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Systematic Review of Economic Evaluations of Primary Caries Prevention in 2- to 5-Year-Old Preschool Children

Open AccessPublished:July 12, 2020DOI:https://doi.org/10.1016/j.jval.2020.04.1823

      Highlights

      • Recent reviews of economic evaluations of oral health interventions identified only a handful of studies conducted in preschool populations.
      • Our review included 39 papers and highlighted a wide variation in (1) types of caries prevention interventions investigated, (2) the effectiveness measures used, (3) the way in which costs and outcomes are reported, and (4) study perspective (when indicated). A lack of inclusion of preference-based health-related quality-of-life measures in preschool oral health evaluations was identified.
      • To identify cost-effective strategies, studies should include economic evaluations using best practice methods guidance and adhere to standards for reporting and presenting.

      Abstract

      Objectives

      To describe and summarize evidence on economic evaluations (EEs) of primary caries prevention in preschool children aged 2 to 5 years and to evaluate the reporting quality of full EE studies using a quality assessment tool.

      Methods

      A systematic literature search was conducted in several databases. Full and partial EEs were included. The reporting quality of full EE studies was assessed using the Consolidated Health Economic Evaluation Reporting Standards (CHEERS) checklist.

      Results

      A total of 808 studies were identified, and 39 were included in the review. Most papers were published between 2000 and 2017 and originated in the United States and the United Kingdom. The most common type of intervention investigated was a complex multicomponent intervention, followed by water fluoridation. Cost analysis and cost-effectiveness analysis were the most frequently used types of EE. One study employed cost-utility analysis. The proportion of full EEs increased over time. The parameters not reported well included study perspective, baseline year, sensitivity analysis, and discount rate. The CHEERS items that were most often unmet were characterizing uncertainty, study perspective, study parameters, and estimating resources and costs.

      Conclusions

      Within the past 2 decades, there has been an increase in the number of EEs of caries prevention interventions in preschool children. There was inconsistency in how EEs were conducted and reported. Lack of preference-based health-related quality-of-life measure utilization in the field was identified. The use of appropriate study methodologies and greater attention to recommended EE design are required to further improve quality.

      Keywords

      Introduction

      Untreated dental caries is one of the most common diseases affecting humans worldwide.
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      Early childhood caries can lead to pain, infections, and difficulties with eating, sleeping, and socializing, thus affecting a child’s general health and child and family quality of life.
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      It often leads to school absenteeism and parents taking time off work to take their children to a dentist or to hospital.
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      York Health Economics Consortium
      A rapid review of evidence on the cost-effectiveness of interventions to improve the oral health of children aged 0-5 years.
      Early childhood caries poses an economic burden to individuals, the health sector, and society more broadly.
      • Phantumvanit P.
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      WHO global consultation on public health intervention against early childhood caries.
      The 2010 global direct financial costs associated with dental caries were estimated to be $298 billion, and indirect costs came to $144 billion.
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      Sugars and dental caries.
      Untreated caries was found to cause 12% of global productivity losses due to dental diseases in 2015: $21.19 billion (11%) owing to untreated caries in permanent teeth and $0.90 billion (0.5%) to caries in deciduous teeth.
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      A number of child public health caries prevention strategies and intervention types currently exist, and choosing between competing oral public health programs is not always an easy decision for public health planners.
      • Morgan M.
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      Economic evaluation of preventive dental programs: what can they tell us?.
      In a recent critique, Watt et al
      • Watt R.G.
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      • Allison P.
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      Ending the neglect of global oral health: time for radical action.
      recommended that the priority for oral health (OH) research should be the promotion of applied health service and implementation research, with methodologies including economic evaluation (EE), so that planners are able to assess program performance comprehensively.
      • Watt R.G.
      • Daly B.
      • Allison P.
      • et al.
      Ending the neglect of global oral health: time for radical action.
      EEs help decision makers to allocate limited resources the best way, to achieve the greatest health benefit. A full EE is “a comparison of two or more alternative courses of action, while considering both inputs (costs) and outputs (consequences) associated with each.”
      • Drummond M.F.
      • Sculpher M.J.
      • Claxton K.
      • Stoddart G.L.
      • Torrance G.W.
      Methods for the Economic Evaluation of Health Care Programmes.
      The most common types of full EEs are cost-benefit analysis (CBA), cost-effectiveness analysis (CEA), and cost-utility analysis (CUA). A partial EE measures a program/intervention or disease costs but does not involve a comparison with alternative options and does not relate costs to outcomes.
      • Rabarison K.M.
      • Bish C.L.
      • Massoudi M.S.
      • Giles W.H.
      Economic evaluation enhances public health decision making.
      Partial EEs include program/intervention cost analysis, cost-outcome description, and cost-of-illness analysis.
      In recent years, the number of published cost-effectiveness studies on the prevention of dental diseases across the life course has increased
      • Marino R.J.
      • Khan A.R.
      • Morgan M.
      Systematic review of publications on economic evaluations of caries prevention programs.
      • Tonmukayakul U.
      • Calache H.
      • Clark R.
      • Wasiak J.
      • Faggion Jr., C.M.
      Systematic review and quality appraisal of economic evaluation publications in dentistry.
      • Rogers H.J.
      • Rodd H.D.
      • Vermaire J.H.
      • et al.
      A systematic review of the quality and scope of economic evaluations in child oral health research.
      ; however, some methodological limitations were found by the previous systematic reviews in the field of EE in dentistry
      • Tonmukayakul U.
      • Calache H.
      • Clark R.
      • Wasiak J.
      • Faggion Jr., C.M.
      Systematic review and quality appraisal of economic evaluation publications in dentistry.
      and caries prevention,
      • Marino R.J.
      • Khan A.R.
      • Morgan M.
      Systematic review of publications on economic evaluations of caries prevention programs.
      ,
      • Rogers H.J.
      • Rodd H.D.
      • Vermaire J.H.
      • et al.
      A systematic review of the quality and scope of economic evaluations in child oral health research.
      such as absence of sensitivity analysis and discounting and insufficient information on how costs and outcomes were measured.
      Experiences and health-related behavior patterns in early life are known to affect OH throughout the life course. Consequently, calls have been made for priority to be given to interventions targeting early ages.
      International Centre for Oral Health Inequalities Research & Policy
      Social Inequalities in Oral Health: From Evidence to Action.
      Therefore, this age group is the focus of the present systematic review. Recent reviews of EEs of OH improvement programs and interventions
      • Coffin D.
      • Craig J.
      • Arber M.
      • Glanville J.
      Literature Review of Economic Evaluations on Oral Health Improvement Programmes and Interventions.
      and of CUAs of OH interventions
      • Hettiarachchi R.M.
      • Kularatna S.
      • Downes M.J.
      • et al.
      The cost-effectiveness of oral health interventions: a systematic review of cost-utility analyses.
      (both of which included subjects of all ages) identified only a handful of studies conducted in preschool populations. To our knowledge, there is only 1 nonsystematic review that looked at the evidence on the cost-effectiveness of interventions to improve the OH of younger children, aged 0 to 5 years, in particular.
      York Health Economics Consortium
      A rapid review of evidence on the cost-effectiveness of interventions to improve the oral health of children aged 0-5 years.
      This rapid review with a narrow search time frame (between 2012 and 2016) found only 5 studies that met the inclusion criteria. The authors found scarce cost-effectiveness evidence but warned that this should not be interpreted as evidence that those interventions were not effective or cost-effective.
      This is the first systematic review of EEs of primary caries prevention focusing specifically on preschool children aged 2 to 5 years, including both full and partial EEs and using a formal quality assessment tool. The objectives of the present systematic review were to describe and summarize currently available scientific literature on EEs of primary caries prevention in preschool children aged 2 to 5 years and to evaluate the reporting quality of full EE studies using an EE quality assessment tool.

