literature review on maternal mortality

• Open access
• Published: 15 December 2022

A systematic review of individual and ecological determinants of maternal mortality in the world based on the income level of countries

• Maryam Tajvar 1 ,
• Alireza Hajizadeh 1 &
• Rostam Zalvand 1  

BMC Public Health volume  22 , Article number:  2354 ( 2022 ) Cite this article

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This systematic review was conducted to map the literature on all the existing evidence regarding individual and ecological determinants of maternal mortality in the world and to classify them based on the income level of countries. Such a systematic review had not been conducted before.

We conducted an electronic search for primary and review articles using “Maternal Mortality” and “Determinant” as keywords or MeSH terms in their Title or Abstract, indexed in Scopus, PubMed, and Google with no time or geographical limitation and also hand searching was performed for most relevant journals. STROBE and Glasgow university critical appraisal checklists were used for quality assessment of the included studies. Data of the determinants were extracted and classified into individual or ecological categories based on income level of the countries according to World Bank classification.

In this review, 109 original studies and 12 review articles from 33 countries or at global level met the inclusion criteria. Most studies were published after 2013. Most literature studied determinants of low and lower-middle-income countries. The most important individual determinants in low and lower-middle-income countries were location of birth, maternal education, any delays in health services seeking, prenatal care and skilled birth attendance. Household-related determinants in low-income countries included improved water source and sanitation system, region of residence, house condition, wealth of household, and husband education. Additionally, ecological determinants including human resources, access to medical equipment and facilities, total fertility rate, health financing system, country income, poverty rate, governance, education, employment, social protection, gender inequality, and human development index were found to be important contributors in maternal mortality. A few factors were more important in higher-income countries than lower-income countries including parity, IVF births, older mothers, and type of delivery.

A comprehensive list of factors associated with maternal death was gathered through this systematic review, most of which were related to lower-income countries. It seems that the income level of the countries makes a significant difference in determinants of maternal mortality in the world.

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Introduction

Maternal mortality is an important public health indicator, reflecting not only poor health condition of mothers and poor quality of health care services, but also in macro levels, it indicates poor economic, social, cultural, and political status in a community and also poor status of women in their society [ 1 , 2 , 3 ]. Nowadays, improving maternal health is the priority of most communities and organizations, including the World Health Organization (WHO) [ 4 ]. The fifth goal of the eight Millennium Development Goals (MDGs) focused on the reduction of maternal mortality by 75% from 1990 to 2015. Despite intense political attention of the countries toward the MDG5, most of them are still far from achieving their MDG5 targets [ 3 ]. The global estimates indicate that 295,000 (UI 279,000 to 340,000) maternal deaths occurred in 2017; 35% lower than 2000 when maternal deaths were estimated at 451,000 (UI 431,000 to 485,000) [ 5 ]. Thus, another target for maternal mortality reduction was set by the WHO to reach a global maternal mortality ratio (MMR) below 70 by 2030, which requires reducing global MMR by an average of 7.5% each year from 2015 to 2030. This means that to achieve this goal, maternal mortality must be reduced by more than three times the 2.3% annual rate of reduction observed globally between 1990 and 2015, which would be a difficult task for many countries [ 6 ].

Based on the existing evidence, there are significant variations in MMR of countries based on their development and income level [ 5 , 7 ]. Annually, half a million women die from pregnancy-related deaths, of which 99% happen in developing countries while most of them are preventable [ 8 ]. This variation is portrayed by WHO; while MMR has never been passed over 115 cases in Europe, America, and Western Pacific regions since 1990, it has always been over 500 cases in Africa [ 9 ].

In order to plan effectively and progress systematically in the reduction of MMR, having a broad knowledge about determinants of maternal mortality is an unavoidable need for policymakers and researchers. These determinants may distantly include socio-economic and cultural factors or intermediately include health status, reproductive status, access to health services, and health care behavior factors, or approximately include biological causes of death [ 10 ]. Causes of maternal death, based on the International Classification of Diseases-Maternal Mortality ( ICD-MM), are classified to direct (e.g., pregnancies with abortive outcome) or indirect (e.g., cardiac disease) causes in six groups by WHO [ 4 , 11 ], with no diversity among them.

The existing review studies on determinants of maternal mortality are generally old, or are non-systematic reviews or only reviewed selected determinants [ 6 , 10 , 12 ]. Recently, only a few studies reviewed the determinants systematically, such as the study of Yakubu et al. [ 13 ], but only focused on some micro determinants or other study on limited number of determinants [ 14 , 15 ]. Given that maternal death is a multidisciplinary phenomenon and different factors are involved, we tried to comprehensively review and overview all determinants, with no limitation, in high quality studies which would be of higher applicability for policymakers and researchers. The aim of this study was to conduct a systematic review on individual and ecological determinants of maternal mortality in the world and to classify them based on the income level of countries. In addition to summarizing distant and intermediate determinants of death in individual or ecological level, we also tried to answer this question that how these determinants might vary among mothers living in countries with various development level.

