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A scoping review of costing methodologies used to assess interventions for underserved pregnant people and new parents

International Journal for Equity in Health volume 23, Article number: 168 (2024) Cite this article

Abstract

Background

Lack of evidence about the long-term economic benefits of interventions targeting underserved perinatal populations can hamper decision making regarding funding. To optimize the quality of future research, we examined what methods and costs have been used to assess the value of interventions targeting pregnant people and/or new parents who have poor access to healthcare.

Methods

We conducted a scoping review using methods described by Arksey and O’Malley. We conducted systematic searches in eight databases and web-searches for grey literature. Two researchers independently screened results to determine eligibility for inclusion. We included economic evaluations and cost analyses of interventions targeting pregnant people and/or new parents from underserved populations in twenty high income countries. We extracted and tabulated data from included publications regarding the study setting, population, intervention, study methods, types of costs included, and data sources for costs.

Results

Final searches were completed in May 2024. We identified 103 eligible publications describing a range of interventions, most commonly home visiting programs (n = 19), smoking cessation interventions (n = 19), prenatal care (n = 11), perinatal mental health interventions (n = 11), and substance use treatment (n = 10), serving 36 distinct underserved populations. A quarter of the publications (n = 25) reported cost analyses only, while 77 were economic evaluations. Most publications (n = 82) considered health care costs, 45 considered other societal costs, and 14 considered only program costs. Only a third (n = 36) of the 103 included studies considered long-term costs that occurred more than one year after the birth (for interventions occurring only in pregnancy) or after the end of the intervention.

Conclusions

A broad range of interventions targeting pregnant people and/or new parents from underserved populations have the potential to reduce health inequities in their offspring. Economic evaluations of such interventions are often at risk of underestimating the long-term benefits of these interventions because they do not consider downstream societal costs. Our consolidated list of downstream and long-term costs from existing research can inform future economic analyses of interventions targeting poorly served pregnant people and new parents. Comprehensively quantifying the downstream and long-term benefits of such interventions is needed to inform decision making that will improve health equity.

Background

Despite improvements in perinatal outcomes in high income countries, socio-economic disparities in outcomes persist. Inequity in perinatal outcomes arises as a result of structural factors that cause social inequality, which in turn impacts the social and health outcomes associated with pregnancy and early parenting through a variety of pathways [1]. Various terminology has been used to describe groups or populations at increased risk of poor perinatal outcomes as a consequence of social inequality, with common terms including ‘vulnerable’, ‘disadvantaged’, or ‘marginalized’ populations. These terms have been critiqued because they are potentially stigmatizing and conceptualize the locus of inequities as arising from individual flaws or deficits [2]. We therefore avoid these terms and instead intentionally conceptualize our focus to be people who are negatively impacted by health and social system factors that limit their access to optimal care to support good perinatal outcomes. In this research we describe the people of interest as those who are poorly served by healthcare systems.

Interventions aimed at addressing disparities in perinatal outcomes are important to redress health inequities and because they potentially have long-term benefits for both the pregnant person and their offspring [3]. The perinatal period offers an opportunity to identify and mitigate the risks to long-term maternal health [4] Additionally, the growing body of evidence on the developmental origins of health and disease has shown that health in pregnancy has a long-term impact on physical health of the offspring [5]. Likewise, research on child development has established that healthy attachment in early childhood has long term impact on emotional well-being and mental health [6]. However, the measurement of long-term outcomes in prospective studies of interventions is often costly, and for that reason may not occur. This can create challenges for funders who need to make decisions about whether it is worthwhile to invest in interventions, as lack of evidence can hamper the calculation of long-term economic benefits associated with the improvements in health and social outcomes.

One type of intervention that has shown promise with respect to improving the pregnancy and early parenting outcomes of populations who are poorly served by healthcare is midwifery-led care [7, 8]. There is a growing body of evidence to support the potential benefits of midwifery-led care for poorly served populations [9–11]. However, we identified a gap in the research with respect to economic evaluation of these kinds of interventions. In order to inform a larger research project aimed at developing a framework to assess the cost implications of midwifery care models that target underserved populations, we conducted a scoping review of the peer-reviewed and grey literature to synthesize previous approaches used to examine the short- and long-term cost implications of interventions targeting underserved pregnant people and new parents. Our research question was what types of costs and costing methodology have been used to assess interventions targeting pregnant people and/or new parents who have poor access to healthcare?