      Methods

      The review followed the Preferred Reporting System for Systematic Reviews and Meta-Analysis (PRISMA) strategy
      • Liberati A.
      • Altman D.G.
      • Tetzlaff J.
      • et al.
      The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration.
      (Fig. 1). The protocol of this systematic review was registered in the international database of prospectively registered systematic reviews in health and social care (PROSPERO), Centre for Reviews and Dissemination, University of York (No. CRD42017083732).
      • Anopa Y.
      • Macpherson L.
      • McIntosh E.
      Economic evaluations of paediatric caries prevention in pre-school children aged two to five years: a systematic review. PROSPERO 2017, CRD42017083732. PROSPERO, Centre for Reviews and Dissemination, University of York.
      Figure thumbnail gr1
      Figure 1PRISMA flow diagram.
      CBA indicates cost-benefit analysis; CEA, cost-effectiveness analysis; CUA, cost-utility analysis; EVPI, expected value of perfect information analysis; PRISMA, Preferred Reporting System for Systematic Reviews and Meta-Analysis.

       Eligibility Criteria

      A Participants, Interventions, Comparators, Outcomes, and Study design (PICOS) approach
      • Liberati A.
      • Altman D.G.
      • Tetzlaff J.
      • et al.
      The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration.
      ,
      • Schardt C.
      • Adams M.B.
      • Owens T.
      • Keitz S.
      • Fontelo P.
      Utilization of the PICO framework to improve searching PubMed for clinical questions.
      was used in developing the eligibility criteria. The OH interventions of interest were interventions aimed at primary caries prevention in preschoolers (eg, water fluoridation, fluoride toothpaste, fluoride varnish/gel, fluoride tablets, fissure sealant, OH educational interventions, etc). Studies on interventions aimed at secondary caries prevention were not included (eg, restorative treatment of existing caries). To be included in this review, a study had to report relevant results for children aged between 2 and 5 years (inclusive). At least some age groups from this range had to be reported. Any types of EEs were included: full EEs (eg, employing CEA, CBA, or CUA) and partial EEs (eg, cost analysis, cost-outcome description). A full list of inclusion and exclusion criteria is presented in Appendix Table 1 (in Supplemental Materials found at https://doi.org/10.1016/j.jval.2020.04.1823).

       Data Sources and Search Strategy

      A systematic literature search was conducted in the following databases: MEDLINE and EMBASE (via the Ovid platform) and EconLit (via the EBSCO platform). Several previous systematic reviews of EEs of OH interventions, their search strategies used, and reference lists were consulted
      York Health Economics Consortium
      A rapid review of evidence on the cost-effectiveness of interventions to improve the oral health of children aged 0-5 years.
      ,
      • Marino R.J.
      • Khan A.R.
      • Morgan M.
      Systematic review of publications on economic evaluations of caries prevention programs.
      ,
      • Tonmukayakul U.
      • Calache H.
      • Clark R.
      • Wasiak J.
      • Faggion Jr., C.M.
      Systematic review and quality appraisal of economic evaluation publications in dentistry.
      ,
      • Coffin D.
      • Craig J.
      • Arber M.
      • Glanville J.
      Literature Review of Economic Evaluations on Oral Health Improvement Programmes and Interventions.
      ,
      • Hettiarachchi R.M.
      • Kularatna S.
      • Downes M.J.
      • et al.
      The cost-effectiveness of oral health interventions: a systematic review of cost-utility analyses.
      ,
      • Kallestal C.
      • Norlund A.
      • Soder B.
      • et al.
      Economic evaluation of dental caries prevention: a systematic review.
      Reference lists of the studies included in this systematic review were checked for any additional eligible studies.
      Search strategies and search terms for this systematic review were developed based on the Canadian Agency for Drugs and Technologies in Health (CADTH) EE filters.,
      • Glanville J.
      • Fleetwood K.
      • Yellowlees A.
      • Kaunelis D.
      • Mensinkai S.
      Development and testing of search filters to identify economic evaluations in MEDLINE and EMBASE.
      The guidance for undertaking systematic reviews in healthcare by the University of York’s Centre for Reviews and Dissemination (CRD)
      Centre for Reviews and Dissemination
      Systematic Reviews. CRD’s Guidance for Undertaking Reviews in Health Care.
      and a series of papers on how to conduct a systematic review of EEs
      • van Mastrigt G.A.
      • Hiligsmann M.
      • Arts J.J.
      • et al.
      How to prepare a systematic review of economic evaluations for informing evidence-based healthcare decisions: a five-step approach (part 1/3).
      • Thielen F.W.
      • Van Mastrigt G.A.P.G.
      • Burgers L.T.
      • et al.
      How to prepare a systematic review of economic evaluations for clinical practice guidelines: database selection and search strategy development (part 2/3).
      • Wijnen B.
      • Van Mastrigt G.
      • Redekop W.K.
      • Majoie H.
      • De Kinderen R.
      • Evers S.
      How to prepare a systematic review of economic evaluations for informing evidence-based healthcare decisions: data extraction, risk of bias, and transferability (part 3/3).
      were used in the process of developing and conducting this review.
      No publication time or language restrictions were applied. It was planned that should any relevant papers be identified in languages other than English or Russian, these would be translated with a help of professional translation services.
      The following blocks of search terms were used: (1) Canadian Agency for Drugs and Technologies in Health–based search filter (including various types of EEs, economics, costs, and economic modeling); (2) OH, caries, and OH intervention terms (OH, caries, early childhood caries, dental decay; toothbrushing, toothpaste, fluoride, fissure sealant, chlorhexidine, mouthwash; educational, preventive, and promotional OH initiatives, etc); and (3) terms related to preschool age (toddler, infant, preschool, early childhood, nursery, kindergarten, early years; Appendix Box 1 in Supplemental Materials found at https://doi.org/10.1016/j.jval.2020.04.1823).

       Study Selection Procedure

      Titles and abstracts of all retrieved records were screened against the inclusion criteria by the lead author, using a method developed by Bramer et al
      • Bramer W.M.
      • Milic J.
      • Mast F.
      Reviewing retrieved references for inclusion in systematic reviews using EndNote.
      employing EndNote (Clarivate Analytics, Philadelphia, PA). Citations with a title but no abstract were assessed for relevance based on the title only. Twenty percent of all titles and abstracts were checked by a second reviewer. Any disagreements were resolved by consensus-seeking discussions between all 3 reviewers. The full texts of all potentially relevant articles were retrieved and screened by the author, with any questionable cases discussed with one of the second reviewers or between all 3 reviewers, depending on the nature of an issue.

       Data Extraction

      Descriptive study data were extracted using a pretested data extraction template. Several sources were used during the development of an initial draft data extraction template: Centre for Reviews and Dissemination’s guidance for undertaking systematic reviews in healthcare,
      Centre for Reviews and Dissemination
      Systematic Reviews. CRD’s Guidance for Undertaking Reviews in Health Care.
      data extraction templates used in 2 previous reviews,
      York Health Economics Consortium
      A rapid review of evidence on the cost-effectiveness of interventions to improve the oral health of children aged 0-5 years.
      ,
      • Hettiarachchi R.M.
      • Kularatna S.
      • Downes M.J.
      • et al.
      The cost-effectiveness of oral health interventions: a systematic review of cost-utility analyses.
      and the Consolidated Health Economic Evaluation Reporting Standards (CHEERS) checklist.
      • Husereau D.
      • Drummond M.
      • Petrou S.
      • et al.
      Consolidated Health Economic Evaluation Reporting Standards (CHEERS) statement.
      Our own preferences, based on the nature and objectives of this review, were also taken into account. Data were extracted by the lead author, and a randomly selected 20% were checked by a second reviewer. Any disagreements were resolved by a discussion within the reviewers’ team.

