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PLOS One logoLink to PLOS One
. 2022 Oct 26;17(10):e0275656. doi: 10.1371/journal.pone.0275656

Societal cost of nine selected maternal morbidities in the United States

Sasigant So O’Neil 1,*, Isabel Platt 1, Divya Vohra 1, Emma Pendl-Robinson 1, Eric Dehus 1, Laurie Zephyrin 2, Kara Zivin 1
Editor: Emily W Harville3
PMCID: PMC9603953  PMID: 36288323

Abstract

Objective

To estimate the cost of maternal morbidity for all 2019 pregnancies and births in the United States.

Methods

Using data from 2010 to 2020, we developed a cost analysis model that calculated the excess cases of outcomes attributed to nine maternal morbidity conditions with evidence of outcomes in the literature. We then modeled the associated medical and nonmedical costs of each outcome incurred by birthing people and their children in 2019, projected through five years postpartum.

Results

We estimated that the total cost of nine maternal morbidity conditions for all pregnancies and births in 2019 was $32.3 billion from conception to five years postpartum, amounting to $8,624 in societal costs per birthing person.

Conclusion

We found only nine maternal morbidity conditions with sufficient supporting evidence of linkages to outcomes and costs. The lack of comprehensive data for other conditions suggests that maternal morbidity exacts a higher toll on society than we found.

Policy implications

Although this study likely provides lower bound cost estimates, it establishes the substantial adverse societal impact of maternal morbidity and suggests further opportunities to invest in maternal health.

Introduction

Maternal morbidity encompasses multiple physical and psychological health conditions that result from or are aggravated by pregnancy [1]. These conditions can start during pregnancy or within a year after delivery. The long-term effects, however, can last months or years, ranging from short acute episodes to longer chronic ailments [2]. These conditions do not necessarily lead to maternal mortality, but they can negatively affect quality of life. The Centers for Disease Control and Prevention identifies 21 severe maternal morbidity indicators, such as blood transfusion, eclampsia, hysterectomy, and sepsis, using hospital discharge data from delivery [1]. The occurrence of severe maternal morbidity has approximately doubled over a 15-year period, affecting 1.4% of birthing people during delivery in the United States [1, 3, 4].

Maternal mortality is the most serious consequence of maternal morbidity. The United States has a maternal mortality ratio of 20 deaths per 100,000 pregnancies, the worst of high-income countries [5]. Other disabilities and chronic illness stemming from maternal morbidity—often referred to as near misses—can have ongoing and compounded effects on a birthing person, their children, and other household members, shaping their workforce participation, nutrition, schooling, and other factors affecting quality of life [68]. Maternal morbidity can lead to adverse outcomes for birthing people, such as cesarean delivery and stroke, and adverse outcomes for their children, such as asthma, preterm birth, and suboptimal breastfeeding [7, 914].

Because of the many conditions associated with maternal morbidity, few studies have attempted to comprehensively estimate its overall costs [15]. This paper estimates the cost of nine maternal morbidity conditions with strong underlying evidence linking them to pregnancies or live births in the United States. We follow each maternal–child pair in the 2019 birth cohort from pregnancy to five years postpartum to highlight near-term economic impacts likely to be most salient to policymakers. To our knowledge, this cost model represents the most comprehensive analysis to date of the economic costs of maternal morbidity in the United States.

This model estimates the costs for select conditions associated with maternal morbidity, though one should interpret results with caution. Our estimate could represent only a lower bound of overall costs because only nine conditions had sufficient information to link to outcomes and costs. At the same time, our results might overestimate certain costs because some birthing people may experience comorbidities (that is, complexities that are not captured in our model). Our model calculates an estimate of the combined independent costs of nine selected maternal morbidities, supplying initial evidence to inform policy.

Our study refers to anyone who has experienced a pregnancy as a “birthing person.” We use the term “maternal morbidity” to describe the adverse medical conditions experienced by birthing people.

Methods

This study used a similar approach to that of Luca et al. [16], who quantified the economic impact of untreated maternal mental health conditions (MMHCs) in the United States. We started with the CDC’s list of 21 severe maternal morbidity indicators, which defines a subset of maternal morbidity conditions through diagnosis codes, and added ten other non-life threatening adverse perinatal medical and mental health conditions directly resulting from pregnancy based on expert recommendations [1]. Through the data extraction process, described in the literature review section below, we narrowed the scope of our analysis to nine maternal morbidity conditions with supporting evidence of subsequent outcomes and associated costs.

