Economic Evaluations of Digital Health Interventions for Children and Adolescents: Systematic Review

Background Digital health interventions (DHIs) are defined as digital technologies such as digital health applications and information and communications technology systems (including SMS text messages) implemented to meet health objectives. DHIs implemented using various technologies, ranging from electronic medical records to videoconferencing systems and mobile apps, have experienced substantial growth and uptake in recent years. Although the clinical effectiveness of DHIs for children and adolescents has been relatively well studied, much less is known about the cost-effectiveness of these interventions. Objective This study aimed to systematically review economic evaluations of DHIs for pediatric and adolescent populations. This study also reviewed methodological issues specific to economic evaluations of DHIs to inform future research priorities. Methods We conducted a database search in PubMed from 2011 to 2021 using the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) checklist. In total, 2 authors independently screened the titles and abstracts of the search results to identify studies eligible for full-text review. We generated a data abstraction procedure based on recommendations from the Panel on Cost-Effectiveness in Health and Medicine. The types of economic evaluations included in this review were cost-effectiveness analyses (costs per clinical effect), cost-benefit analyses (costs and effects expressed in monetary terms as net benefit), and cost-utility analyses (cost per quality-adjusted life year or disability-adjusted life year). Narrative analysis was used to synthesize the quantitative data because of heterogeneity across the studies. We extracted methodological issues related to study design, analysis framework, cost and outcome measurement, and methodological assumptions regarding the health economic evaluation. Results We included 22 articles assessing the cost-effectiveness of DHI interventions for children and adolescents. Most articles (14/22, 64%) evaluated interventions delivered through web-based portals or SMS text messaging, most frequently within the health care specialties of mental health and maternal, newborn, and child health. In 82% (18/22) of the studies, DHIs were found to be cost-effective or cost saving compared with the nondigital standard of care. The key drivers of cost-effectiveness included population coverage, cost components, intervention effect size and scale-up, and study perspective. The most frequently identified methodological challenges were related to study design (17/22, 77%), costing (11/22, 50%), and economic modeling (9/22, 41%). Conclusions This is the first systematic review of economic evaluations of DHIs targeting pediatric and adolescent populations. We found that most DHIs (18/22, 82%) for children and adolescents were cost-effective or cost saving compared with the nondigital standard of care. In addition, this review identified key methodological challenges directly related to the conduct of economic evaluations of DHIs and highlighted areas where further methodological research is required to address these challenges. These included the need for measurement of user involvement and indirect effects of DHIs and the development of children-specific, generic quality-of-life outcomes.


Information sources
6 Specify all databases, registers, websites, organisations, reference lists and other sources searched or consulted to identify studies.Specify the date when each source was last searched or consulted.
page 4 Search strategy 7 Present the full search strategies for all databases, registers and websites, including any filters and limits used.pages 4-5, Textbox 1 Selection process 8 Specify the methods used to decide whether a study met the inclusion criteria of the review, including how many reviewers screened each record and each report retrieved, whether they worked independently, and if applicable, details of automation tools used in the process.pages 4-5; Figure 1 Data collection process 9 Specify the methods used to collect data from reports, including how many reviewers collected data from each report, whether they worked independently, any processes for obtaining or confirming data from study investigators, and if applicable, details of automation tools used in the process.
pages 4-5 Data items 10a List and define all outcomes for which data were sought.Specify whether all results that were compatible with each outcome domain in each study were sought (e.g. for all measures, time points, analyses), and if not, the methods used to decide which results to collect.pages 4-5; Textbox 2 10b List and define all other variables for which data were sought (e.g.participant and intervention characteristics, funding sources).Describe any assumptions made about any missing or unclear information.pages 4-5; Textbox 2

Study risk of bias assessment
11 Specify the methods used to assess risk of bias in the included studies, including details of the tool(s) used, how many reviewers assessed each study and whether they worked independently, and if applicable, details of automation tools used in the process.
n/a (not all studies were based on individual patient data; quality assessment focused on methodological issues rather than risk of bias itself) Effect measures 12 Specify for each outcome the effect measure(s) (e.g.risk ratio, mean difference) used in the synthesis or presentation of results.
pages 4-5, Textbox 2, Table 2 Synthesis methods 13a Describe the processes used to decide which studies were eligible for each synthesis (e.g.tabulating the study intervention characteristics and comparing against the planned groups for each synthesis (item #5)).
page 4; Figure 1 13b Describe any methods required to prepare the data for presentation or synthesis, such as handling of missing summary statistics, or data conversions.
pages 4-5 13c Describe any methods used to tabulate or visually display results of individual studies and syntheses.page 5, Textbox 2

Section and Topic
Item # Checklist item Location where item is reported 13d Describe any methods used to synthesize results and provide a rationale for the choice(s).If meta-analysis was performed, describe the model(s), method(s) to identify the presence and extent of statistical heterogeneity, and software package(s) used.
Narrative synthesis.No metaanalysis conducted due to high heterogeneity across studies 13e Describe any methods used to explore possible causes of heterogeneity study results (e.g.subgroup analysis, meta-regression).
n/a (due to high heterogeneity across studies) 13f Describe any sensitivity analyses conducted to assess robustness of the synthesized results.n/a Reporting bias assessment 14 Describe any methods used to assess risk of bias due to missing results in a synthesis (arising from reporting biases).
No missing results

Certainty assessment
15 Describe any methods used to assess certainty (or confidence) in the body of evidence for an outcome.n/a

Study selection 16a
Describe the results of the search and selection process, from the number of records identified in the search to the number of studies included in the review, ideally using a flow diagram.
page 6; Figure 1 16b Cite studies that might appear to meet the inclusion criteria, but which were excluded, and explain why they were excluded.pages 6-7; Table 1 Study characteristics 17 Cite each included study and present its characteristics.Table 1, Table 2 Risk of bias in studies 18 Present assessments of risk of bias for each included study.Table 3 (see Item 11)

Results of individual studies
19 For all outcomes, present, for each study: (a) summary statistics for each group (where appropriate) and (b) an effect estimate and its precision (e.g.confidence/credible interval), ideally using structured tables or plots.
Table 2 Results of syntheses 20a For each synthesis, briefly summarise the characteristics and risk of bias among contributing studies.pages 6-8; Table 2 20b Present results of all statistical syntheses conducted.If meta-analysis was done, present for each the summary estimate and its precision (e.g.confidence/credible interval) and measures of statistical heterogeneity.
explain any amendments to information provided at registration or in the protocol.n/a Support 25 Describe sources of financial or non-financial support for the review, and the role of the funders or sponsors in the review. of the following are publicly available and where they can be found: template data collection forms; data extracted from included studies; data used for all analyses; analytic code; any other materials used in the review.none of these are publicly available.From: Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al.The PRISMA 2020 statement: an updated guideline for reporting systematic reviews.BMJ 2021;372:n71.doi: 10.1136/bmj.n71