IJCA-28913; No of Pages 7

International Journal of Cardiology xxx (2020) xxx

Contents lists available at ScienceDirect

International Journal of Cardiology

journal homepage: www.elsevier.com/locate/ijcard

Economic consequences of rheumatic heart disease: A scoping review

Chinonso C. Opara a, Negeen Aghassibake b, David A. Watkins a,c,⁎
a
Department of Medicine, University of Washington, Seattle, WA, USA
b
Health Sciences Library, University of Washington, Seattle, WA, USA
c
Department of Global Health, University of Washington, Seattle, WA, USA

a r t i c l e i n f o a b s t r a c t

Article history: Background: Rheumatic heart disease (RHD) remains endemic in less-resourced regions and countries and results
Received 8 June 2020 in high medical and non-medical costs to households, health systems, and society. This scoping review maps out
Accepted 7 September 2020 the available evidence on the economic impact of RHD and its antecedents and suggests future research priori-
Available online xxxx ties.
Method: We followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Extension for
Keywords:
Scoping Reviews. We identified articles through systematic electronic database search supplemented by expert
Rheumatic fever
Rheumatic heart disease
knowledge of unpublished literature. Studies were included if they collected empirical RHD-related costing
Costs and cost analysis data as a primary or secondary objective and if the data were collected from 2000 onward. Main quantitative
findings by intervention, costing perspective, and location were charted, and a standardized quality assessment
tool was used to appraise included studies.
Results: The index search identified 2519 electronic records and two grey-literature graduate theses. Six full texts
were included in the review. Primary prevention costs were modest, while secondary and especially tertiary pre-
vention were more costly. Most estimates were of health sector costs and for tertiary interventions. Only two
studies described RHD-related costs in non-high-income countries. Most studies were of adequate methodolog-
ical quality.
Conclusion: Research into the costs of RHD has mostly been done in wealthy countries. Costs from the household
perspective, which are particularly important in countries with limited public healthcare finance, are lacking. To
inform advocacy and guide implementation of the 2018 World Health Assembly resolution on RHD, high-quality,
local cost estimates will be needed from a range of representative, RHD-endemic countries.
© 2020 Elsevier B.V. All rights reserved.

1. Introduction
Over the past two decades, the incidence of acute rheumatic fever
(ARF) and chronic rheumatic heart disease (RHD) have dramatically de-
clined in wealthier regions of the world as a result of prevention pro-
grams, improved living standards, and access to cardiac surgery.
Nevertheless, ARF and RHD are still public health problems in less-
developed regions of the world such as Oceania, South Asia and Sub-
Saharan Africa [1].
Due to its chronic nature, RHD care implies high costs that persist
over time. These costs include direct medical and non-medical costs re-
lated to valve surgery and use of long-term medications, as well as lost
wages and time spent on medical care. These costs could be especially
devastating to households in poorer countries where RHD is endemic.
⁎ Corresponding author at: University of Washington, 325 9th Ave, Box 359931, Seattle,
WA 98104, USA.
E-mail address: davidaw@uw.edu (D.A. Watkins).
Over the last decade, increasing evidence from less-developed countries
has shown how households affected by chronic illness can get stuck in a
“poverty trap” due to repeated disruptions in household income, intra-
household labor substitution, and loss of assets [2].
Research into the economic consequences of RHD and its anteced-
ents (streptococcal pharyngitis and rheumatic fever) can potentially
aid governments in allocating resources for RHD prevention and treat-
ment and mitigating the household effects mentioned previously. Yet
much of the research on RHD prevention and treatment efforts, includ-
ing cost and cost-effectiveness evidence, was conducted decades ago
and in relatively more-developed settings [3]. The 2018 World Health
Assembly resolution on RHD generated a new urgency to critically re-
view existing data on the cost of RHD-related care and determine prior-
ities for future research in this area [4].
Since we expected the literature on the cost of RHD to be diverse and
heterogeneous, we conducted a scoping review in order to systemati-
cally map research done in this area and identify existing gaps in knowl-
edge. Our review sought to identify all relevant empirical information

https://doi.org/10.1016/j.ijcard.2020.09.020
0167-5273/© 2020 Elsevier B.V. All rights reserved.

