
Baltussen and Measurement: DALYs. Millions Cost over 10 y (in millions). Africa: Passive screening: Type: Univariate and School screening at secondary
Smith31 (2009) of DALYs averted (over 10 y). Africa: Passive screening: Int$998. ICER (1° school multivariate sensitivity analysis. schools only or primary and
Africa: Passive screening: 0.07. Int$692. 1° school only: only): Dominated. ICER (2° Variations: Discounting DALYs, secondary schools together is
1° school only: 0.58. 2° school Int$920. 2° school only: school only): Int$1347. ICER age weighing, catchment area ‘‘cost-effective’’ under the
only: 0.31. 1° and 2° school: Int$416. 1° and 2° school: (1° and 2° school): Int$1472. per screening worker, ratio of WHO guidelines of \3 times
0.87. SE Asia: Passive Int$1248. SE Asia: Passive SE Asia: Passive screening: false positives to true average income per capita.
screening: 1.99. 1° school only: screening: Int$2093. 1° school Int$1053. ICER (1° school positives, useful life of hearing
0.83. 2° school only: 0.61. 1° only: Int$1086. 2° school only: only): Dominated. ICER (2° aids. Results: No change of
and 2° school: 1.40. Discount Int$661. 1° and 2° school: school only): Int$1079. ICER study conclusions under
rate: 3%. Source: WHO- Int$1,607. Discount rate: 3% (1° and 2° school): Int$1201 parameters that were varied
CHOICE methodology

(continued)

833
834
Table 3. (continued)
Study Health Utilities Costs Cost-effectiveness Ratios Sensitivity Analysis Author Conclusions

Baltussen et al25 NA Cost per child fitted with hearing NA NA Costs are lowest when doing
(2009) aids: Beijing. Health care: follow-up and fitting hearing
US$306. Societal: US$365. aids in a primary care setting
Nanning. Health care: US$257. vs tertiary care setting
Societal: US$277. Guigang.
Health care: US$209. Societal:
US$227. Discount: 3.5%
Bamford et al33 Measurement: QALYs. Expected Cost per child. 1-y horizon: No ICER (1-y horizon): £2445 Type: Probabilistic sensitivity School-entry hearing screening
(2007) QALYs of screening (1-y screening: £0.22. Screening: analysis and CEACs. is more cost-effective than no
horizon): No screening: 0.979. £10. 6-y horizon: No Variations: Expected costs, screening, although
Screening: 0.983. Expected screening: £2. Screening: £25. QALYs, WTP thresholds. considerable uncertainty due
QALYs of screening (6-y 11-y horizon: No screening: Results: 1-y horizon: to wide estimates makes
horizon): No screening: 5.27. £3. Screening: £30. Prices Probability of screening being results indicative and
Screening: 5.37. Expected standardized to 2004. cost-effective is 90% under exploratory. Targeted school
QALYs of screening (11-y Discount rate: 3.5% WTP of £30,000 and 50% screening may be more cost-
horizon): No screening: 8.91. under WTP of £2250. 6-y effective than universal school-
Screening: 9.07. Discount rate: horizon: Probability of entry screening
None. Source: Systematic screening being cost-effective
review, previous utility is .99% under WTP of
literature on adults (Barton £2000. 11-y horizon:
et al 2004, Dolan et al 1995) Probability of screening being
and expert opinion cost-effective is .99% under
WTP of £2000. Range of
expected costs, QALYs, and
net benefits was broad,
indicating significant
uncertainty
Fortnum et al13 Measurement: QALYs. QALY Cost per child: PTS: £1.93. HC No screening dominates PTS Type: Threshold analysis. School hearing screening is not
(2016) gain over 4 y: No screening: screener: £2.49. Total costs: and HC screener screening Variations: Sensitivity of cost-effective in the UK vs no
35.9. PTS: 35.21. HC screener: No screening: £182,333. PTS: screening, prevalence of screening within the
35.16. Discount rate: 3.5%. £209,637. HC screener: hearing loss, number of constraints posed by the
Source: Previous utility £215,323. Discount rate: 3.5% referrals generated. Results: model
literature on adults (Barton No change in cost-
et al 2004, Summerfield et al effectiveness conclusions
2002) and modeling
assumptions

(continued)
Yong et al 835

Abbreviations: CEAC, cost-effectiveness acceptability curve; DALY, disability-adjusted life year; HC, HearCheck; ICER, incremental cost-effectiveness ratio; NA, not available; PTS, pure tone screen; QALY, qual-
interventions remained either very cost-effective or cost-

screening is cost-effective vs
Mobile telemedicine-enabled
effective under the WHO definition. An earlier study by
Author Conclusions