Methods

We conducted a scoping review in accordance with the methodology described by Arksey and O’Malley (2005) [12], including the following five stages: (1) identifying the research question; (2) identifying relevant studies; (3) study selection; (4) charting the data; (5) collating, summarizing, and reporting the results. We selected a scoping review as the best approach to systematically identify all previous economic and cost analyses, including both peer-reviewed and grey literature, that examine the cost implications of interventions targeting underserved pregnant people and/or new parents. The methodology allowed us to compare the range of analytical approaches used to evaluate costs to inform the development future cost analysis frameworks.

Identifying relevant studies

We developed a comprehensive search strategy for peer-reviewed and grey literature in consultation with a research librarian at the McMaster Health Sciences Library. The librarian provided guidance on the selection of keywords, refinement of the search strategy, and identification of relevant databases to search. Search terms related to ‘underserved populations,’ ‘economic evaluations,’ and ‘pregnant people and new parents’ were used to structure the search. Our search was not limited by year or language of publication. We drew on concurrent research we were conducting to scope the research literature regarding populations who have poor access to sexual and reproductive health care to define ‘underserved populations’ taking a broad approach. The full search strategy is included in Appendix A, which provides details regarding what populations were considered ‘underserved.’

We searched the following peer-reviewed academic literature databases: Ovid MEDLINE, Ovid Emcare, EMBASE (Ovid), Ovid Healthstar, Cochrane Library, Cumulative Index to Nursing and Allied Health Literature (CINAHL) EBSCO, EconLit EBSCO, and Business Source Premier EBSCO. To identify relevant grey literature, AJ hand-searched the websites of relevant organizations (e.g., Nurse Family Partnership, The Canada Prenatal Nutrition Program (CPNP), and Special Supplemental Nutrition Program for Women, Infants, and Children (WIC)). In addition, we reviewed the reference lists of key publications and relevant review articles to identify further sources. We imported the citation information for the retrieved publications into the citation management software, EndNote, and removed duplicate records prior to screening the results.

Study selection

We screened the retrieved publications for inclusion or exclusion using the systematic review software, Covidence, to manage the screening process. We first screened records by title and abstract against the inclusion and exclusion criteria. Second, we retrieved publications that passed the first stage of screening to screen the full text. Two reviewers (AJ, and RG or BJ or BA) independently screened the articles at both stages. We flagged any conflicts between researchers at either stage in Covidence and resolved difference through discussion. A third reviewer (ED) was consulted when consensus could not be reached.

Inclusion and exclusion criteria

To be included in this review, the publication needed to have: (1) described an intervention targeting pregnant people and/or new parents; (2) targeted an underserved population; (3) included a cost analysis of the intervention; and (4) been set in one of the top 20 Gross Domestic Product (GDP) per capita Organisation for Economic Co-operation and Development (OECD) countries in USD in 2020 [2, 13]. The purpose of the fourth criterion was to ensure that the research was conducted in countries with a similar context, i.e., a high-income economy and high Human Development Index (HDI). Included countries were Ireland, Luxembourg, Norway, United States, Switzerland, Denmark, Belgium, Sweden, Austria, Germany, France, Netherlands, Iceland, Finland, United Kingdom, Canada, Turkey, Australia, Italy, and Spain. A study was excluded if: (1) the goal of the described intervention was the prevention or termination of pregnancy; or (2) it was a review-level publication. We retained relevant reviews and hand-searched the reference lists for original studies meeting our criteria.

Charting the data

We charted relevant data from the publications identified for inclusion in a Microsoft Excel spreadsheet. Initially, two reviewers (AJ, RG) conducted data extraction independently, compared their results, and resolved disagreements by consensus. Both reviewers extracted data from approximately 10% of the included articles, until a high level of agreement was achieved. Then one reviewer (AJ) extracted the remaining data independently. Extracted data included publication details (i.e., authors, title, journal), study design, setting (location, duration), population characteristics, description of the intervention, type of economic evaluation conducted, types of costs included in the economic analysis, and sources of cost data.