Please cite this article as: C.C. Opara, N. Aghassibake and D.A. Watkins, Economic consequences of rheumatic heart disease: A scoping review, In-
ternational Journal of Cardiology, https://doi.org/10.1016/j.ijcard.2020.09.020
C.C. Opara, N. Aghassibake and D.A. Watkins
on the economic burden of RHD from the health sector, household, and/
or societal perspective.
2. Methods
We followed the Preferred Reporting Items for Systematic Reviews
and Meta-Analyses Extension for Scoping Reviews (PRISMA-ScR) [5].
A completed PRISMA-ScR checklist is included in Appendix A.
We sought to identify all empirical studies that estimated the cost
(s) of one or more components of RHD-related healthcare from any per-
spective. By “empirical” we mean that the cost(s) estimates were devel-
oped predominately based on data collected from patient surveys,
clinical records, and/or administrative records containing patient-level
data (e.g., insurance claims databases). As such, unit cost estimates de-
veloped principally for the purpose of conducting economic evaluations
were generally excluded. For example, a cost-minimization analysis
that cited “average” unit costs from administrative databases [6], or a
cost-effectiveness analysis that combined literature estimates with pa-
tient surveys focused solely on out-of-pocket costs [7], would both be
excluded from this review.
“RHD-related healthcare” was defined as any intervention that spe-
cifically addressed risk factors for RHD (streptococcal pharyngitis or
rheumatic fever) or addressed disease outcomes (such as heart failure,
stroke, or need for valve surgery). Studies were eligible for inclusion if
they met the following criteria: (i) the study reported at least one esti-
mate of the cost of RHD-related healthcare (as a primary or secondary
objective of the study); (ii) the study produced this estimate using em-
pirical methods and collection of primary data (as noted above); and
(iii) the study's data were collected in or after the year 2000. The latter
was justified because standards of clinical care for ARF and RHD have
changed substantially over the past several decades; further, older stud-
ies do not provide data relevant to current policy and advocacy efforts
[8,9]. We excluded non-English publications, reviews, case reports, edi-
torials, opinion articles, or commentaries.
The search was conducted on March 29, 2019 by one author (NA).
Databases included were: MEDLINE/PubMed, Embase, EconLit,
Cochrane Library Reviews, and NHS-EED. The search strategy included
terms related to rheumatic heart disease and to economic costs (see Ap-
pendix B for full search strategy). After duplicates were removed, one
author (CCO) independently selected publications with pertinent titles
and abstracts. Potentially relevant full text articles were reviewed by
two authors (CCO and DAW) and included by consensus. We also
reviewed reference lists of full-text studies identified through the data-
base search. Although we did not systematically search global academic
databases for published theses, we were aware of two master's theses
recently published the University of Cape Town, so we included these
manuscripts in the review. The selection process was managed using
Zotero 5.0.66® software.
One reviewer (CCO) used a standardized data extraction form to
gather information on study characteristics and cost estimates. When
necessary, corresponding authors were contacted for further costing
data. We charted the data using eight distinct clinical categories: strepto-
coccal pharyngitis treatment, management of ARF, provision of secondary
prophylaxis, RHD screening and diagnosis, chronic medical management
of RHD, medical treatment of sequelae such as heart failure and stroke,
surgical management of RHD, and interventional management of RHD
(e.g., percutaneous transvenous mitral commissurotomy).
The data charting exercise also considered other study characteris-
tics such as costing perspective and country/region studied. In some
cases, simple calculations were made using published study data so
that we could present comparable cost estimates (e.g., annual cost of
chronic medical management per participant) across similar studies.
Topline cost estimates from each study were standardized to 2017 US
dollars using methods published previously and exchange rate and con-
sumer price index data from the World Bank World Development Indi-
cators data (April 2019 update) [10].
2
International Journal of Cardiology xxx (2020) xxx
To date, no tools or guidelines have been published that specifically
assess the quality of costing studies. Drummond and colleagues suggest
a “checklist” for economic evaluation studies, but their checklist is con-
cerned principally with the evaluation component of the study, and
only a few items speak to the conduct of the costing itself [11]. More re-
cently, the Global Health Cost Consortium produced a draft “reference
case” that specified standards for costing study design, conduct, and
reporting [12]. The 17 items in this reference case were adapted into a
checklist (see appendix C); the possible quality score for a study
would thus range from zero to 17 out of 17.
The sponsor of this research was not involved in the design of the re-
view, collection of data, analysis and interpretation of data, or drafting of
the report.
3. Results
Fig. 1 presents a flow diagram of the study selection process. 2519
records were identified on the index bibliometric search, and two re-
cords were identified through an alternate source (theses from the Uni-
versity of Cape Town that were known previously to the authors). After
removal of duplicates and screening of titles and abstracts, we reviewed
60 candidate full text articles. Of these, 54 were excluded for the follow-
ing reasons: 27 insufficient information on costing methods and/or re-
sults; 15 not costing studies; four reviews; three commentaries; three
with primary data collection prior to 2000; one editorial; and one
study not published in English. The remaining six studies were included
this review. Two master of public health theses were included; the re-
maining four studies were published in peer-reviewed journals.
Characteristics of included studies are listed in Table 1. Two studies
focused largely on the economic aspects of group A streptococcal phar-
yngitis prevention and treatment [13,16]. Cannon et al. estimated the
health and economic burden of several group A streptococcus-
attributable diseases in Australia as part of a modeling study to assess
the potential value of a new vaccine. Pfoh et al. described the morbidity
and medical and non-medical costs associated with streptococcal phar-
yngitis in school-aged children at two outpatient pediatric clinics in the
city of Boston.
The remaining studies focused mostly on the costs of RHD treat-
ments [9,14,15,17]. Milne reviewed ARF and RHD hospitalization costs
in New Zealand in children and adults. Studies by Hellebo, Oyebamiji,
and Seo looked at chronic outpatient costs in addition to hospitalization
costs in South Africa and South Korea.
Table 2 summarizes the main quantitative findings from each study.
Three studies estimated costs from a health sector perspective [9,13,14].
Only one study (Cannon et al.) reported costs of managing acute phar-
yngitis and of secondary prevention; the other studies (as well as
Cannon et al) reported costs of ARF and RHD hospitalization, including
valve surgery. Three studies estimated costs from a household perspec-
tive: Cannon et al. for acute pharyngitis, and Seo et al. and Oyebamiji for
chronic medical management of RHD [13,15,17]. Finally, one study
(Pfoh et al.) estimated costs from a societal perspective, evaluating the
cost for acute pharyngitis [16]. There were no studies assessing the
cost of RHD screening, nor were there studies assessing cost of percuta-
neous interventions.
Table 3 illustrates gaps in evidence on costs across the spectrum
of RHD-related care by World Bank region. The economic burden of
primary, secondary and tertiary RHD care are described in East Asia
and the Pacific, though only in high-income countries in this region.
North America and sub-Saharan Africa are represented by studies
detailing cost of either primary or tertiary prevention, respectively.
However, the only data from sub-Saharan Africa come from South
Africa, an upper-middle-income country with declining rates of
RHD. We did not find any studies in Europe and Central Asia, Latin
America and the Caribbean, Middle East and North Africa, or South
Asia.
C.C. Opara, N. Aghassibake and D.A. Watkins International Journal of Cardiology xxx (2020) xxx