       Reporting Quality Assessment of Full EEs

      The quality of full EEs was assessed using the CHEERS checklist,
      • Husereau D.
      • Drummond M.
      • Petrou S.
      • et al.
      Consolidated Health Economic Evaluation Reporting Standards (CHEERS) statement.
      ,
      • Husereau D.
      • Drummond M.
      • Petrou S.
      • et al.
      Consolidated Health Economic Evaluation Reporting Standards (CHEERS)—explanation and elaboration: a report of the ISPOR Health Economic Evaluation Publication Guidelines Good Reporting Practices Task Force.
      which was developed specifically to optimize the reporting of health EEs. The CHEERS checklist contains 24 items subdivided into 6 categories (Appendix Table 2 in Supplemental Materials found at https://doi.org/10.1016/j.jval.2020.04.1823).
      Each item of the CHEERS checklist was scored as “1”/”Yes,” if the paper met the criteria in full; “0”/”No,” if it did not meet the criteria; or “Not applicable.” Partial scores were not assigned. Items from the “Other” category (“Source of funding” and “Conflicts of interest”) were not taken into account when calculating a total score for each paper, as these are not directly relevant to the economics-related reporting quality of a paper. A total of 22 was the maximum possible score. Each study’s reporting quality was expressed as a proportion of items fully met for each paper. The lead author assessed all of the selected papers using the CHEERS checklist, with a second reviewer reassessing 20% of these papers, selected at random. Any discrepancies were resolved by discussion between the 2 reviewers.

      Results

       Literature Databases Search Results

      A total of 808 studies were identified, of which 42 met the inclusion criteria. Figure 1, a Preferred Reporting System for Systematic Reviews and Meta-Analysis flow diagram, illustrates the study selection process. A set of 3 papers reported on the same study,
      • Ast D.B.
      • Cons N.C.
      • Carlos J.P.
      • Maiwald A.
      Time and cost factors to provide regular, periodic dental care for children in a fluoridated and nonfluoridated area.
      • Ast D.B.
      • Cons N.C.
      • Carlos J.P.
      • Polan A.
      Time and cost factors to provide regular, periodic dental care for children in a fluoridated and nonfluoridated area: progress report. II.
      • Ast D.B.
      • Cons N.C.
      • Pollard S.T.
      • Garfinkel J.
      Time and cost factors to provide regular, periodic dental care for children in a fluoridated and nonfluoridated area: final report.
      with 2 reporting on intermediate results. Only the latest of the 3, reporting the study in full, was included in the review.
      • Ast D.B.
      • Cons N.C.
      • Pollard S.T.
      • Garfinkel J.
      Time and cost factors to provide regular, periodic dental care for children in a fluoridated and nonfluoridated area: final report.
      Two papers reported on another study, one of them being a health technology assessment report
      • Tickle M.
      • O’Neill C.
      • Donaldson M.
      • et al.
      A randomised controlled trial to measure the effects and costs of a dental caries prevention regime for young children attending primary care dental services: the Northern Ireland Caries Prevention In Practice (NIC-PIP) trial.
      and another a conventional journal paper.
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      Only the latter
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      was included into the review. The final number of papers included in the review was therefore 39.

       Study Characteristics

      Of the 39 papers, 25 (64%) were published between 2000 and 2017 (inclusive). The earliest of the identified papers was published in 1968. Twenty-three (59%) were partial EEs, namely cost analyses, and 16 (41%) were full EEs. The type of EE was reported in relation to the age group of interest: 2 to 5 years. There was 1 study that was a CBA for a full age range of participants (2 to 16 years), but only cost data were reported for the 2- to 7-year-old group.
      • Potapova L.V.
      Medical and economic effectiveness of drinking water fluoridation in a district with an intensive dental caries affection in children [in Russian].
      This study was classified as partial EE. Table 1 reports various study characteristics.
      Table 1Study characteristics of all included studies.
      CharacteristicNo. of studies (%) (total = 39)
      Year of publication
       1968-19702 (5%); 2 partial EEs
       1971-19806 (15%); 6 partial EEs
       1981-19902 (51%); 1 partial and 1 full EE
       1991-20005 (13%); 3 partial and 2 full EEs
       2001-201010 (26%); 6 partial and 4 full EEs
       2011-201714 (36%); 5 partial and 9 full EEs
      Type of EE
       Cost analysis
      One study36 was a CBA for a full age range of participants, 2 to 16 years, but only cost data were reported for the 2- to 7-year-old group For the purposes of our review, this study was classified as partial economic evaluation, cost analysis.
      23 (59%)
       CEA12 (31%)
       CBA1 (3%)
       CEA + CBA1 (3%)
       CEA + CUA1 (3%)
       CEA + EVPI1 (3%)
      Type of study
       Observational in nature (non-RCT/nonmodeling study)24 (61%)
       Alongside an RCT4 (10%)
       Markov model4 (10%)
       Calculations using previously published data (but not a formal model)3 (8%)
       System dynamics modeling2 (5%)
       Based on an RCT, but not alongside it (costs of a hypothetical prevention program, based on RCT results)1 (3%)
       EVPI + cost-effectiveness model1 (3%)
      Study country
       United States16 (41%)
       United Kingdom9 (23%)
       Canada3 (8%)
       Sweden3 (8%)
       Australia2 (5%)
       USSR2 (5%)
       Chile1 (3%)
       Finland1 (3%)
       Taiwan1 (3%)
       Uzbekistan1 (3%)
      Type of intervention
       Complex/multicomponent intervention12 (31%)
       Water fluoridation7 (18%)
       Oral health education (with or without additional elements)4 (10%)
       Topical fluoride (varnish, foam, gel)4 (10%)
       Multiple interventions compared3 (8%)
       Primary molar sealants2 (5%)
       Systemic fluoride (with or without additional elements)2 (5%)
       Toothbrushing2 (5%)
       Fluoridated milk and cereal1 (3%)
       Microbiological screening1 (3%)
       Preventive dental visit1 (3%)
      CBA indicates cost-benefit analysis; CEA, cost-effectiveness analysis; CUA, cost-utility analysis; EE, economic evaluation; EVPI expected value of perfect information; RCT, randomized controlled trial.
      One study
      • Potapova L.V.
      Medical and economic effectiveness of drinking water fluoridation in a district with an intensive dental caries affection in children [in Russian].
      was a CBA for a full age range of participants, 2 to 16 years, but only cost data were reported for the 2- to 7-year-old group For the purposes of our review, this study was classified as partial economic evaluation, cost analysis.