Collating, summarizing, and reporting the results

Once data extraction and charting were completed, the team reviewed the findings to determine the most useful way to collate the results and synthesize key findings. We created tables summarizing the interventions, the analytical approaches used to calculate costs (e.g. cost -benefit, cost-minimization, cost effectiveness, cost utility, cost-consequences, or costing analyses) and consolidated a list of costs considered in the analyses (i.e., program costs, health care costs, societal costs) as well as whether immediate, short or long-term costs were determined. We used descriptive statistics, such as frequencies and percentages, to summarize the data and calculated these using Microsoft Excel.

Results

Final searches were completed on May 28, 2024. Figure 1 summarizes the results of our searches. We identified a total of 4866 publications from the peer-reviewed databases, all of which were in English. After removing duplicates, we identified 2507 publications for screening. We included 65 publications identified through database searches, 28 publications identified through reference chaining, and 11 publications identified through targeted web-searches. The final number of articles included in the review from these three sources was 104.

Fig. 1
figure 1

PRISMA diagram

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A descriptive summary of the included articles is presented in Table 1. Sixty-four (n = 64) of the publications focused on interventions in the United States [14–77], 29 in the United Kingdom [78–106], six in Australia [107,108,109,110,111,112], three in Canada [113,114,115], and one in each Sweden [116] and Germany [116]. The interventions described in these publications targeted pregnant people (n = 50), new parents (n = 16), or both (n = 37). Thirty-six (n = 36) underserved populations were targeted by the interventions, including people with low income, who were targeted in 35 interventions; people who smoke, who were targeted in 20 interventions; Medicaid recipients, who were targeted in 15 interventions; and people who use substances, who were targeted in 11 interventions. The publications described a range of interventions, most commonly home visiting programs (n = 19), smoking cessation interventions (n = 19), prenatal care (n = 11), perinatal mental health interventions (n = 11), and substance use treatment (n = 10). Of the 50 interventions that began in or continued into the postpartum period, the most common duration of interventions was between six months to two years postpartum (n = 30). Thirty-seven (n = 37) interventions were delivered in part or in full in the client’s home, 21 in the hospital, 21 digitally, and 16 in primary care clinics. Nurses (n = 33), midwives or nurse-midwives (n = 21), social workers (n = 11), and family physicians (n = 9) were the professionals most commonly involved in delivering the interventions.

Table 1 Study characteristics
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Table 2 lists all 33 cost categories that were used in the publications, grouped into program costs, health care costs, and other societal costs. Sixteen (n = 16) studies considered only program costs. Most publications considered health care costs (n = 82). While 45 studies considered costs that we labelled ‘other societal costs’, these were frequently immediate costs incurred during the time frame of the intervention (e.g., costs of subsidies and social services). A third (n = 35) of the included studies considered long-term costs that occurred more than one year after birth (for interventions occurring only in pregnancy) or after the end of the intervention.

Table 2 Categories of costs considered in included studies
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Table 3 summarizes the types of analytical approaches described in the included publications, as well as the sources of cost data, the types of costs considered, and the timeframe for included cost outcomes. The most common analytic approaches to determine the costs were through cost-effectiveness analyses (n = 43), followed by costing analyses (n = 28) and cost benefit analyses (n = 19). Cost utility (n = 8), cost consequence (n = 4) and cost-minimization (n = 1) analyses were less commonly used. The majority of analyses drew cost data from administrative health data sources (n = 77) and directly measured program costs (n = 58). Costs were also commonly sourced from the literature (n = 51). Hospital costs (n = 70) and salaries and wages (n = 62) were the most common categories of costs considered. Thirty (n = 30) analyses included costs occurring only within the duration of the intervention (immediate); 37 also included costs that occurred up to one year after birth or after the end of the intervention (short-term); and 36 also included costs that occurred longer than one year after birth or after the end of the intervention (long-term).

Table 3 Analytical approaches
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Discussion

The purpose of this review was to report on the state of the literature with respect to economic evaluations of interventions targeting pregnant people and/or new parents and to summarize the methods and costs used in the included studies. The 104 articles included in this review show that a range of methods, costs, and timeframes have been used to assess the value of interventions targeting pregnant people and new parents. Research on home visiting programs was most likely to consider long-term outcomes, and studies looking at long-term outcomes tended to take a wider view of the downstream costs and savings by considering long-term societal costs (e.g., costs related to child apprehension, abuse or neglect, crime, education, employment, lost productivity, addiction, etc.) in addition to program and health care costs. While we identified some economic evaluations that considered a comprehensive set of long-term societal costs, our assessment of the literature demonstrates that evaluations of interventions targeting underserved pregnant people and/or new parents frequently do not consider costs related to long-term outcomes that are necessary to assess their true value for money.