Fig. 1. Flow diagram of study selection.

Table 1
Studies included in this review.

Author Start year Study Location Setting Methods Costing endpoints Quality
of data duration score
collection

Cannon April 10 years Australia outpatient, emergency retrospective analysis using Streptococcal pharyngitis, ARF, secondary prophylaxis, 13/17
2018 2005 department, and administrative databases RHD chronic medical management, RHD acute medical
[13] inpatient centers management, RHD surgery
Hellebo January 1 year South governmental tertiary retrospective analysis using RHD chronic medical management, RHD acute medical 15/17
2017 2017 Africa care center patient charts, administrative management
[14] databases and surveys
Milne January 10 years New inpatient centers retrospective analysis using RHD surgery 11/17
2012 [5] 2000 Zealand patient charts and
administrative databases
Oyebamiji January 14 months South governmental tertiary cross-sectional cohort analysis RHD chronic medical management, RHD acute medical 16/17
2018 2017 Africa care center management
[15]
Pfoh 2008 October 4 months United two pediatric retrospective telephone Streptococcal pharyngitis 15/17
[16] 2005 States outpatient centers interviews
Seo 2013 2008 1 year South outpatient and retrospective analysis using RHD chronic medical management, RHD acute medical 10/17
[17] Korea inpatient centers administrative databases management, RHD surgery

3
C.C. Opara, N. Aghassibake and D.A. Watkins International Journal of Cardiology xxx (2020) xxx

Table 2
Economic cost of RHD by intervention category and cost perspective.