Inclusion criteria

All types of studies, except descriptive studies, that aimed to identify determinants of maternal mortality were included. In these studies the relationship of possible determinants with maternal mortality was statistically tested. Studies that only focused on the cause of maternal mortality as an approximate determinant, published in non-English journals, or when their full-texts were unavailable were excluded. We considered WHO definition for maternal mortality as “death of a woman while pregnant or beyond 42 weeks of gestation, irrespective of duration and site of pregnancy, due to any cause related to pregnancy or its management, but not due to accidental causes” [ 9 ]. No time limit was considered in searches.

Search strategy

To identify relevant studies, an electronic literature search was first conducted on “PubMed” and “Scopus” databases. Then, some of the most relevant journals were searched specifically (Table 1 ). Additionally, references of included articles were screened.

Except for PubMed database in which “Maternal Mortality” was searched using MeSH term, this term was searched just as a keyword in the rest of search procedure (Table 1 ). All articles retrieved from electronic databases and other studies were entered into EndNote X8 for further selection.

Study selection

The process of identification and selection of studies is shown in Fig.  1 . Initially, 902 and 901 studies were found through electronic searching in databases and journals, respectively. Furthermore, 1855 records were identified based on the bibliography of the included studies. Duplicate studies ( n  = 98) were removed. Then, screening of titles and abstracts was done by the researchers and studies not relevant were excluded ( n  = 3308). Then, the assessment of the full text of articles was done for eligibility of 252 studies. Based on the exclusion criteria, 123 studies were excluded due to being only descriptive and not reporting statistical results of relationship between independent variables and maternal mortality (except for review articles), or only focusing on biological causes of maternal deaths, or focusing on the relationship between determinants rather than their relationship with mortality. 16 studies were also excluded as their full-text were not found [ 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 ] and eight others were not in English [ 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 ]. In addition, 16 relevant articles were added through searching on Google. Finally, 121 relevant studies obtained from electronic databases, bibliographies, and hand searching were selected based on the inclusion criteria (Table 1 and Fig.  1 ). No study was excluded after quality assessment. Two reviewers (R.Z. and A.H.) performed the literature searching, checking the inclusion and exclusion criteria and selection of studies and finally data extraction, all phases independently. To reach consensus in the case of variations, in all stages of this review, the reviewers met frequently for discussion, and in the case of lasting disagreement, the third reviewer (M.T.), has been intervened.

PRISMA flow diagram for systematic review

Quality assessment

We used the STROBE checklist for original studies and also for ecological studies. It is designed for quality assessment of observational studies especially cohort, case–control, or cross-sectional studies based on 22 items [ 40 ]. There were some limitations in quality assessment of the included studies; i.e., some items of this checklist related to participants (item 6), or sample sizes (item 10), or reasons for non-participation (item 13) were not reported in ecological studies that used MMR as a dependent variable. Considering those items as “can’t tell”, the quality scores of those studies decreased. Other items such as clearly defining potential confounders in item 7, reporting potential sources of bias in item 9, way of handling quantitative variables in item 11, and reporting statistical methods used to control confounders or missing data in item 12 of the STROBE checklist were the main limitations of the selected studies, which were not often reported in their methodology. In some included studies, their limitations were not mentioned in the discussion. Each question in this checklist had one score, so the highest quality of the article would obtain score 22.

We used the critical appraisal checklist from Glasgow University [ 41 ] for the quality assessment of review articles. As shown in Table 3 , except for two studies containing nine review articles, the quality assessment was not performed for the rest of them, and their results were not reported precisely because determinants of maternal mortality were not based on the confidence interval. It is worth noting that the quality of the studies was independently assessed by two researchers in different places. Then, discrepancies were resolved by participation of a third researcher to reduce the risk of bias.

Data extraction

We extracted the required data from the included review articles and original articles using a purposefully designed data extraction form. Year of publication, number of maternal deaths, time span of the study, setting, design, and methods, and maternal mortality determinants were extracted from the included studies as shown in the Tables 2 and 3 .

Data analysis

The included studies were developed with diverse objectives, used various methods and different statistical techniques, and included participants with different characteristics and were widely distributed among countries. This diversity made formal meta-analysis almost impossible. Some of the included studies were developed with similar objectives, and participants had similar characteristics and used similar methods and measures, but we also classified the determinants of maternal mortality into three main individual, household, and community levels to be higher applicability for policymakers and researchers.