Decision-makers who fund programs targeting underserved populations need evidence to assess the potential value of investing in such programs, and the evidence gap we identified is a barrier to sustaining and spreading interventions that have the potential to improve health equity. The term value for money refers to finding an ideal balance between economy (costs), efficiency (minimizing the ratio of input to output), and effectiveness (achieving the desired outcomes), or the “three Es” [117]. Authors of several of the studies included in our scoping review acknowledge that because long-term or societal costs were not considered, their calculations represent only a small part of the economic benefit to women, infants, and families and this was often noted as a limitation of the study [41, 42, 54, 75, 87, 89]. Some authors called for further research considering these types of costs when the data or resources were available [25,26,27]. Many authors speculated that due to the limited time frame or scope of benefits considered, their calculations were likely an underestimation of the true long-term value for money of the intervention [16, 19, 21, 32, 50, 63].

Our findings align with those of other systematic reviews of economic evaluations of similar interventions [118,119,120,121,122]. In their review of economic evaluations of home visiting programs for young or vulnerable pregnant women, Stamuli et al. noted that many studies considered outcomes only within the length of the trial (what we have described as the immediate time frame) [121]. They argue that for interventions targeting vulnerable pregnant people, the benefits are expected to accrue over the lifetime of the child and the parent rather than in the immediate time frame. They recommend that these types of evaluations consider multiple perspectives, including a societal perspective, and long-term outcomes.

Ruger and Lazar (2012) systematically reviewed economic evaluations of drug abuse treatment and HIV prevention programs in pregnant women [119]. They echoed Stamuli et al.’s recommendation that costs should be reported from a societal perspective [121]. Neither Stamuli et al. [121] nor Ruger and Lazar [119] were able to reach conclusions on whether the interventions represented value for money from the results of the studies, citing a lack of consistency between studies with respect to costs considered, methods used, and perspectives taken. Both articles recommend that for best practice, detailed cost and outcome data should be collected alongside randomized control trials.

Verbeke et al.’s 2022 systematic review of the cost-effectiveness of mental health interventions during pregnancy and up to two years postpartum [122], Koegl et al.’s 2023 review of cost-benefit analyses of developmental crime prevention programs [118], and Sampaio et al.’s 2024 systematic review of interventions aimed at improving child health [120] all concluded that these interventions represented value for money. However, these authors all noted significant limitations in the literature including an overall shortage of published evaluations [118, 122], lack of consistency in methods [118, 120, 122], a deficit of data looking at outcomes for both parents and the child together [122], and insufficient analysis of the long-term effectiveness of the included interventions [118, 120, 122]. For future research, they recommend considering a broader range of outcomes for both parents as well as the child over a longer time frame. Like Stamuli et al., Verbeke et al. noted that by not considering the impact over the lifetime of the child, the real-world cost-effectiveness is likely underestimated [122].

Our results will be of value to people interested in a wide range of interventions and may assist those planning and conducting economic evaluations to improve the quality of such research. Readers can use the information provided in Table 3 to identify publications that considered long-term outcomes and societal costs and can refer to the original publications for further details regarding definitions and data sources for such costs to aid in planning their own analyses. Those planning evaluations of interventions targeting underserved pregnant people and/or new parents should collect prospective long-term outcome data pertaining to both parent and child outcomes whenever possible. Long-term health outcomes and long-term health care costs should be considered. Important long-term societal costs to consider, where relevant, include child welfare services, legal services, incarceration, other criminal justice costs, costs of crime, domestic violence, child abuse/neglect, addiction, lost productivity, employment status/income, housing, education, and earning potential. We noted a gap in the existing literature with respect to the consideration of patient priorities in the identification of costs considered. This might be addressed through patient engagement when planning economic evaluations, and through the inclusion of costs that have been identified a patient-oriented outcome measures for economic research [123]. Our findings suggest that economic evaluations that consider multiple perspectives, including a societal perspective, and a long-term time horizon will most fully describe the value for money of interventions underserved pregnant people and/or new parents. Inclusion of a patient perspective evaluation may also better address equity considerations [124].