       Partial EEs

      Twenty-three partial EE studies were reviewed in total. All partial EEs were cost analyses. Seven studies (30% of the total number of partial EEs) investigated the costs of water fluoridation.
      • Ast D.B.
      • Cons N.C.
      • Pollard S.T.
      • Garfinkel J.
      Time and cost factors to provide regular, periodic dental care for children in a fluoridated and nonfluoridated area: final report.
      ,
      • Potapova L.V.
      Medical and economic effectiveness of drinking water fluoridation in a district with an intensive dental caries affection in children [in Russian].
      • Dowell T.B.
      The economics of fluoridation.
      • Fidler P.E.
      A comparison of treatment patterns and costs for a fluoride and non-fluoride community.
      • Lewis D.W.
      • Hunt A.M.
      • Kawall K.
      • Feasby R.E.
      Initial dental care time, cost and treatment requirements under changing exposure to fluoride during tooth development.
      • Rugg-Gunn A.J.
      • Carmichael C.L.
      • French A.D.
      • Furness J.A.
      Fluoridation in Newcastle and Northumberland: a clinical study of 5-year-old children.
      • Trubman A.
      • Silberman S.L.
      • Meydrech E.F.
      Treatment costs for carious primary teeth related to fluoride exposure.
      Six (26%) investigated multicomponent interventions,
      • Buckingham S.
      • John J.H.
      Outcomes and costs of pre-school and school-based fluoride varnish pilots.
      • Gisselsson H.
      • Birkhed D.
      • Bjorn A.L.
      Effect of a 3-year professional flossing program with chlorhexidine gel on approximal caries and cost of treatment in preschool children.
      • Jokela J.
      • Pienihakkinen K.
      Economic evaluation of a risk-based caries prevention program in preschool children.
      • Jong A.
      • Leske G.S.
      Utilization and cost of dental services for preschool children in Boston's Head-Start program.
      • Kaakko T.
      • Skaret E.
      • Getz T.
      • et al.
      An ABCD program to increase access to dental care for children enrolled in Medicaid in a rural county.
      • Lewis D.W.
      • Magid S.C.
      • Jarrett M.E.
      The Waterloo preschool children’s denticare study.
      3 investigated topical fluoride (varnish, foam, or gel),
      • Chen S.F.
      • Lin H.C.
      Dental service utilization and costs before and after introduction of fluoride gel application for preschool children in Taiwan.
      • Hawkins R.
      • Noble J.
      • Locker D.
      • et al.
      A comparison of the costs and patient acceptability of professionally applied topical fluoride foam and varnish.
      • Kranz A.M.
      • Rozier R.G.
      • Preisser J.S.
      • Stearns S.C.
      • Weinberger M.
      • Lee J.Y.
      Preventive services by medical and dental providers and treatment outcomes.
      and 2 compared several different interventions.
      • Edelstein B.L.
      • Hirsch G.
      • Frosh M.
      • Kumar J.
      Reducing early childhood caries in a Medicaid population: a systems model analysis.
      ,
      • Hirsch G.B.
      • Edelstein B.L.
      • Frosh M.
      • Anselmo T.
      A simulation model for designing effective interventions in early childhood caries.
      There were single studies investigating each of the following: fluoride drops (for younger children) and fluoride varnish (for older children),
      • Pashaev K.P.
      Economic effectiveness of dental caries prevention with fluorine preparations [in Russian].
      microbiological screening,
      • Zavras A.I.
      • Edelstein B.L.
      • Vamvakidis A.
      Health care savings from microbiological caries risk screening of toddlers: a cost estimation model.
      age at the first preventive dental visit,
      • Savage M.F.
      • Lee J.Y.
      • Kotch J.B.
      • Vann Jr., W.F.
      Early preventive dental visits: effects on subsequent utilization and costs.
      OH education of parents with several additional components,
      • Wennhall I.
      • Norlund A.
      • Matsson L.
      • Twetman S.
      Cost-analysis of an oral health outreach program for preschool children in a low socioeconomic multicultural area in Sweden.
      and supervised toothbrushing in nurseries.
      • Anopa Y.
      • McMahon A.D.
      • Conway D.I.
      • Ball G.E.
      • McIntosh E.
      • Macpherson L.M.
      Improving child oral health: cost analysis of a national nursery toothbrushing programme.
      Reporting of sensitivity analysis, study perspective, discount rate, and base year are shown in Table 2. For further study descriptions, see Appendix Tables 5 and 6 in Supplemental Materials found at https://doi.org/10.1016/j.jval.2020.04.1823.
      Table 2Sensitivity analysis, study perspective, discount rate, and base year (by type of EE: partial, full).
      ParameterPartial EEs: No. of studies (%) (total = 23)Full EEs: No. of studies (%) (total = 16)
      Sensitivity analysis
       Some form of sensitivity analysis7 (30%)
      • Jokela J.
      • Pienihakkinen K.
      Economic evaluation of a risk-based caries prevention program in preschool children.
      ,
      • Lewis D.W.
      • Magid S.C.
      • Jarrett M.E.
      The Waterloo preschool children’s denticare study.
      ,
      • Edelstein B.L.
      • Hirsch G.
      • Frosh M.
      • Kumar J.
      Reducing early childhood caries in a Medicaid population: a systems model analysis.
      ,
      • Hirsch G.B.
      • Edelstein B.L.
      • Frosh M.
      • Anselmo T.
      A simulation model for designing effective interventions in early childhood caries.
      ,
      • Zavras A.I.
      • Edelstein B.L.
      • Vamvakidis A.
      Health care savings from microbiological caries risk screening of toddlers: a cost estimation model.
      ,
      • Wennhall I.
      • Norlund A.
      • Matsson L.
      • Twetman S.
      Cost-analysis of an oral health outreach program for preschool children in a low socioeconomic multicultural area in Sweden.
      ,
      • Anopa Y.
      • McMahon A.D.
      • Conway D.I.
      • Ball G.E.
      • McIntosh E.
      • Macpherson L.M.
      Improving child oral health: cost analysis of a national nursery toothbrushing programme.
      12 (75%)

      In addition, 1 (6%) with no formal sensitivity analysis but 2 types of intrachild correlation were modeled, resulting in 2 sets of results
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
       Not employed12 (52%)3 (19%)
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
       Not applicable4 (17%)
      • Fidler P.E.
      A comparison of treatment patterns and costs for a fluoride and non-fluoride community.
      ,
      • Lewis D.W.
      • Hunt A.M.
      • Kawall K.
      • Feasby R.E.
      Initial dental care time, cost and treatment requirements under changing exposure to fluoride during tooth development.
      ,
      • Gisselsson H.
      • Birkhed D.
      • Bjorn A.L.
      Effect of a 3-year professional flossing program with chlorhexidine gel on approximal caries and cost of treatment in preschool children.
      ,
      • Kranz A.M.
      • Rozier R.G.
      • Preisser J.S.
      • Stearns S.C.
      • Weinberger M.
      • Lee J.Y.
      Preventive services by medical and dental providers and treatment outcomes.
      0 (0%)
      EE perspective
       Stated4 (17%), as follows:

      Healthcare system perspective: 3 (13%)
      • Rugg-Gunn A.J.
      • Carmichael C.L.
      • French A.D.
      • Furness J.A.
      Fluoridation in Newcastle and Northumberland: a clinical study of 5-year-old children.
      ,
      • Chen S.F.
      • Lin H.C.
      Dental service utilization and costs before and after introduction of fluoride gel application for preschool children in Taiwan.
      ,
      • Anopa Y.
      • McMahon A.D.
      • Conway D.I.
      • Ball G.E.
      • McIntosh E.
      • Macpherson L.M.
      Improving child oral health: cost analysis of a national nursery toothbrushing programme.


      Medicaid perspective: 1 (4%)
      • Savage M.F.
      • Lee J.Y.
      • Kotch J.B.
      • Vann Jr., W.F.
      Early preventive dental visits: effects on subsequent utilization and costs.
      9 (56%), as follows:

      Healthcare payer perspective: 3 (19%)
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
      • Quinonez R.B.
      • Stearns S.C.
      • Talekar B.S.
      • Rozier R.G.
      • Downs S.M.
      Simulating cost-effectiveness of fluoride varnish during well-child visits for Medicaid-enrolled children.
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.