Intervention category Costa perspective

Health sector Household Societal

Primary Streptococcal pharyngitis
Secondary ARF treatment
Secondary prophylaxis
Tertiary RHD screening and
diagnosis
RHD chronic medical
management
RHD acute medical
management
RHD surgery
Catheter-based
interventions for RHD
US$ 55 per episode [13] US$ 39 per episode [13]
US$ 260 per episode
- US$ 150 medical costs
- US$ 110 non-medical
costs [16]
US$ 6200 per episode for
non-indigenous child
US$ 6600 per episode for
non-indigenous adult
US$ 9000 per episode for indigenous
child
US$ 9700 per episode for indigenous
adultb [13]
US$ 1300 per non-indigenous patient
US$ 1500 per indigenous patient [13]
– – –
US$ 1600 per non-indigenous patient US$ 470 per patient for direct costs
US$ 2300 per indigenous patient [13] US$ 300 per patient for indirect costs [17]
US$ 990 per patient [14] US$ 0 per patient for direct medical costsd
US$ 21 per patient for direct non-medical costs US$ 32 per patient
for indirect costs [15]
US$ 5800 per episode for
non-indigenous child
US$ 4500 per episode for
non-indigenous adult
US$ 8800 per episode for indigenous
child
US$ 7300 per episode for indigenous
adult [13]
US$ 190 per medical & ICU episode US$ 0 per episode for direct medical costs
US$ 2000 per catheterization lab US$ 31 per episode for direct non-medical costs US$ 28 per
episode [14] episode for indirect costse [15]
US$ 48,000 per episode for
non-indigenous child
US$ 46,000 per episode for
non-indigenous adult
US$ 56,000 per episode indigenous
child
US$ 54,000 per episode indigenous
adult [13]
US$ 12,000 per episode [14]
US$ 39,000 per ARF/RHD surgical valve
admissionc [9]
– – –

Notes: (a) All costs are in 2017 US dollars and reported to two significant digits regardless of the of the estimates as reported in the studies; (b) authors stratified costs according to
indigeneity in the Australian population; (c) authors did not disaggregate surgical costs associated with ARF and RHD; (d) public healthcare is generally free for low-income clients in
South Africa; (e) authors did not disaggregate costs based on surgical and non-surgical admissions, but most episodes were non-surgical.

4. Discussion
Table 3
Empirical evidence on the cost of RHD by world region and spectrum of care. This scoping review sought to map out the current evidence on the
economic consequences of RHD and its antecedents worldwide. We
Wolrd Bank region Spectrum of RHD care
found only six empirical studies; four peer-reviewed studies were con-
Primary Secondary Tertiary ducted in high-income settings in North America and East Asia and the
East Asia and Pacific Cannon 2018 Cannon 2018 Cannon 2018 Pacific, and two masters' theses were conducted in an upper-middle-
Milne 2012 income setting with reasonable access to advanced cardiac care. We
Seo 2013 did not identify any studies conducted in low- or lower-middle-
Europe and Central Asia – – –
Latin America and the Caribbean – – –
income settings in Africa or Asia, where the burden of RHD is estimated
Middle East and North Africa – – – to be the highest [1].
North America Pfoh 2008 The range of cost estimates in this study is consistent with evidence
South Asia – – – from other health areas. For example, costs of primary care interven-
Sub-Saharan Africa Oyebamiji 2018
tions for cardiovascular disease are on the order of US$ 10–100 per
Hellebo 2018
year (or per episode, in the case of screening), whereas costs of acute in-
Notes: Studies in italics represent high-income countries as defined by the World Bank for terventions are on the order of $1000–100,000 per episode [10]. When a
the year 2017.
household perspective on costs is taken, direct non-medical costs and
indirect costs of RHD together appear to contribute significantly to