We define societal cost as the combined estimated excess medical and nonmedical costs from maternal morbidity for birthing people and their children based on a conceptual model (Fig 1). Medical costs include those directly incurred through the health care delivery system, such as hospitalization costs for birthing people and their children. Nonmedical costs include costs incurred outside the health care delivery system such as productivity loss and absenteeism (that is, missing days of work). To avoid overestimating the risk of outcomes associated with maternal morbidity, our conceptual model focused on primary outcomes that are directly associated with maternal morbidity conditions. For example, infants who experience suboptimal breastfeeding—one of our modeled outcomes—have a higher risk of death from sudden infant death syndrome. We did not include the cost of this secondary outcome in our model because we already include sudden infant death syndrome as a primary outcome of MMHCs, one of the nine maternal morbidity conditions we studied.

Fig 1. Conceptual framework of our model that calculates the costs of outcomes attributed to nine maternal morbidity conditions, 2019 birth cohort.

Fig 1

Our model estimated the excess medical and nonmedical costs of maternal and child outcomes associated with maternal morbidity conditions.

Literature review

To identify the maternal morbidity conditions and subsequent outcomes we used in our model, we conducted a comprehensive literature review following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (Fig 2) [17]. We conducted three searches: (1) the prevalence or incidence of each maternal morbidity condition, (2) the likelihood of adverse medical and nonmedical outcomes associated with maternal morbidity, and (3) the associated medical and nonmedical costs of each outcome. We searched key databases to identify original articles and meta-analyses published in peer-reviewed journals, restricting the searches to articles published from 2010 to 2021. We supplemented these articles with grey literature and other reports focusing on the impacts and costs of maternal morbidity.

Fig 2. Title: Preferred reporting items for systematic reviews and meta-analyses flowchart of article selection for final model, using data primarily from 2010 to 2020.

Fig 2

This figure details the number of articles we found in each of our three searches, the number we selected for full-text review after screening title and abstracts, the number of articles we excluded from the model for each reason, and the final number of articles identified for model inclusion.

From the results of our database searches, we reviewed titles, abstracts, and full text to determine high-quality estimates for our model using the following criteria: (1) study used adequate controls to address confounding factors, (2) outcomes had direct associations with maternal morbidity conditions, (3) outcomes were quantifiable in monetary terms, and (4) incidence and prevalence estimates and medical costs came from the United States. To ensure that we modeled maternal morbidity–outcome connections with strong evidence, we included only connections with evidence in three or more studies and that had at least one cost estimate for the outcomes (either from the Healthcare Cost and Utilization Project’s Nationwide Inpatient Sample or the literature) [18]. We narrowed our original 31 maternal morbidity conditions to nine for inclusion in the model, five of which represent severe maternal morbidity conditions. Details of the literature search strategy appear in S1 Appendix.

Baseline rates

We obtained the baseline incidence or prevalence rates for maternal morbidity conditions using the most recent and relevant statistics available, relying primarily on the Healthcare Cost and Utilization Project’s Nationwide Inpatient Sample. When estimates were unavailable through published reports using these data, we drew from other government agencies or peer-reviewed studies from our literature review. We used a similar process to identify the baseline incidence or prevalence rates for the outcomes associated with maternal morbidity. When possible, we limited the population to pregnant and postpartum people and children through age five. S2 Appendix contains the maternal morbidity and outcome incidence and prevalence estimates we used in the main model.

Cost estimates

To calculate the cost of maternal morbidity in the United States, we estimated the average incremental cost per birthing person or child for each outcome attributable to a maternal morbidity condition. Each outcome resulted in medical costs (such as a longer stay for a delivery hospitalization), nonmedical costs (such as lost wages), or some combination of them. We used the most recent and relevant estimates from the Agency for Healthcare Research and Quality, CDC, and other government agencies along with estimates from peer-reviewed articles from our literature review. Although the severity of each outcome can differ by person, we used the average estimates from the literature to populate our model. We standardized the costs to annual units and converted them to 2019 dollars using the medical component of the Consumer Price Index. S3 Appendix describes the sources we used to obtain cost estimates for maternal morbidity outcomes.

Modeling

We created a model that estimated the costs of maternal morbidity among birthing people and their children for the 2019 birth cohort through five years postpartum, the period most salient to policymakers and fiscal funding time periods. Our key parameters included the following:

  1. Number of pregnancies or live births in the United States in 2019, depending on whether the outcome from the condition occurred during pregnancy, at delivery, or postpartum

  2. Prevalence of each maternal morbidity condition

  3. Incidence of each outcome associated with a maternal morbidity condition

  4. Impact estimates of each morbidity–outcome connection, such as the odds of a preterm birth given a maternal hemorrhage

  5. Documented medical and nonmedical costs for each outcome (understanding that costs vary by severity of each condition or outcome)

  6. Medical inflation rate and discount rate for all conditions, and remission rate for MMHCs

For each maternal morbidity condition associated with a given outcome, we identified the highest and lowest impact estimate from our literature search results as our range and used the midpoint from across the papers as the main estimate. If we identified only one estimate that met our inclusion criteria after further review of the studies, we used the 95% confidence interval as the high and low estimates. This strategy allowed us to include the full range of estimates meeting our criteria in the model. S4 Appendix contains the full set of impact estimates used in our modeling, and S5 Appendix contains the full set of model parameters.