      Public payer perspective: 3 (19%)
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
      ,
      • Chi D.L.
      • van der Goes D.N.
      • Ney J.P.
      Cost-effectiveness of pit-and-fissure sealants on primary molars in Medicaid-enrolled children.


      Societal perspective: 2 (12%)
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.


      Different perspectives (for the base-case and subgroup analyses): 1 (6%)
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
       Not stated19 (83%)7 (44%)
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      ,
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
      • Widenheim J.
      • Birkhed D.
      Caries-preventive effect on primary and permanent teeth and cost-effectiveness of an NaF tablet preschool program.
      Discount rate.
       Stated3 (13%)
      • Dowell T.B.
      The economics of fluoridation.
      ,
      • Zavras A.I.
      • Edelstein B.L.
      • Vamvakidis A.
      Health care savings from microbiological caries risk screening of toddlers: a cost estimation model.
      ,
      • Wennhall I.
      • Norlund A.
      • Matsson L.
      • Twetman S.
      Cost-analysis of an oral health outreach program for preschool children in a low socioeconomic multicultural area in Sweden.
      11 (69%)
       Not stated6 (26%)
      • Potapova L.V.
      Medical and economic effectiveness of drinking water fluoridation in a district with an intensive dental caries affection in children [in Russian].
      ,
      • Kranz A.M.
      • Rozier R.G.
      • Preisser J.S.
      • Stearns S.C.
      • Weinberger M.
      • Lee J.Y.
      Preventive services by medical and dental providers and treatment outcomes.
      • Edelstein B.L.
      • Hirsch G.
      • Frosh M.
      • Kumar J.
      Reducing early childhood caries in a Medicaid population: a systems model analysis.
      • Hirsch G.B.
      • Edelstein B.L.
      • Frosh M.
      • Anselmo T.
      A simulation model for designing effective interventions in early childhood caries.
      • Pashaev K.P.
      Economic effectiveness of dental caries prevention with fluorine preparations [in Russian].
      ,
      • Savage M.F.
      • Lee J.Y.
      • Kotch J.B.
      • Vann Jr., W.F.
      Early preventive dental visits: effects on subsequent utilization and costs.
      3 (19%)
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      ,
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      ,
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
       Not applicable (or authors explained why costs/outcomes were not discounted)14 (61%)
      • Ast D.B.
      • Cons N.C.
      • Pollard S.T.
      • Garfinkel J.
      Time and cost factors to provide regular, periodic dental care for children in a fluoridated and nonfluoridated area: final report.
      ,
      • Fidler P.E.
      A comparison of treatment patterns and costs for a fluoride and non-fluoride community.
      • Lewis D.W.
      • Hunt A.M.
      • Kawall K.
      • Feasby R.E.
      Initial dental care time, cost and treatment requirements under changing exposure to fluoride during tooth development.
      • Rugg-Gunn A.J.
      • Carmichael C.L.
      • French A.D.
      • Furness J.A.
      Fluoridation in Newcastle and Northumberland: a clinical study of 5-year-old children.
      • Trubman A.
      • Silberman S.L.
      • Meydrech E.F.
      Treatment costs for carious primary teeth related to fluoride exposure.
      • Buckingham S.
      • John J.H.
      Outcomes and costs of pre-school and school-based fluoride varnish pilots.
      • Gisselsson H.
      • Birkhed D.
      • Bjorn A.L.
      Effect of a 3-year professional flossing program with chlorhexidine gel on approximal caries and cost of treatment in preschool children.
      • Jokela J.
      • Pienihakkinen K.
      Economic evaluation of a risk-based caries prevention program in preschool children.
      • Jong A.
      • Leske G.S.
      Utilization and cost of dental services for preschool children in Boston's Head-Start program.
      • Kaakko T.
      • Skaret E.
      • Getz T.
      • et al.
      An ABCD program to increase access to dental care for children enrolled in Medicaid in a rural county.
      • Lewis D.W.
      • Magid S.C.
      • Jarrett M.E.
      The Waterloo preschool children’s denticare study.
      • Chen S.F.
      • Lin H.C.
      Dental service utilization and costs before and after introduction of fluoride gel application for preschool children in Taiwan.
      • Hawkins R.
      • Noble J.
      • Locker D.
      • et al.
      A comparison of the costs and patient acceptability of professionally applied topical fluoride foam and varnish.
      ,
      • Anopa Y.
      • McMahon A.D.
      • Conway D.I.
      • Ball G.E.
      • McIntosh E.
      • Macpherson L.M.
      Improving child oral health: cost analysis of a national nursery toothbrushing programme.
      2 (12%)
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
      Base year
       Stated13 (61%)11 (75%)
       Not stated9 (39%)
      • Potapova L.V.
      Medical and economic effectiveness of drinking water fluoridation in a district with an intensive dental caries affection in children [in Russian].
      ,
      • Trubman A.
      • Silberman S.L.
      • Meydrech E.F.
      Treatment costs for carious primary teeth related to fluoride exposure.
      ,
      • Buckingham S.
      • John J.H.
      Outcomes and costs of pre-school and school-based fluoride varnish pilots.
      ,
      • Kaakko T.
      • Skaret E.
      • Getz T.
      • et al.
      An ABCD program to increase access to dental care for children enrolled in Medicaid in a rural county.
      ,
      • Hawkins R.
      • Noble J.
      • Locker D.
      • et al.
      A comparison of the costs and patient acceptability of professionally applied topical fluoride foam and varnish.
      ,
      • Edelstein B.L.
      • Hirsch G.
      • Frosh M.
      • Kumar J.
      Reducing early childhood caries in a Medicaid population: a systems model analysis.
      ,
      • Pashaev K.P.
      Economic effectiveness of dental caries prevention with fluorine preparations [in Russian].
      • Zavras A.I.
      • Edelstein B.L.
      • Vamvakidis A.
      Health care savings from microbiological caries risk screening of toddlers: a cost estimation model.
      • Savage M.F.
      • Lee J.Y.
      • Kotch J.B.
      • Vann Jr., W.F.
      Early preventive dental visits: effects on subsequent utilization and costs.
      4 (25%)
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
      EE indicates economic evaluation.