4
C.C. Opara, N. Aghassibake and D.A. Watkins
total costs, a finding consistent with other cost-of-illness studies for
chronic diseases [18].
The health sector perspective on costs is relatively well-represented
among the studies in this review; less is known about the household im-
pact of RHD prevention and care. Evidence on the latter is particularly
important for low- and middle-income countries as they seek to expand
their portfolios of interventions in universal health coverage systems.
Health interventions that are less attractive from a cost-effectiveness
standpoint (such as heart surgery) may be considered candidates for
public finance if they provide substantial financial protection to house-
holds or address conditions (such as RHD) that disproportionately affect
the poor [19]. Still, the lack of published estimates of health sector costs
in African and Asian settings makes the planning of national RHD pre-
vention and treatment programs in these settings much more
challenging.
We found no cost estimates related to echocardiography-based
screening for RHD and catheter-based interventions, such as percutane-
ous transvenous mitral commissurotomy. In recent years, a number of
groups have begun to explore, or even advocate for, screening for RHD
among children and pregnant women who live in endemic settings
[20]–[21]. The absence of good empirical data on the cost of screening
at scale suggests that it is premature to recommend screening pediatric
populations for RHD. Similarly, a major objective of screening for RHD in
pregnancy is to identify women who would be eligible for catheter-
based interventions that could improve pregnancy outcomes; without
data on the costs of percutaneous transvenous mitral commissurotomy,
it is difficult to argue that antenatal screening is cost-effective, scalable,
and affordable in resource-limited settings.
According to the quality scoring system that we developed based on
the Global Health Cost Consortium draft reference case, the studies in-
cluded in this review were generally of good or very good quality,
with scores ranging from 10/17 to 16/17. The reference case items
that were adhered to in half or fewer of the studies included:
(i) describing, adequately, the data sources and methods used for esti-
mating the quantities of inputs (including the handling of shared
costs); (ii) description of sources of price data and price inputs in a man-
ner that allows for adjustment across settings; and (iii) consideration
and appropriate handling of capital costs (e.g., through depreciation or
annuitization) in line with the time horizon used in the study. Future re-
ports should ensure that these relatively technical points are adequately
addressed throughout the conduct of the research, including reporting.
Disaggregating data by cost component (e.g., human resources, com-
modities, etc.), risk groups (e.g., Indigenous, non-Indigenous), facility
characteristics (e.g., health post vs. community health center, public
vs. private-for-profit), and other relevant parameters could greatly in-
crease the usefulness and generalizability of data across settings.
Notedly, in this scoping review, we identified 27 studies with insuf-
ficient costing information related to methods and results. For instance,
Gilbert et al. performed a retrospective analysis of hospital records and
charges for children admitted to a tertiary hospital in New Zealand with
ARF or RHD [16]. While their estimates of costs had face validity (in our
view), there was very little elaboration on costing methods and data
sources used, and only a couple of point estimates (of total cost) were
reported. These factors restricted our ability to appraise the study's
methods or draw conclusions from the results. Similarly, Neuner et al.
explored the cost-effectiveness of various diagnostic and therapeutic
modalities for streptococcal pharyngitis; their model parameters'
input values were derived from published literature and available data-
bases, and no doubt were adequate for use in a decision-analysis prob-
lem, but they did not provide insights into the economic burden of
pharyngitis in a real-world setting [22]. Considering the limited empir-
ical evidence on the economic burden of RHD, researchers making use
of patient databases that include RHD costs might consider devoting
more attention to the conduct and reporting of their cost analyses to en-
sure they adhere to the standards proposed by the Global Health Cost
Consortium.
5
International Journal of Cardiology xxx (2020) xxx
Our scoping review has some limitations. While we included two
masters theses from the University of Cape Town because we had
prior knowledge of their existence, we did not systematically search ac-
ademic databases to identify theses and dissertations at other academic
institutions. Further, while focusing on English studies was practical for
the purposes of this study, we may be missing data from areas that have
a substantial volume of non-English-language literature (e.g., Latin
America and China). Finally, excluding literature with primary data col-
lection prior to the year 2000 limited the number of eligible studies but