Our model used the input parameters to calculate the medical and nonmedical costs of each morbidity–outcome connection, which reflect the number of birthing people or children who would experience an outcome directly because of a maternal morbidity condition. For each outcome, we used context from the literature to assume whether the costs occurred once (such as stillbirth) or on an ongoing basis (such as juvenile onset type 1 diabetes). S6 Appendix contains more detail on how we calculated the annual incremental excess costs of each outcome.

Following the U.S. Public Health Service’s recommendations for cost models, we discounted costs at an annual rate of 3% to reflect the lower economic value of future expenses and accounted for increases in prices of healthcare services and commodities across time [19]. For medical costs, we used the medical care component of the Consumer Price Index to adjust prices to 2019 dollars. For non-medical costs, we used the Consumer Price Index for all items, less food and energy, to adjust prices to 2019 dollars [20]. We added the medical and nonmedical costs associated with each maternal morbidity condition to estimate the total societal costs of nine maternal morbidity conditions for the 2019 birth cohort through five years postpartum.

We conducted a series of deterministic sensitivity analyses to understand the sensitivity of our cost model to variation in prevalence, cost, and impact estimates, and we identified which model parameters had the greatest effect on changes to the total societal cost of maternal morbidity. We varied each incidence or prevalence rate individually using the high and low estimates to determine how each condition or outcome affected the total cost of maternal morbidity. Then, we varied each impact estimate individually. S7 Appendix contains more information on the sensitivity analyses we conducted.

Results

In our literature review, we initially identified 8,337 studies through our database search and 95 papers through our review of the grey literature. After deduplication, the total number of unique records was 5,365. Using a systematic narrowing process (see S1 Appendix), we identified 32 studies that supported connections between nine maternal morbidity conditions and 24 maternal and child outcomes (Fig 3). To complete our data sources, we included 32 summaries or data briefs of publicly available data for the most recent prevalence and cost estimates and 44 studies from Luca et al. related to MMHCs [16]. Using the prevalence estimates for each of the nine conditions included in our model, we aggregated costs associated with each condition for all pregnancies or live births in 2019 to estimate a total cost of $32.3 billion from conception to five years postpartum. Of the total cost, we estimated $18.7 billion in medical costs and $13.6 billion in nonmedical costs. Two-thirds of these costs occurred within the first year postpartum.

Fig 3. Connections between nine maternal morbidity conditions and 24 outcomes, 2019 birth cohort.

Fig 3

This figure illustrates the connections included in our final model between nine maternal morbidity conditions and 24 outcomes. Each connection has a statistically significant evidence base or expert recommendation for inclusions and documented associated costs.

MMHCs ($18.1 billion), hypertensive disorders ($7.5 billion), gestational diabetes mellitus ($4.8 billion), and hemorrhage ($1.8 billion) generated the largest costs, in part because these are the most prevalent conditions among those with documented cost information. Child outcomes accounted for about 74% of the total costs ($24.0 billion), and maternal outcomes accounted for about 26% ($8.3 billion). The specific child outcomes driving the costs of these conditions included preterm birth ($13.7 billion), developmental disabilities ($6.5 billion), and respiratory distress syndrome ($2.1 billion). The maternal outcomes with the highest costs included productivity loss ($6.6 billion), cesarean section delivery ($895 million), and increased peripartum stay ($350 million). These nine maternal morbidity conditions and outcomes amounted to an average of $8,624 in additional costs to society for each maternal–child pair of the more than 3.7 million births in the United States annually. Table 1 summarizes our findings, and our full results appear in S8 Appendix.

Table 1. Cost estimates of maternal morbidity conditions in the United States for the 2019 birth cohort (in millions $).