Baltussen and Smith,31 focusing more on school and commu-

nity screening in Africa and Southeast Asia, showed that pas-
the status quo sive screening, secondary school screening, and primary and
secondary school screening were all cost-effective as compared
with no screening and other alternate screening combinations
and that these combinations dominated screening at primary
schools only. Sensitivity analyses did not alter the authors’
conclusions.
Bamford et al33 found that with a 1-year time horizon
probability of acceptable ICER

Screening rate achieved: 75%-

specificity, treatment effects,
analysis. Variations: Hearing

ICER of £2445 under a WTP threshold of £30,000, school-

Screening cost per child: Status Starting at age 3: ICER: A$656. Type: Probabilistic sensitivity

loss prevalence, screening

under $50,000 per QALY.

coverage, screening test
Sensitivity Analysis

entry hearing screening was more cost-effective than no

treatment costs, utility
weights. Results: 98%

screening. This also held true across 6- and 11-year time

ity-adjusted life year; SE, Southeast; WHO-CHOICE, World Health Organization Choosing Interventions That Are Cost-effective; WTP, willingness to pay. horizons. However, there was considerable uncertainty due
to the wide range of expected costs, QALYs, and net bene-
fits, and it was felt that targeted school screening may be
more cost-effective than universal school hearing screening.
Fortnum et al13 found that no screening dominated both
90%

screening methods used in the trial and that sensitivity anal-

ysis supported the robustness of the conclusions. However,
referral rates for children with potential hearing loss were
Cost-effectiveness Ratios

Starting at age 4 y: ICER:

actually higher in the region where only ad hoc surveillance

A$117. Annual equivalent cost: Screening is dominant.

was performed (34.4 per 1000) as compared with the region

where school entry screening was implemented (21.9 per
1000). Furthermore, screening did not result in a signifi-
cantly higher number of true cases of hearing loss (3.04 vs
2.51 per 1000). Lastly, the authors noted that by varying the
baseline referral rate in the model, there were 2 scenarios
where school-entry screening became cost-effective.
The last study, by Nguyen et al,34 evaluated school hear-
ing screening in Australian Indigenous schoolchildren. The
Status quo: A$78,243. New
quo: A$88. New screening:

authors found that screening administered by their mobile

screening: A$236,200.

van unit was cost-effective in comparison with the status

quo at an ICER of A$656. In addition, changing the starting
Discount rate: 5%
Costs

age of screening from 3 to 4 years made screening the

dominant choice. Probabilistic sensitivity analysis showed a
98% probability that the acceptable ICER was under a WTP
threshold of A$50,000.
Quality of Evidence
Overall, the reporting quality of the studies was good with
Previous utility literature on
adults (Clemens et al 2014)
QALYs: Status quo: 15.902.

Discount rate: 5%. Source:

Measurement: QALYs. Total

regard to cost-effectiveness methods and data reporting

New screening: 15.944.

(Supplemental Table S1, available online). On average,

Health Utilities

cost-effectiveness analyses reported 88% of CHEERS items

with adequate detail. The main concern was that many stud-
ies failed to include adequately detailed explanations of the
hearing screening protocols themselves, such as information
on audiometric frequencies, thresholds used for referral, and
rescreening procedures.
Table 3. (continued)