       Full EEs

      Sixteen full EE papers were reviewed. Six investigated complex multicomponent interventions,
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      ,
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
      ,
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
      3 looked at OH education (with or without additional components),
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      ,
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
      2 examined primary molar sealants,
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
      ,
      • Chi D.L.
      • van der Goes D.N.
      • Ney J.P.
      Cost-effectiveness of pit-and-fissure sealants on primary molars in Medicaid-enrolled children.
      and there was 1 study investigating each of the following: fluoridated milk and milk cereal,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      fluoride varnish,
      • Quinonez R.B.
      • Stearns S.C.
      • Talekar B.S.
      • Rozier R.G.
      • Downs S.M.
      Simulating cost-effectiveness of fluoride varnish during well-child visits for Medicaid-enrolled children.
      sodium fluoride tablets (plus other underlying interventions),
      • Widenheim J.
      • Birkhed D.
      Caries-preventive effect on primary and permanent teeth and cost-effectiveness of an NaF tablet preschool program.
      toothbrushing,
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      and comparing multiple interventions.
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.
      With regard to study settings, 7 (44%) were modeling studies,
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
      ,
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      ,
      • Quinonez R.B.
      • Stearns S.C.
      • Talekar B.S.
      • Rozier R.G.
      • Downs S.M.
      Simulating cost-effectiveness of fluoride varnish during well-child visits for Medicaid-enrolled children.
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.
      • Chi D.L.
      • van der Goes D.N.
      • Ney J.P.
      Cost-effectiveness of pit-and-fissure sealants on primary molars in Medicaid-enrolled children.
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
      3 (19%) were conducted in dental settings (dental practice, dental clinic),
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
      ,
      • Widenheim J.
      • Birkhed D.
      Caries-preventive effect on primary and permanent teeth and cost-effectiveness of an NaF tablet preschool program.
      2 (13%) studies were conducted in multiple settings,
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
      ,
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
      2 (13%) at home,
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      ,
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      1 study (6%) was kindergarten based,
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
      and 1 (6%) was community based.
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      The most frequently used type of framework for full EE was CEA (12 papers, 75%).
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      ,
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
      • Quinonez R.B.
      • Stearns S.C.
      • Talekar B.S.
      • Rozier R.G.
      • Downs S.M.
      Simulating cost-effectiveness of fluoride varnish during well-child visits for Medicaid-enrolled children.
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.
      • Chi D.L.
      • van der Goes D.N.
      • Ney J.P.
      Cost-effectiveness of pit-and-fissure sealants on primary molars in Medicaid-enrolled children.
      ,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      ,
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
      ,
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
      • Widenheim J.
      • Birkhed D.
      Caries-preventive effect on primary and permanent teeth and cost-effectiveness of an NaF tablet preschool program.
      Other studies employed CBA
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
      or a combination of CEA with 1 of the following: CBA,
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      CUA,
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      and expected value of perfect information.
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
      Appendix Table 3 (in Supplemental Materials found at https://doi.org/10.1016/j.jval.2020.04.1823) further illustrates the breakdown of full EE studies by type of intervention and EE framework used, and Appendix Tables 7 and 8 (in Supplemental Materials found at https://doi.org/10.1016/j.jval.2020.04.1823) contain study descriptions.
      A wide variety of OH and economic outcomes measures were used in the reviewed full EE studies (Table 3). Four studies
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      ,
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      ,
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
      used mean dmft, which is the mean number of deciduous teeth decayed (d), missing due to decay (m) or filled (f), with the unit being the tooth (t) and/or dmfs (the same principle as for dmft applies, but the unit of measurement is the tooth surface [s]). Two studies used conversion from caries free to caries active (plus other secondary measures).
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      Nine other OH outcome measures were used in 1 study each. Five different cost-effectiveness outcomes were used in individual studies. Only 1 study used the quality-adjusted life-year (QALY) as an outcome.
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      The most widely used options for reporting of costs and outcomes were the incremental cost-effectiveness ratio and the average cost-effectiveness ratio.
      Table 3Full EE studies: oral health and economic outcomes used and reporting and presentation of costs and outcomes.
      CharacteristicNo. of studies (%) (total = 16)
      Oral health outcomes
       dmft or/and dmfs4 (25%)
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      ,
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      ,
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
       Conversion from caries-free to caries-active (plus other secondary measures)2 (12%)
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
       Cavity-free months1 (6%)
      • Quinonez R.B.
      • Stearns S.C.
      • Talekar B.S.
      • Rozier R.G.
      • Downs S.M.
      Simulating cost-effectiveness of fluoride varnish during well-child visits for Medicaid-enrolled children.
       fs, dfsa1 (6%)
      • Widenheim J.
      • Birkhed D.
      Caries-preventive effect on primary and permanent teeth and cost-effectiveness of an NaF tablet preschool program.
       Mean No. of restorations and extractions averted1 (6%)
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
       No. of carious surfaces1 (6%)
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
       No. of carious teeth1 (6%)
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
       No. of cases/incidence rates of caries and stomatitis1 (6%)
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
       Rates of dental treatment1 (6%)
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
       Reduction in dental treatments1 (6%)
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
       Reduction in No. of carious teeth; reduction in full-mouth dental reconstructions1 (6%)
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.
       N/A (tooth-level model)1 (6%)
      • Chi D.L.
      • van der Goes D.N.
      • Ney J.P.
      Cost-effectiveness of pit-and-fissure sealants on primary molars in Medicaid-enrolled children.
      Preference-based outcomes
       QALY (based on Child Health Utility 9 Dimensions [CHU9D]–parental proxy questionnaire)1 (6%)
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      Reporting and presentation of costs and outcomes
       ICER7 (44%)
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
      ,
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
      ,
      • Quinonez R.B.
      • Stearns S.C.
      • Talekar B.S.
      • Rozier R.G.
      • Downs S.M.
      Simulating cost-effectiveness of fluoride varnish during well-child visits for Medicaid-enrolled children.
      ,
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      ,
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
       ACER
      One study60 reported both average C/E and B/C ratios.
      3 (19%)
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      ,
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.
      ,
      • Widenheim J.
      • Birkhed D.
      Caries-preventive effect on primary and permanent teeth and cost-effectiveness of an NaF tablet preschool program.
       B/C ratio
      One study60 reported both average C/E and B/C ratios.
      2 (12%)
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      ,
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
       Cost per carious surface averted, cost-saving threshold1 (6%)
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
       Cost per event avoided (tooth is not restored or extracted)1 (6%)
      • Chi D.L.
      • van der Goes D.N.
      • Ney J.P.
      Cost-effectiveness of pit-and-fissure sealants on primary molars in Medicaid-enrolled children.
       Cost per incremental change in dmfs1 (6%)
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
       Cost per tooth saved, cost per child saved from caries experience, cost per child saved from extraction experience1 (6%)
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
       Number of avoided (reduced) restorative or surgical treatment visits in the ambulatory dental clinic or operating room at the hospital1 (6%)
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
      ACER indicates average cost-effectiveness ratio; ICER, incremental cost-effectiveness ratio; N/A, not applicable; QALY, quality-adjusted life-year.
      One study
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      reported both average C/E and B/C ratios.

       Cost-Effectiveness/Cost-Benefit Results

      Six of 15 studies that employed CEA concluded that the intervention was cost-effective. The interventions were a complex dental disease management program,
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
      OH education programs,
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      ,
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
      fluoridated milk and milk-cereal,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      and a study comparing 5 different caries prevention interventions.
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.
      In 4 cases, the intervention was cost-effective for certain subgroups or for certain scenarios but not the others.
      • Ramos-Gomez F.J.
      • Shepard D.S.
      Cost-effectiveness model for prevention of early childhood caries.
      ,
      • Stearns S.C.
      • Rozier R.G.
      • Kranz A.M.
      • Pahel B.T.
      • Quinonez R.B.
      Cost-effectiveness of preventive oral health care in medical offices for young Medicaid enrollees.
      ,
      • Donaldson C.
      • Forbes J.F.
      • Smalls M.
      • Boddy F.A.
      • Stephen K.W.
      • McCall D.
      Preventive dentistry in a health centre: effectiveness and cost.
      ,
      • Widenheim J.
      • Birkhed D.
      Caries-preventive effect on primary and permanent teeth and cost-effectiveness of an NaF tablet preschool program.
      In 2 studies on primary molar sealants, the “always seal” intervention was more effective but more costly than standard care.
      • Ney J.P.
      • van der Goes D.N.
      • Chi D.L.
      Economic modeling of sealing primary molars using a “value of information” approach.
      ,
      • Chi D.L.
      • van der Goes D.N.
      • Ney J.P.
      Cost-effectiveness of pit-and-fissure sealants on primary molars in Medicaid-enrolled children.
      Two studies on fluoride varnish indicated that the intervention was not cost-saving.
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      ,
      • Quinonez R.B.
      • Stearns S.C.
      • Talekar B.S.
      • Rozier R.G.
      • Downs S.M.
      Simulating cost-effectiveness of fluoride varnish during well-child visits for Medicaid-enrolled children.
      The authors of a postal toothbrushing program evaluation
      • Davies G.M.
      • Worthington H.V.
      • Ellwood R.P.
      • et al.
      An assessment of the cost effectiveness of a postal toothpaste programme to prevent caries among five-year-old children in the North West of England.
      did not draw any conclusions on its cost-effectiveness.
      Of the 2 studies that employed CBA, 1 study showed that the benefits of a combined hand hygiene and OH promotion program outweighed costs at each discount rate level considered.
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
      The other study compared dental health education with several other caries prevention strategies.
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      The results showed that the dental health education program provided better benefit-costs ratios than other preventive programs.
      The only study that used QALY as an outcome evaluated the cost-effectiveness of a home visit intervention conducted by OH therapists relative to a telephone-based alternative and no intervention.
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      The home visits and telephone interventions resulted in 7 and 6 QALYs, respectively, gained over the usual care group for the 100 children older than 5.5 years. Both interventions were dominant, as they saved costs and produced health benefits over usual care.