Maternal morbidity condition Total Year 0 Year 1 Year 2 Year 3 Year 4 Year 5
Amniotic fluid embolism 4.4 4.4 0.0 0.0 0.0 0.0 0.0
Cardiac arrest 10.9 10.9 0.0 0.0 0.0 0.0 0.0
Gestational diabetes mellitus 4,843.9 4,049.2 158.9 166.1 173.6 181.5 189.7
Hemorrhage 1,828.9 1,828.9 0.0 0.0 0.0 0.0 0.0
Hypertensive disorders 7,540.8 6,231.4 261.8 273.7 286.1 299.1 312.6
Maternal mental health conditions 18,059.0 9,782.6 1,655.0 1,730.0 1,808.4 1,890.3 1,975.9
Renal disease 3.0 3.0 0.0 0.0 0.0 0.0 0.0
Sepsis 3.3 3.3 0.0 0.0 0.0 0.0 0.0
Venous thromboembolism 6.4 6.4 0.0 0.0 0.0 0.0 0.0
Total 32,300.6 21,920.1 2,075.7 2,169.8 2,268.1 2,370.9 2,478.2

This table shows the total estimated costs of nine maternal morbidity conditions from conception to five years postpartum, estimated for the U.S. 2019 birth cohort. Maternal morbidity conditions with acute outcomes have one-time costs, while conditions with chronic outcomes have ongoing costs, modeled through five years postpartum. S8 Appendix contains the full results of the model by condition and outcome.

More than half the costs (58%) were borne by the medical system, and the rest (42%) were borne by employers and other nonmedical sectors. Nonmedical costs included losses in productivity ($6.6 billion), additional social service provision for behavioral and developmental disorders in children ($6.5 billion), and increased family enrollment in and use of social programs, such as the Supplemental Nutrition Assistance Program; the Special Supplemental Nutrition Program for Women, Infants, and Children; Medicaid; and Temporary Assistance for Needy Families ($239 million).

The sensitivity analyses showed that our model estimated a range of $12.1 billion to $57.6 billion by varying all prevalence and impact estimates at once. Preterm birth associated with MMHCs proved the most sensitive to variation––the incidence of preterm birth could decrease the total costs of maternal morbidity by up to $6 billion or increase them by up to $5 billion relative to our main model results. S5 Appendix provides the range of parameters we tested, and S7 Appendix shows the results of the sensitivity analyses in a tornado diagram.

Discussion

Our findings demonstrate that maternal morbidity places a substantial economic toll on society. In particular, MMHCs, gestational diabetes, and hypertension have the highest costs, consistent with the results of Moran et al.’s systematic review of the incremental costs of maternal morbidity conditions [15]. In addition, our estimate far exceeds recent findings of Phibbs et al. and Chen et al., who projected an excess of $250 million and $630 million, respectively, in direct medical costs because of severe maternal morbidity [21, 22]. Our model projects substantially higher costs by including the costs of outcomes associated with morbidity conditions—rather than limiting costs to the medical conditions themselves—and expanding the scope of the analysis to maternal morbidity conditions more broadly. The costs could rise if we extended our model beyond five-years postpartum (the period we considered most salient to policymakers), although most costs accrued during the first year.

We found evidence to support inclusion of nine maternal morbidity conditions in the cost model. Among all 2019 births, we estimated these nine conditions cost society $32.3 billion—with a lower bound of $12.1 billion and upper bound of $57.6 billion—from the beginning of pregnancy to five years postpartum. These significant costs show a need for further investments in evidence-based maternal health initiatives, such as midwifery models of care that extend beyond birth, comprehensive gender-specific primary health care that provides seamless transitions in and out of pregnancy, and community-based models of maternity care [2326]. Initiatives should consider how social and structural factors beyond clinical care, such as unstable housing, lack of transportation, and racism, drive maternal outcomes [24, 27]. Policies that extend postpartum coverage or expand insurance coverage more broadly could incorporate these initiatives, and programming that incorporates a holistic approach to maternal health. It is important, however, to consider the implications of our findings in the context of the study limitations discussed here.

Limitations

Our cost estimates for the nine maternal morbidity conditions are limited by the completeness of costs documented in the literature and the accuracy of model parameters. In addition, unaccounted-for interactions between the modeled conditions might amplify or moderate the costs of individual conditions. We also lack data for maternal morbidity conditions beyond the nine we selected and for costs by subgroups.

Completeness of costs for nine selected conditions

The preponderance of medical costs during the delivery period implies our estimates might have missed costs associated with longer-term consequences of maternal morbidity, such as associated chronic conditions that develop later for birthing people and their children, as well as stroke, educational challenges, or future earnings for children. In addition, our model does not account for costs of readmissions because of maternal morbidity, which could substantially increase our estimate [28, 29].