allowed us to produce findings of greater relevance to current policy
and advocacy.
In summary, we found a handful of studies on the economic burden
of RHD-related healthcare to health systems, households, and society.
Prevention strategies appear to be relatively modest in terms of their
costs, while surgery and other tertiary care are quite expensive. Unfor-
tunately, except for two masters' theses, contemporary research on
the cost of RHD has been done only in high-income settings. Consider-
ing the recent political commitment by WHO Member States to imple-
ment RHD control programs, high-quality cost estimates are urgently
needed to assist in raising awareness, planning, and allocating resources
efficiently.
Declaration of Competing Interest
The authors have no conflicts of interest to declare.
Acknowledgements
This study was supported by a grant from the American Heart Asso-
ciation (17SFRN33670611) to Dr. Watkins.
Appendix A. Completed PRISMA-ScR checklist.
PRISMA-ScR Item Section where
addressed
1 Identification of the report as a scoping review Title
2 Summary that includes background, objectives, eligibility Abstract
criteria, sources of evidence, charting methods, results and
conclusions that relate to review questions and objectives
3 Rational for the review in the context of what is known, Introduction
explaining why the review questions/objectives lend
themselves to a scoping review approach
4 Explicit statement of questions and objectives being Introduction
addressed with reference to their key elements used to
conceptualize the review questions and/or objectives
5 Indication of whether a review protocol exists and where it Methods
can be accessed
6 Outlining characteristics of the sources of evidence used as Methods
eligibility criteria (years considered, language, and
publication status), and provide a rationale
7 Description of all information sources in the search Methods
(e.g., databases with dates of coverage and contact with
authors to identify additional sources), as well as the date
the most recent search was executed
8 Full electronic strategy for at least one database, including Appendix B
any limits used, such that it could be repeated
9 Statement of the process for selecting sources of evidence Methods
(i.e., screening and eligibility) included in the scoping
review
10 Describing the methods of charting data from the included Methods
sources of evidence (e.g. calibrated forms or forms that
have been tested by the team before their use, and
whether data charting was done independently or in
duplicate) and any processes for obtaining and confirming
data from investigators
11 List and definition of all variables for which data were Methods
sought and any assumptions and simplifications made
12 If done, a rational for conducting a critical appraisal of Methods
(continued on next page)
C.C. Opara, N. Aghassibake and D.A. Watkins
(continued)
PRISMA-ScR Item
included sources of evidence; describing the methods used
and how this information was used in any data synthesis
(if appropriate)
13 Describing the methods of handling and summarizing the
data that were charted
14 Numbers of sources of evidence screened, assessed for
eligibility, and included in the review, with reasons for
exclusions at each stage, ideally using a flow diagram
15 For each source of evidence, presentation of characteristics
for which data were charted and provide the citations
16 If done, presentation of data on critical appraisal of
included sources of evidence (see item 12)
17 For each included source of evidence, presentation of the
relevant data that were charted that relate to the review
questions and objectives
18 Summary and/or presentation of the charting results as
they relate to the review questions and objectives
19 Summary of the main results (including an overview of
concepts, themes, and types of evidence available), link to
the review questions and objectives, and consider the
relevance to key groups
20 Discussion of the limitations of the scoping review process
21 Provision of a general interpretation of the results with
respect to the review questions and objectives, as well as
potential implications and/or next steps
22 Description of sources of funding for the included sources
of evidence, as well as sources of funding for the scoping
review; description of the role of the funders of the
scoping review
Appendix B. Search strategies.
PubMed search strategy
((rheumatic heart disease) OR (acute rheumatic fever) OR (strepto-
coccal pharyngitis))
AND
(economy OR economies OR economic OR economics OR socioeco-
nomic OR socioeconomics OR cost OR costs OR costing OR financial OR
finances OR expenditure OR expenditures OR income OR incomes OR
payer OR payers OR payee OR payees OR payment OR payments OR
poverty OR “standard of living” OR social determinant OR social deter-
minants OR environmental determinant OR environmental
determinants)
AND 2000:3000[pdat] AND english[la].
Embase search strategy
((rheumatic heart disease) OR (acute rheumatic fever) OR (strepto-
coccal pharyngitis))
AND
(economy OR economies OR economic OR economics OR socioeco-
nomic OR socioeconomics OR cost OR costs OR costing OR financial OR