       Results of Reporting Quality Assessment of Full EEs

      The results of the assessment of full EE studies using the CHEERS checklist showed substantial variation in reporting quality. Figure 2 shows the proportion of studies that did not meet each criterion. Not every single item was applicable to every study. The items that were most often unmet were #20a, characterizing uncertainty (for single-study–based EEs), with 67% of all studies to which this item was applicable not meeting this criterion; #6, study perspective, with 44% of studies not meeting this criterion; #18, study parameters, 38% not met; and #13a, estimating resources and costs (for single-study–based EEs), 33% not met. Several items were met by all studies to which these items were applicable: items 1 and 2, title and abstract; #10, choice of health outcomes; #11a and #11b, measurement of effectiveness (for single-study–based and model-based EEs, respectively); #13b, estimating resources and costs (model-based EEs); #15, choice of model; #16, assumptions (model); and #21, characterizing heterogeneity (applicable to 2 studies only). Item 12, measurement and valuation of preference-based outcomes, was applicable to 1 study only and was met. Scores by item by study are shown in Appendix Table 4 (Supplementary Material found at https://doi.org/10.1016/j.jval.2020.04.1823).
      Figure thumbnail gr2
      Figure 2Proportion of CHEERS items not met, by item (% of total numbers of studies for which each of the items was applicable). Key: Chequered fill – over 40% of eligible studies did not meet a criterion; Horizontal lines fill – 30% < 40% not met; Dotted fill – 20% < 30% not met; Solid fill – < 20% not met. Notes: 1) The total (100%) was different for various items, as some items were not applicable to all studies. 2) “Other” items, namely “Source of funding” and “Conflicts of interest” were not included when rating the reporting quality of the reviewed papers.
      CHEERS indicates Consolidated Health Economic Evaluation Reporting Standards.
      The overall CHEERS score for each reviewed study is shown in Figure 3. More recent papers were of higher reporting quality than earlier ones. Papers published from 2014 onward met 100% of applicable CHEERS items. For all 16 papers combined, the median proportion of all applicable CHEERS items met was 97.5% and the mean was 81.8%. The range was 50% to 100%. With regard to “other” items (not shown in Fig. 2 and not included in the total score in Fig. 3), only three papers (19%) provided information on their authors’ conflict of interest, whereas 9 papers (56%) indicated a source of funding.
      Figure thumbnail gr3
      Figure 3CHEERS score for each reviewed full EE study (% of total applicable items). Note: “Other” items, namely “Source of funding” and “Conflicts of interest” were not included when rating the reporting quality of reviewed papers.
      CHEERS indicates Consolidated Health Economic Evaluation Reporting Standards; EE, economic evaluation.