Furthermore, the literature contained few nonmedical costs for conditions other than MMHCs. Studies of other health conditions found that nonmedical costs from lost earnings, productivity loss, and other indirect costs can account for more than half the overall costs [3032]. For our study, nonmedical costs accounted for 42% of total costs. These documented nonmedical costs mainly stem from MMHCs, the maternal morbidity condition for which we had the most complete outcomes and cost information. More information on nonmedical costs for the other maternal morbidity conditions could greatly increase our estimate of nonmedical and total costs. Finally, although we recognize that maternal morbidity can affect nonmaternal family members, such as sibling behavioral development, we only modeled the costs of the nine maternal morbidity conditions as they related to the birthing person and child.

Accuracy of model parameters

Our model uses data from secondary data and peer-reviewed literature that calculate statistical estimates to varying levels of statistical precision. In addition, the computer software used in this model introduced a slight rounding error. The precision could affect the accuracy of the societal cost and other statistical estimates reported.

Unaccounted-for interactions and secondary outcomes

To develop costs for each outcome, we modeled the likelihood of an outcome for each person with a given condition and the associated costs. Our model, however, cannot account for the effects of comorbidities. For example, a pregnant person with hypertension and gestational diabetes faces an increased risk for a preterm birth, and our model calculates only the independent costs of the individual conditions. These comorbidities could interact to increase or decrease the likelihood of a preterm birth beyond our impact estimate, resulting in an ambiguous effect on costs. In this example, the cost of delivery for a pregnant person with hypertension and gestational diabetes could end up substantially higher than the cost of delivery for a pregnant person with only one of these conditions. Despite the limitation that the model cannot account for comorbidities, the general lack of comprehensive data for other morbidities and outcomes suggests that maternal morbidity could have a much higher societal cost impact than we found.

Furthermore, to avoid overestimating the risk of outcomes associated with any given maternal morbidity condition, we chose not to model secondary outcomes. This approach might have led to our underestimating the costs of maternal morbidity. For example, we included child developmental disorders as one of the model outcomes associated with MMHCs. Many of these children might subsequently require use of social services, such as Supplemental Security Income, but we did not include this set of costs in the model because we already included the risk of social service use directly from MMHCs. Other researchers could revise the model to account for interaction effects of multiple conditions or secondary outcomes, which can provide a more comprehensive cost estimate for maternal morbidity overall.

Lack of information for all maternal morbidity conditions and long-term costs, and by subgroup

Our estimates likely do not capture the full costs of maternal morbidity for several reasons. First, although maternal morbidity includes many conditions and connections to outcomes, we could only find literature meeting our criteria to support modeling 32 connections between nine conditions and 24 outcomes (S1 Appendix). Second, we designed this model to focus on a six-year period (pregnancy to five years postpartum) so that stakeholders could understand the immediate impacts of maternal morbidity, especially as they consider allocating resources to interventions. We recognize that maternal morbidity can have long-term effects on the birthing person and the child, indicating that our estimates might represent only a fraction of the lifetime costs.

Ideally, we would have examined costs by various subgroups, such as race and ethnicity and Medicaid status, to understand the variation of cost burden for specific populations. For example, research has shown that Black birthing people have higher rates of severe maternal morbidity, which corresponds to a higher incidence of outcomes such as preterm birth and their associated costs [33]. Further, maternal mental health conditions get screened and diagnosed at a higher rate among non-Hispanic whites than Hispanic or Black birthing people, contributing to even greater disparities in measuring the true cost burden among minoritized populations.

The literature did not provide sufficient evidence of morbidity-outcome connections by racial or ethnic subgroup for us to include in our model. Additional data and analyses documenting incidence and prevalence of maternal morbidity conditions and costs by subgroup would deliver a more complete understanding of populations that bear a disproportionate burden and experience inequities in structural and social factors that lead to disparities in outcomes. Such information can better support decision making on medical and non-medical interventions that could have the greatest impact on maternal health–related outcomes and costs.

Conclusions

Today’s case of maternal morbidity can result in tomorrow’s maternal death, which means that better measurement and reporting of maternal morbidity and its associated costs is critical to addressing the maternal health crisis in the United States. To develop effective maternal programming and policies and assess their impacts on outcomes, researchers and program staff will need strong data on the prevalence and incidence of maternal morbidity and associated inequities, including information on whether any changes observed over time are due to reporting changes or true increases or decreases in prevalence or incidence. Our study demonstrates ongoing gaps in measurement of maternal morbidity, however, and lack of data to identify associated disparities. Heath care and public health agencies, measure stewards, and health systems can do more to define measures beyond those for severe maternal morbidity and incorporate an equity lens to better understand the differential impact on various subgroups of the population. These subgroups for measurement can include individual demographics and community characteristics to provide further insight into social and structural conditions driving maternal outcomes. These analyses will support a systems-wide response to reducing maternal morbidity and addressing the underlying societal and structural causes.