Our scoping review is the first of its kind and has several strengths. Our method allowed us to consider grey literature and include studies of all methodologies while maintaining a systematic approach. We looked at a wider range of interventions and underserved populations than previous reviews. Another strength of our review is the extensive set of search terms used to identify underserved populations, which minimized the possibility of missing eligible articles. One limitation of our review is that we did not extract detailed information about the economic analysis methods that were used, including the outcomes associated with the full economic evaluations (e.g., quality-adjusted-life-years for cost-utility analyses, net benefit for cost-benefit analyses) and time horizons considered. Another limitation is that we did not conduct the optional stakeholder consultation step in the Arksey and O’Malley (2005) scoping review framework [12]. This step is most commonly conducted in the search phase to inform keyword selection [125]. Based on another review conducted as part of our larger program of research, we compiled an extensive set of search terms to identify relevant studies and felt confident that omission of stakeholder consultation did not sacrifice the quality of our review. Secondly, due to the nature of the scoping review methodology, we did not appraise the quality of the methods or results of the studies included in this review. Limitations and methodological concerns about some of the articles included in this review have been reported previously [34, 57, 121]. Lastly, in order to ensure included articles were relevant to our context, we did not include publications focused on interventions set outside of the top 20 GDP OECD countries. This means that the findings from this article, including the particular downstream costs considered in the analyses, may not be applicable to low- and middle-income countries.

Conclusion

This scoping review can be used to inform future economic analyses of interventions targeting poorly served pregnant people and new parents. It consolidates a list of costs that researchers and health economists may wish to consider when conducting these types of analyses and identifies studies that look at a range of downstream and long-term costs. It is important to include all costs relevant to the underserved population in question in the evaluation. Policy and resource allocation decisions are often informed by evidence generated through economic evaluations to ensure the efficient allocation of available resources. Economic evaluations that are able to capture the downstream and long-term benefits of interventions targeting underserved populations may support the implementation of policy and funding decisions that will benefit underserved pregnant people and new parents, their children, and society in general and that will reduce health inequities.

Availability of data and materials

The datasets used and/or analysed during the current study are available from the corresponding author on reasonable request.

Abbreviations

OVID:

Ovid Emcare, EMBASE

CINAHL:

Cumulative Index to Nursing and Allied Health Literature

CPNP:

The Canada Prenatal Nutrition Program

WIC:

Special Supplemental Nutrition Program for Women, Infants, and Children

GDP:

Gross Domestic Product

OECD:

Organisation for Economic Co-operation and Development

HDI:

Human Development Index

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Acknowledgements

We acknowledge the contributions of Riley Graybrook to the data acquisition process and Bella Arless to updating the searches in 2024, screening results, and extracting data, and the support of Lindsay Grenier in preparing the final manuscript for submission. We thank Emmanuel Guindon for his review of an earlier version of the manuscript.

Funding

This research was funded by a grant from the Canadian Institutes of Health Research (#MRC-167971) awarded to Elizabeth Darling (PI). Aisha Jansen was also supported by a Mitacs Research Training Award. The funders had no role in study design; in the collection, analysis and interpretation of data; in writing of the report; or in the decision to submit the article for publication.

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  1. McMaster Midwifery Research Centre, McMaster University, 1280 Main St. W., HSC 4H24, Hamilton, ON, L8S 4K1, Canada

    Elizabeth K. Darling, Aisha Jansen & Bismah Jameel

  2. Department of Health Research Methods, Evidence, and Impact, Communication Research Lab (CRL) 227, McMaster University, 1280 Main Street West, Hamilton, ON, L8K 4K1, Canada

    Jean-Éric Tarride

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  1. Elizabeth K. Darling
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Contributions

The study was conceptualized by E.D. E.D., J-E.T., and A.J. contributed to the study design. A.J. and B.J. conducted the study selection and full text screening, and A.J. completed the data extraction. E.D. was consulted when consensus could not be reached. The findings were collated and summarized by A.J. The first draft of the manuscript was written by A.J. and E.D. All authors critically revised the manuscript. All authors read and approved the final manuscript.

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Correspondence to Elizabeth K. Darling.

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Darling, E.K., Jansen, A., Jameel, B. et al. A scoping review of costing methodologies used to assess interventions for underserved pregnant people and new parents. Int J Equity Health 23, 168 (2024). https://doi.org/10.1186/s12939-024-02252-x

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International Journal for Equity in Health

ISSN: 1475-9276

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