      Discussion

      This is the first systematic review to focus on the EEs of OH improvement interventions targeted at the early life. This period has been shown to be crucial in influencing health in later years.
      International Centre for Oral Health Inequalities Research & Policy
      Social Inequalities in Oral Health: From Evidence to Action.
      ,
      • Colak H.
      • Dulgergil C.T.
      • Dalli M.
      • Hamidi M.M.
      Early childhood caries update: A review of causes, diagnoses, and treatments.
      According to a review of publications on EEs of caries prevention programs in all ages,
      • Marino R.J.
      • Khan A.R.
      • Morgan M.
      Systematic review of publications on economic evaluations of caries prevention programs.
      the main methodological problems identified were the limited information provided on adjustments for discounting in addition to inadequate sensitivity analyses. These are similar findings to those of a systematic review of EE publications in dentistry,
      • Tonmukayakul U.
      • Calache H.
      • Clark R.
      • Wasiak J.
      • Faggion Jr., C.M.
      Systematic review and quality appraisal of economic evaluation publications in dentistry.
      which also identified insufficient information on how costs and outcomes had been measured and valued. In addition, a more recent systematic review of EEs in child OH research, which included full EE studies involving children aged 18 years old and under,
      • Rogers H.J.
      • Rodd H.D.
      • Vermaire J.H.
      • et al.
      A systematic review of the quality and scope of economic evaluations in child oral health research.
      highlighted that a wide range of outcome measures was employed across the reviewed studies, which prevented interstudy comparisons. Lack of meaningful involvement of children and of consideration of their own perspectives and preferences were also emphasized.
      The present review has identified that the most widely used type of analysis were cost analysis and CEA, which is similar to the findings of a recent systematic review of EEs in wider child OH research.
      • Rogers H.J.
      • Rodd H.D.
      • Vermaire J.H.
      • et al.
      A systematic review of the quality and scope of economic evaluations in child oral health research.
      More than 60% of the reviewed papers were published between 2000 and 2017 (inclusive). The proportion of full EEs increased over time, especially from 2000 onward. Most studies were conducted in the United States and the United Kingdom. Just less than one-third of the studies reviewed investigated complex multicomponent interventions and approximately one-fifth focused on water fluoridation.
      Unlike the previous reviews,
      • Marino R.J.
      • Khan A.R.
      • Morgan M.
      Systematic review of publications on economic evaluations of caries prevention programs.
      ,
      • Tonmukayakul U.
      • Calache H.
      • Clark R.
      • Wasiak J.
      • Faggion Jr., C.M.
      Systematic review and quality appraisal of economic evaluation publications in dentistry.
      the current review did not find a mismatch between the study descriptor and actual type of analysis used. All studies that were labeled a CEA or a CBA or a combination of methods were indeed those study types. This may be explained by the fact that the vast majority of full EEs in the field of interest were published relatively recently, by which time guidance on EE methods was established and widely used.
      Although the reporting quality of partial EEs was not formally assessed in our systematic review, some parameters, which were included in the data extraction template, indicate that, on the whole, partial EEs were inferior to full EEs in relation to reporting these parameters. For example, more than half of the reviewed partial EEs did not employ sensitivity analysis, whereas three-quarters of full EEs studies did. Most full EEs stated the discount rate used, whereas only 3 partial EE studies did so. In addition, a higher proportion of full EE papers indicated the baseline year for their analysis. Previous reviews identified similar methodological limitations, namely, absence of sensitivity analysis, limited information on adjustments for discounting, and not reporting the base year.
      • Marino R.J.
      • Khan A.R.
      • Morgan M.
      Systematic review of publications on economic evaluations of caries prevention programs.
      • Tonmukayakul U.
      • Calache H.
      • Clark R.
      • Wasiak J.
      • Faggion Jr., C.M.
      Systematic review and quality appraisal of economic evaluation publications in dentistry.
      • Rogers H.J.
      • Rodd H.D.
      • Vermaire J.H.
      • et al.
      A systematic review of the quality and scope of economic evaluations in child oral health research.
      ,
      • Hettiarachchi R.M.
      • Kularatna S.
      • Downes M.J.
      • et al.
      The cost-effectiveness of oral health interventions: a systematic review of cost-utility analyses.
      A significant proportion of the papers did not state the perspective used in the analysis: 83% of partial EEs and 44% of all full EEs, which is similar to the results of a previous systematic review of CUAs of OH interventions.
      • Hettiarachchi R.M.
      • Kularatna S.
      • Downes M.J.
      • et al.
      The cost-effectiveness of oral health interventions: a systematic review of cost-utility analyses.
      The review identified 16 full EEs, which used a variety of EE methods and techniques and OH outcome and economic outcome measures. This variation makes it challenging to compare the cost-effectiveness of individual caries prevention interventions. This concurs with the conclusions of 2 previous systematic reviews.
      • Rogers H.J.
      • Rodd H.D.
      • Vermaire J.H.
      • et al.
      A systematic review of the quality and scope of economic evaluations in child oral health research.
      ,
      • Hettiarachchi R.M.
      • Kularatna S.
      • Downes M.J.
      • et al.
      The cost-effectiveness of oral health interventions: a systematic review of cost-utility analyses.
      Interpretation of cost-effectiveness ratios for dental health outcomes is similar to the standard challenges of using CEA when comparing different outcomes. Without use of an accepted threshold for a generic outcome such as a QALY, comparability is not possible. It is not clear how much the payer (eg, a healthcare system, public payer, or society) is willing to pay per decayed surface/tooth avoided or per child kept caries free.
      • Lord J.
      • Longworth L.
      • Singh J.
      • et al.
      Oral Health Guidance—Economic Analysis of Oral Health Promotion Approaches for Dental Teams.
      Only 1 study used a preference-based health-related quality-of-life measure that allows calculation of QALY as one of the outcomes, which, in turn, allows a comparison of cost-utility results of various interventions’ evaluations. This lack of evidence reveals a clear gap in relation to preschoolers’ OH research.
      More than 40% of the reviewed full EE papers concluded in favor of the intervention(s) under investigation. Nevertheless, there were small numbers of studies investigating each intervention type (eg, fluoride varnish, OH education, dental sealants, toothbrushing, fluoridated food and drinks, water fluoridation). The studies were underpowered,
      • Kowash M.B.
      • Toumba K.J.
      • Curzon M.E.
      Cost-effectiveness of a long-term dental health education program for the prevention of early childhood caries.
      ,
      • Koh R.
      • Pukallus M.
      • Kularatna S.
      • et al.
      Relative cost-effectiveness of home visits and telephone contacts in preventing early childhood caries.
      ,
      • Pukallus M.
      • Plonka K.
      • Kularatna S.
      • et al.
      Cost-effectiveness of a telephone-delivered education programme to prevent early childhood caries in a disadvantaged area: a cohort study.
      used simple spreadsheet-based calculations,
      • Atkins C.Y.
      • Thomas T.K.
      • Lenaker D.
      • Day G.M.
      • Hennessy T.W.
      • Meltzer M.I.
      Cost-effectiveness of preventing dental caries and full mouth dental reconstructions among Alaska Native children in the Yukon-Kuskokwim delta region of Alaska.
      ,
      • Marino R.
      • Morgan M.
      • Weitz A.
      • Villa A.
      The cost-effectiveness of adding fluorides to milk-products distributed by the National Food Supplement Programme (PNAC) in rural areas of Chile.
      ,
      • Ataniyazova R.
      • Negmatov J.
      • Parpiev Z.
      A cost-benefit analysis of early childhood hygiene interventions in Uzbekistan.
      or were pilot studies,
      • Samnaliev M.
      • Wijeratne R.
      • Kwon E.G.
      • Ohiomoba H.
      • Ng M.W.
      Cost-effectiveness of a disease management program for early childhood caries.
      making it challenging to draw reliable conclusions with regard to the value of primary caries prevention.
      The only full EE, conducted alongside a well-powered randomized controlled trial and deemed to be of high reporting quality, was O’Neill et al,
      • O’Neill C.
      • Worthington H.V.
      • Donaldson M.
      • et al.
      Cost-effectiveness of caries prevention in practice: a randomized controlled trial.
      which concluded that the costs of providing a combined fluoride intervention (fluoride varnish, free toothbrush and fluoride toothpaste, and standardized dental health education) in general dental practice settings outweighed savings in treatment over the 3-year follow-up period. This intervention was unlikely to produce a cost-saving.
      The results of our quality assessment of full EEs using the CHEERS checklist showed substantial variation in reporting quality. The items most often unmet were “characterizing uncertainty,” “study perspective,” “study parameters,” and “estimating resources and costs.” Of note, more recently published papers were of higher reporting quality. The CHEERS is the most recently developed EE checklist that was created to update previous guidelines.
      • Husereau D.
      • Drummond M.
      • Petrou S.
      • et al.
      Consolidated Health Economic Evaluation Reporting Standards (CHEERS) statement.
      ,
      • Frederix G.W.J.
      Check your checklist: the danger of over- and underestimating the quality of economic evaluations.
      It has been widely used as a single tool for assessing the quality in systematic reviews of EE studies, including some focusing on OH.
      • Hettiarachchi R.M.
      • Kularatna S.
      • Downes M.J.
      • et al.
      The cost-effectiveness of oral health interventions: a systematic review of cost-utility analyses.
      ,
      • Geisler B.P.
      • Ji Y.D.
      • Peacock Z.S.
      Value in oral and maxillofacial surgery: a systematic review of economic analyses.
      One of the limitations of this systematic review is that because of the time constraints, only 20% of randomly selected records were assessed or checked independently by a second reviewer. The reporting quality was formally assessed for only full EE studies. The CHEERS checklist cannot be meaningfully used for partial EEs assessment, as many of the items are not applicable. In addition, the overall methodological quality of the reviewed studies was not formally assessed.

      Conclusions

      Although the number of EE studies relating to OH improvement interventions in preschoolers has been increasing in recent years, a number of items were inadequately reported in a substantial proportion of the reviewed studies. Our review has highlighted wide variation in (1) types of caries prevention interventions investigated, (2) effectiveness measures used, (3) how costs and outcomes are reported, and (4) study perspective (when indicated).
      Importantly, only 1 study employed CUA, using a preference-based outcome measure. This notable lack of use of preference-based health-related quality-of-life measures in the field of preschoolers’ OH likely reflects the challenges with conducting EE in this young age group, the availability of suitable preference-based measures, and also flags the limitations with the use of these studies for the purposes of decision making in dental healthcare.
      Although variation in prevention interventions investigated is entirely expected, the methodological limitations identified preclude meaningful comparisons across studies as well as compromise the evidence base for strategies in relation to the prevention of this disease in this age group. Because of the small numbers of studies investigating each intervention type and the questionable methodological quality of many of the reviewed EEs, it was not possible to arrive at reliable conclusions with regard to the economic value of primary caries prevention.
      With dental caries being one of the most common diseases affecting humans worldwide, the identification of cost-effective prevention strategies in children should be a global public health priority. This agrees with the recommendations in the recent articles outlining the challenges and priorities for global OH. For this to be achieved, studies should be designed to include EEs using best practice methods guidance and adhering to standards for reporting and presenting. Such improvements to the evidence base will serve to increase both the availability and quality of economic evidence in this important area.

      Acknowledgment

      The authors would like to thank Mr Paul Cannon, College Librarian, Medical, Veterinary, and Life Sciences, University of Glasgow, for his support in developing the literature search strategies.

      Supplemental Material

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