Supporting information

S1 Appendix. Description of literature review and search terms.

(DOCX)

S2 Appendix. Prevalence of maternal morbidity conditions and outcomes.

(DOCX)

S3 Appendix. Studies and data sources used to inform the cost estimates used in the model.

(DOCX)

S4 Appendix. Evidence of the association between maternal morbidity conditions and maternal and child health outcomes.

(DOCX)

S5 Appendix. Model inputs: Parameters and costs used to estimate the economic impact of maternal morbidity conditions among 2019 births.

(DOCX)

S6 Appendix. Modeling method.

(DOCX)

S7 Appendix. Sensitivity analyses.

(DOCX)

S8 Appendix. Full results of the model.

(DOCX)

S9 Appendix. Glossary.

(DOCX)

Acknowledgments

The authors would like to thank Jodie Katon and Kay Johnson for their expert guidance in refining our conceptual model. We would also like to thank Caroline Margiotta and Jessica Gao for their support conducting the literature review, and Erin Lipman for her support in developing the Excel model. Finally, we thank all the researchers and practitioners for conducting and publishing the studies used in our review.

Data Availability

All relevant data are within the paper and its Supporting information files.

Funding Statement

The Commonwealth Fund (https://www.commonwealthfund.org/) supported this work under contract number 20212979, received by SO. The funder had no role in study design, data collection, or in the analysis of results, and this article does not necessarily reflect the funder’s views or opinions. However, the funder provided a critical reading of the manuscript and offered suggestions for revisions.

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Decision Letter 0

Emily W Harville

18 May 2022

PONE-D-22-11649Societal cost of nine selected maternal morbidities in the United StatesPLOS ONE

Dear Dr. ONeil,

Thank you for submitting your manuscript to PLOS ONE. After careful consideration, we feel that it has merit but does not fully meet PLOS ONE’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process.

 The reviewers concurred on the importance of the project and generally approved the methods.  Their comments, particularly reviewer 2's comments, deserve thorough addressing.

Please submit your revised manuscript by Jul 02 2022 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

Please include the following items when submitting your revised manuscript:

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We look forward to receiving your revised manuscript.

Kind regards,

Emily W. Harville

Academic Editor

PLOS ONE

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

Reviewer #2: Yes

**********

2. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #2: Yes

**********

3. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #2: Yes

**********

4. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

**********

5. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: Thank you for the opportunity to review this critically important and timely study. O’Neil et al. address an important policy and public health issue – societal costs of maternal morbidity. The paper is well written and applies appropriate methods to advance the topic. I have several suggestions and requests for points of clarification that I believe will help to strengthen the paper. These concerns and minor feedback are detailed below, by section:

Introduction:

- Line 13 - I suggest replacing 'developed' with high-income or industrialized countries.

- Line 25 - Is 2019 in the "maternal-pair from pregnancy to five years postpartum" referring to the end of postpartum period? It is confusing to read about the 2019 birth cohort and tracking the maternal-child pair 5 years postpartum (meaning to 2023?) - please clarify.

Methods:

- Line 93 - Could you please include a reference/citation to the Healthcare Cost and Utilization Project from which the Nationwide Inpatient Sample was drawn for those readers not familiar with it?

- Lines 126-128 - I am not convinced about the causal language used in key parameters 3 and 4 - does this mean you only selected those studies that estimated the impact (causal relationship)? Or how do you know the incidence of outcome (3) was caused by the maternal morbidity? Also, odds ratio estimates the association between morbidity-outcome, and not the impact of morbidity on outcome. I advise the authors to either clarify the criteria or adjust the language used to avoid inferences about the causality between morbidity-outcome.

Results:

- Table 1 - please include a brief explanation as to why some maternal morbidity outcomes have cost estimates for 1 year and other for up to 5 years?

Discussion:

- Lines 223-224 - I think the language like this has to be attenuated unless you only included those studies that looked at the causal effect and thus provide evidence that those outcomes truly resulted from / were caused by maternal morbidity?

- Limitations - I appreciate the detailed discussion (and acknowledgment) of study limitations!

S4 Appendix. Effects of Exposure to Maternal Morbidity Conditions – Again, I would suggest adjusting the language implying causality between maternal morbidity conditions and maternal and child health outcomes (e.g. “Evidence of the association between MMC and maternal and child health” or “MCH outcomes associated with MMC” )

I am impressed with the authors’ rigorous and transparent approach to documenting the conduct of this study and providing so much supporting information in the appendices. This is very helpful and greatly appreciated.