finances OR expenditure OR expenditures OR income OR incomes OR
payer OR payers OR payee OR payees OR payment OR payments OR
poverty OR “standard of living” OR “social determinant” OR “social de-
terminants” OR “environmental determinant” OR “environmental de-
terminants”) AND [2000−3000]/py.
EconLit search strategy
((rheumatic heart disease) OR (acute rheumatic fever) OR (strepto-
coccal pharyngitis))
AND
(economy OR economies OR economic OR economics OR socioeco-
nomic OR socioeconomics OR cost OR costs OR costing OR financial OR
finances OR expenditure OR expenditures OR income OR incomes OR
payer OR payers OR payee OR payees OR payment OR payments OR
poverty OR “standard of living” OR social determinant OR social deter-
minants OR environmental determinant OR environmental
determinants)
AND LA english
International Journal of Cardiology xxx (2020) xxx
NOTE: Date was filtered manually in EconLit
Section where
Cochrane Library Reviews search strategy
addressed ((rheumatic heart disease) OR (acute rheumatic fever) OR (strepto-
coccal pharyngitis))
AND
(economy OR economies OR economic OR economics OR socioeco-
Methods nomic OR socioeconomics OR cost OR costs OR costing OR financial OR
finances OR expenditure OR expenditures OR income OR incomes OR
Results
payer OR payers OR payee OR payees OR payment OR payments OR
poverty OR “standard of living” OR social determinant OR social deter-
Results minants OR environmental determinant OR environmental
determinants)
Results
NOTE: Date and language were manually filtered
Results NHS-EED search strategy
(((rheumatic heart disease) OR (acute rheumatic fever) OR (strepto-
coccal pharyngitis)) AND (economy OR economies OR economic OR
Results
economics OR socioeconomic OR socioeconomics OR cost OR costs OR
Discussion costing OR financial OR finances OR expenditure OR expenditures OR in-
come OR incomes OR payer OR payers OR payee OR payees OR payment
OR payments OR poverty OR standard of living OR social determinant
OR social determinants OR environmental determinant OR environ-
Discussion
mental determinants)) IN NHSEED FROM 2000 TO 2019.
Discussion
NOTE: Language was filtered manually in NHS-EED.
Results
Funding Date of search: March 29, 2019
PubMed: 614
Embase: 1781
EconLit: 0
Cochrane Library Reviews: 114
NHS-EED: 10
Other: 2
Total articles: 2521
Duplicates: 546
Total less duplicates: 1975
Appendix C. Quality checklist from the global health cost consor-
tium (September 2017 revision).
Study design
1 The purpose, the population, and the intervention and/or service/output of
the cost estimation [are] defined
2 The perspective of the cost estimation [is] defined
3 The type of cost being estimated [is] defined (e.g., economic vs financial,
real world vs guideline, incremental vs full, and whether cost is net of future
savings); the type of cost is justified relevant to purpose
4 The ‘units’ in the unit costs for strategies, services, and interventions [are]
defined, relevant for the costing purpose, and generalizable
5 The time horizon [is] explicit and of sufficient length to capture costs
relevant to the purpose (and consideration [is] given to disaggregating costs
into separate time periods where they vary over time)
Resource measurement
6 The scope of the inputs to include in the cost estimation [is] defined and
justified and relevant to purpose (where inputs are excluded for pragmatic
reasons, these [are] explicitly reported)
7 The methods for estimating the quantities of inputs [are] described,
including data sources, criteria for allocating shared costs, and exclusion of
research costs
8 The sampling frame, method, and size [are] determined by the precision
demanded by the costing purpose and designed to minimize bias
9 The selection of the data source(s) and methods for estimating the ‘units’
for unit costs [are] described and potential biases reported in the study
limitations
10 Consideration [is] given to the timing of data collection to minimize recall
bias and, where relevant, the impact of seasonality and other differences
over time
Pricing and valuation
11 Sources of price data… reflect the price relevant to the purpose of the
costing and are described in a way that allows for adjustment across
settings
6
C.C. Opara, N. Aghassibake and D.A. Watkins
(continued)
Study design
12 Capital costs [are] appropriately annuitized or depreciated to reflect the
opportunity cost of capital inputs over the timeframe relevant to the
[research question]
13 Where relevant, appropriate discount rate, inflation, and currency
conversion rate [are] used to adjust costs over setting and time
14 The use and source of shadow prices to value inputs without a market price
and the opportunity cost of time [are] reported
Analyzing and presenting results
15 Variation in the cost of the intervention by site size/organization,
sub-populations, or by other drivers of heterogeneity [is] explored and
reported
16 The uncertainty associated with the cost estimates [is] appropriately
characterized
17 Cost estimates, including the methods used, [are] communicated clearly and
transparently to… interpret and use the results
Adapted from Vassal and colleagues [12].
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