Discussion
Summary of Evidence
Nguyen et al34

School hearing screening programs represent a unique

opportunity to reduce the health and economic burden of
(2015)
Study

childhood hearing loss. The results of this review indicate

that school hearing screening is a potentially valuable
836
public health intervention that should be further considered,
lending support to the current literature surrounding the ben-
efits of childhood hearing screening after the newborn
period, including recommendations from the WHO.5,12,37-39
However, the substantial variability found in the existing lit-
erature base plays a key role in the interpretation of results.
Screening test accuracy has a notable impact on assump-
tions in cost-effectiveness models and can be highly depen-
dent on many factors, such as the level of ambient noise,
screening referral thresholds, combination of tests adminis-
tered, and rescreening protocol.15,16,23,38 False positives due
to varying amounts of ambient noise, differing frequency
and intensity thresholds for referral, and the combination of
tympanometry, otoscopy, and otoacoustic emission testing
have all been shown to affect overall screening sensitivity
and specificity.12,16,23,38 Moreover, when a child is referred
upon initial screening, rescreening is a valuable step in the
protocol, reducing the amount of false-positive referrals for
further audiometric assessment.14,16
The sources from which studies based their probability and
utility assumptions are important to examine. Primary survey
evaluation of utility estimates related to childhood hearing loss
were missing from all studies, presumably due to the signifi-
cant logistical challenges associated with this task in the pedia-
tric population.40 Instead, studies used utility estimates based
on systematic literature reviews or single studies. However,
many of these sources represented response data from older
adults completing standardized quality-of-life questionnaires
after interventions such as cochlear implantation. The effect of
these assumptions is generally not well studied and indicates a
need to improve estimates of hearing-related utility measures
among the pediatric population.41
Cost perspective was also a source of considerable varia-
bility. Although several studies adopted a societal perspec-
tive, the patient costs that were reported were often
inconsistent among studies.35 Health care cost inclusion was
variable as well, illustrated by the scenario where Fortnum
et al13 excluded the cost of postoperative care in the case of
surgical treatment while Nguyen et al34 did not specify
whether these postoperative costs were included.
Fortnum et al13 were the only authors to find that school
hearing screening was not cost-effective as compared with
no screening. This was a study conducted strictly on hearing
screening at the time of school entry. In addition, the high
baseline referral rate of the ad hoc surveillance system in
the United Kingdom indicates that school hearing screening
may not yield substantial additional health utility while
requiring significant costs, making screening in this circum-
stance an economically unattractive intervention. However,
it is unlikely that ad hoc surveillance referral rates would be
as high in low- and middle-income countries as those in the
United Kingdom.
Limitations
As noted in the quality-of-evidence assessment against the
CHEERS checklist, several studies failed to include detailed
explanations of the hearing screening protocols. This is a
Otolaryngology–Head and Neck Surgery 162(6)
1. Inclusion of detailed hearing screening protocols
with particular attention to re-screening and
follow-up procedures.
2. More robust evaluation of the sensitivity and
specificity of the chosen hearing screening tests.
3. More accurate estimates of regional prevalence of
hearing loss based on epidemiologic data.
4. Quantification of the number of patients lost to
follow-up.
5. Clearly state cost perspectives and consistent
inclusion of all applicable costs.
6. Elucidation of pediatric-specific preferences used
to determine measures of benefit, which may
differ from those extrapolated from adults.
Figure 2. Future economic evaluation research needs.
significant gap because screening test characteristics play an
important role in the overall assumptions that contribute to
uncertainty in the cost-effectiveness model. A lack of ade-
quate detail in screening protocol description may also
make it difficult to replicate the findings of previous cost-
effectiveness analyses.
Another substantial limitation of this review was the
small number of studies that performed a cost-effectiveness
analysis. Results in this review were specific to the respec-
tive regions of Africa, Australia, Southeast Asia, and the
United Kingdom and are likely to have limited usefulness
when extrapolated to other populations. Several of these
study authors cautioned that further investigation into the model
assumptions and more robust primary data are needed.13,33,34
As such, the conclusions found here should not be used to dic-
tate policy in other geographic regions where prevalence of
hearing loss, accuracy of screening, and the current state of ad
hoc surveillance may differ greatly.
In addition, the general conclusion that school hearing
screening is cost-effective in the majority of studies must be
considered within the context of highly heterogeneous meth-
odology, including differences in screening protocols, included
costs, and sources of model parameter inputs. These inconsis-
tencies make direct comparison of study conclusions difficult.
Implications for Research
The small number of cost-effectiveness analyses on school
hearing screening programs, as well as the lack of standardi-
zation of study design, indicates a need for more research to
establish standards for school hearing screening cost-
effectiveness analysis and gather more region-specific esti-
mates of costs and utilities. Figure 2 presents a list of
future economic evaluation research needs identified as a
result of this review.
Yong et al
Implications for Practice
The results of this review should be interpreted with caution.
When one considers whether to implement a school hearing
screening program in any setting, reasonable estimates of
region-specific disease prevalence, screening test accuracy,
and costs are all needed to inform an accurate assessment of
the cost-effectiveness of the proposed intervention.
Conclusions
The results of this review indicate that school hearing
screening is cost-effective overall. However, the small
number of studies and significant differences in study meth-
odologies make it difficult to extrapolate these conclusions
to other geographic regions that are considering the imple-
mentation of school hearing screening programs.
Acknowledgments
Donna Hesson reviewed and edited the search strategy.
Author Contributions
Michael Yong, project inception, search algorithm, data extraction,
data analysis, manuscript writing and editing; Jiahe Liang, search
algorithm, data extraction, data analysis; Jeromie Ballreich,
manuscript editing critical for intellectual content; Jane Lea,
manuscript editing critical for intellectual content; Brian D.
Westerberg, manuscript editing critical for intellectual content;
Susan D. Emmett, project inception, search algorithm, manuscript
editing critical for intellectual content.
Disclosures
Competing interests: None.
Sponsorships: Michael Yong is supported by the University of
British Columbia Clinician Investigator Program.
Funding source: None.
Supplemental Material
Additional supporting information is available in the online version
of the article.
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