Reviewer #2: This is a meaningful contribution to the body of literature about maternal morbidity. Akin to the referenced publication by Luca et all assigning a cost to the impact of MMHC, this manuscripts attempts to estimate the cost of several maternal morbidities. This analysis addresses the question of how to assign economic metrics to maternal morbidity beyond the associated hospitalization and recognizes that these experiences have ramifications for maternal and early childhood health.

As an obstetric reader of this, I need a better explanation of why this list of 9 morbidities. AFE, Cardiac arrest, AKI, sepsis, VTE are all severe maternal morbidities. GDM, hemorrhage, HTN, MMHC are not. Do the categories of outcomes and predisposing condition perhaps deserve separate analyses? There should at minimum be an acknowledgement of the mixed list. It is also notable that the list includes AFE (estimated 1/40,000 births) and hypertension (1/5 births).

In the small proportion of outcomes that increase maternal costs in this analysis I don't see any reference to level of care -- use of intensive care rather than typical maternity postpartum stay, yet this would be an outcome of AFE, cardiac arrest, possibly hemorrhage, possibly sepsis.

There is a vulnerability in this model -- for example, hypertension is linked to cardiac arrest and renal disease, but the authors do not "amplify" the cost of hypertension by linking the outcomes attributed to these. An explanation for these omissions would be reasonable.

The opportunity to build on this to create more complex models that would include amplifications should be mentioned. This manuscript offers an excellent precedent and warrants publication with its analysis as it stands.

While the authors mention in their second to last paragraph that it would have been "ideal" to examine costs by race and ethnicity, they do not disclose why they didn't. It is true, as they mention, that SMM is more common in Black birthing populations, as are their other morbidities of interest (hemorrhage, GDM, hypertension). The compounded impact of lost productivity, income, mental health toll etc on a population already structurally marginalized may tell a very different story. Especially as there is data that MMHC are identified in non-hispanic white people at a greater rate than in hispanic or NHB populations due to underscreening of the latter -- that is, this analysis is vulnerable to structural racism due to the morbidities included: there are inherently more white patients in the MMHC group with massive cost associated with its sequelae, and more Black patients in the hypertension group with is nearly 3x lower cost. Perhaps in the interest of building the literature around cost of maternal morbidities this discrepancy can be acknolwedged not unpacked, but the manuscript deserves a better explanation of why race wasn't investigated.

**********

6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

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Reviewer #1: No

Reviewer #2: No

[NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files.]

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PLoS One. 2022 Oct 26;17(10):e0275656. doi: 10.1371/journal.pone.0275656.r002

Author response to Decision Letter 0


17 Jun 2022

Please see the "Response to Reviewers" letter included in this submission for a table outlining how we addressed each reviewer's suggestions.

Attachment

Submitted filename: Response to Reviewers.docx

Decision Letter 1

Emily W Harville

14 Sep 2022

PONE-D-22-11649R1Societal cost of nine selected maternal morbidities in the United StatesPLOS ONE

Dear Dr. ONeil,

Thank you for submitting your manuscript to PLOS ONE. After careful consideration, we feel that it has merit but does not fully meet PLOS ONE’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process.

We thank you for your patience with the delays in reviews.  The reviewers agree the paper is substantially approved, but suggest a few minor clarifications. Please respond to these.

Please submit your revised manuscript by Oct 29 2022 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

Please include the following items when submitting your revised manuscript:

  • A rebuttal letter that responds to each point raised by the academic editor and reviewer(s). You should upload this letter as a separate file labeled 'Response to Reviewers'.

  • A marked-up copy of your manuscript that highlights changes made to the original version. You should upload this as a separate file labeled 'Revised Manuscript with Track Changes'.

  • An unmarked version of your revised paper without tracked changes. You should upload this as a separate file labeled 'Manuscript'.

If you would like to make changes to your financial disclosure, please include your updated statement in your cover letter. Guidelines for resubmitting your figure files are available below the reviewer comments at the end of this letter.

If applicable, we recommend that you deposit your laboratory protocols in protocols.io to enhance the reproducibility of your results. Protocols.io assigns your protocol its own identifier (DOI) so that it can be cited independently in the future. For instructions see: https://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols. Additionally, PLOS ONE offers an option for publishing peer-reviewed Lab Protocol articles, which describe protocols hosted on protocols.io. Read more information on sharing protocols at https://plos.org/protocols?utm_medium=editorial-email&utm_source=authorletters&utm_campaign=protocols.

We look forward to receiving your revised manuscript.

Kind regards,

Emily W. Harville

Academic Editor

PLOS ONE

Journal Requirements:

Please review your reference list to ensure that it is complete and correct. If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references. Any changes to the reference list should be mentioned in the rebuttal letter that accompanies your revised manuscript. If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice.

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #1: All comments have been addressed

Reviewer #3: All comments have been addressed

**********

2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

Reviewer #3: Yes

**********

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #3: Yes

**********

4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #3: Yes

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #3: Yes

**********

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: (No Response)

Reviewer #3: The authors have responded appropriately to the reviewers comments. A few points of clarification remain, and it would improve the paper if a little more clarity were added.

First, the study estimates costs over 5 years. Clearly the annual costs fall over this period, and it looks like most of the high costs consequences occur in this period. It would help if there were a clearer justification of the choice of 5 years, and a comment on the likely scale of underestimation of costs that result from this.

Second, i was a little unclear about the way in which health care inflation was used. There are two main reasons why costs in healthcare rise - general increases in pay and prices (which are not really relevant, since for decision making real rather than nominal costs are relevant), and increases in costs that are specific to the health sector, some of which are Baumol effects. The latter are relevant but are probably a small part of the increases.

Third, the co-morbidity point has been expanded, but i think we could still get a little more useful comment around this, particularly around the way in which costs for a single disease can be much higher in the context of co-morbidity.

Fourth, it is commented that the outcomes in question have risen substantially - is it clear if this is a real increase or a change in reporting?

Finally, and less seriously, it is a little odd to describe maternal deaths as one of the more serious outcomes. Some would argue it is the most serious!

**********

7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

Reviewer #3: No

**********

[NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files.]

While revising your submission, please upload your figure files to the Preflight Analysis and Conversion Engine (PACE) digital diagnostic tool, https://pacev2.apexcovantage.com/. PACE helps ensure that figures meet PLOS requirements. To use PACE, you must first register as a user. Registration is free. Then, login and navigate to the UPLOAD tab, where you will find detailed instructions on how to use the tool. If you encounter any issues or have any questions when using PACE, please email PLOS at figures@plos.org. Please note that Supporting Information files do not need this step.

Decision Letter 2

Emily W Harville

21 Sep 2022

Societal cost of nine selected maternal morbidities in the United States

PONE-D-22-11649R2

Dear Dr. ONeil,

We’re pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it meets all outstanding technical requirements.

Within one week, you’ll receive an e-mail detailing the required amendments. When these have been addressed, you’ll receive a formal acceptance letter and your manuscript will be scheduled for publication.

An invoice for payment will follow shortly after the formal acceptance. To ensure an efficient process, please log into Editorial Manager at http://www.editorialmanager.com/pone/, click the 'Update My Information' link at the top of the page, and double check that your user information is up-to-date. If you have any billing related questions, please contact our Author Billing department directly at authorbilling@plos.org.

If your institution or institutions have a press office, please notify them about your upcoming paper to help maximize its impact. If they’ll be preparing press materials, please inform our press team as soon as possible -- no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org.

Kind regards,

Emily W. Harville

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments:

Acceptance letter

Emily W Harville

28 Sep 2022

PONE-D-22-11649R2

Societal cost of nine selected maternal morbidities in the United States

Dear Dr. O’Neil:

I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department.

If your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org.

If we can help with anything else, please email us at plosone@plos.org.

Thank you for submitting your work to PLOS ONE and supporting open access.

Kind regards,

PLOS ONE Editorial Office Staff

on behalf of

Dr. Emily W. Harville

Academic Editor

PLOS ONE

Associated Data

    This section collects any data citations, data availability statements, or supplementary materials included in this article.

    Supplementary Materials

    S1 Appendix. Description of literature review and search terms.

    (DOCX)

    S2 Appendix. Prevalence of maternal morbidity conditions and outcomes.

    (DOCX)

    S3 Appendix. Studies and data sources used to inform the cost estimates used in the model.

    (DOCX)

    S4 Appendix. Evidence of the association between maternal morbidity conditions and maternal and child health outcomes.

    (DOCX)

    S5 Appendix. Model inputs: Parameters and costs used to estimate the economic impact of maternal morbidity conditions among 2019 births.

    (DOCX)

    S6 Appendix. Modeling method.

    (DOCX)

    S7 Appendix. Sensitivity analyses.

    (DOCX)

    S8 Appendix. Full results of the model.

    (DOCX)

    S9 Appendix. Glossary.

    (DOCX)

    Attachment

    Submitted filename: Response to Reviewers.docx

    Attachment

    Submitted filename: Response to Reviewers.docx

    Data Availability Statement

    All relevant data are within the paper and its Supporting information files.


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