Classification of the included studies based on income levels of countries

We classified each original study based on low-, lower-middle-, upper-middle-, and high-income countries according to World Bank classification. Some points considered in this classification; for example, if a study in a country was in a specific income group for a some years and then its level changed for next years, we chose the income group that were for a longer period. In the study of Simo ˜es et al. [ 94 ], Brazil was in the upper-middle-income group for two years (2000–2001) and then changed as a lower-middle-income country in 2002; thus we considered Brazil in the upper-middle-income group. If a study included different countries in two different income groups, we chose the group with more countries. For example, in the study of Graham et al. [ 131 ], among 11 different countries, only two countries were in lower-middle group and the rest were in low-income group; so, we considered it as a low-income country. We also chose the last years of income level for the studies that used the same number of years for two different levels of income. For example, in the study of Taguchi et al. [ 77 ], Indonesia was lower-middle-income country for two years (1996 and 1997) and was low-income for the other two years (1998 and 1999), so we considered Indonesia as low-income country. Studies with more than two income groups in different countries were considered as global/regional studies, illustrated in the last column of Table 4 .

The results of this systematic review are structured here in two main sections; first, according to Tables 2 and 3 , which are a summary of all included original studies and review articles, respectively, and provide a description of the characteristics of included studies. Then, maternal mortality determinants are reported for review articles and original studies, summarized at individual, household, and community levels by income groups in Table 4 .

Description of the included studies

Of 121 included studies, 12 were review articles (Table 3 ) and the rest were original studies (Table 2 ) in which 33%, 28%, 27%, 10%, and 2% were ecological, case–control, cross-sectional, longitudinal (including prospective cohort, panel and time-series studies), and cohort studies, respectively. Among the 12 review articles, six of them with a total of 160 studies were conducted systematically and the rest were narrative reviews (Table 3 ). Most of the literature evaluated the determinants of maternal mortality globally with special focus on low- and lower-middle-income countries. Moreover, the results by countries show that most of the included original studies have been conducted in China, followed by Niagara and Indonesia (details provided in Fig.  2 ), and most of them were published after 2013 (Fig.  3 ).

Number of included studies by setting

Number of included studies by publication years during 1991–2021

Determinants of maternal mortality in the world

In this section, first the results of the review articles then the original study’s findings are reported based on determinant levels and income levels of countries.

Based on our review articles, some macro factors such as economic growth, poverty, inequality, improved water and sanitation, education, accessibility of health care services, etc. (details are shown in Table 3 ), and some micro factors such as age, parity, type of delivery, early marriage, etc. have led to maternal mortality.

Maternal mortality determinants based on individual, household, and community levels

Determinants at individual level mainly show the health status and reproductive-demographic characteristics of deceased mothers. A summary of the determinants of mothers who died due to pregnancy-related reasons, based on original studies, is described in Table 4 . As shown in column three of the table, among demographic factors, maternal age over 34 or below 18 is one of the main factors affecting pregnancy-related deaths, and nearly 70% of 36 examined studies with 7632 aggregate deaths confirmed this association. In addition, higher gravidity and cesarean delivery (as opposed to natural delivery) were recognized as significant threatening factors in 90% and nearly 75% of the studies, respectively. Furthermore, the results of this review show that mothers attended by skilled birth personnel, under prenatal care, as well as educated women are less likely to die from pregnancy-related causes. Moreover, delay in seeking health services and making decisions for delivery in all the eight observational evidences and factors related to mothers’ health (including the history of underlying disease, medical comorbidities, having complications during pregnancy or postpartum period, and hospitalization during pregnancy) were significant contributors according to most studies. However, in a few studies, some variables such as BMI, race, skin color, and caste were not recognized as associated factors. In contrast, each of the factors of birth weight, substance misuse, nationality, and previous pregnancy history were significant factors in one study, which we refused to include in Table 4 . At this level, there is no consensus on the effect of age at marriage, age at first birth, birth weight, and postnatal care coverage. Details about other determinants at this level were described in Table 4 .

Determinants at household level show that improved water sources and sanitation systems were the main contributors in most studies. In addition, low family income or wealth, and rural residence were associated factors in nearly 87% and 70% of studies, respectively. Also in this level, access to electricity in all the evaluated studies, and higher levels of husband education were recognized as significant factors in 80% of the observational studies (details in Table 4 ). Other factors mainly related to the condition of living, including the type of flour and toilet, building material of the house, and type of family were the associated factors, which are not shown in Table 4 .

At community or social levels, poor human resources in 73% of the examined studies, and poor accessibility or availability of health facilities (including the number of hospitals, primary health care, hospital beds, and equipment, etc.) in 79% of the 19 examined studies were the main contributors that threaten maternal health. Moreover, health expenditure shares (including higher share of out-of-pocket or private expenditures, and lower share of total health of GDP or public expenditures) and higher total fertility rate per woman were other associated factors in 89% and 77% of the included studies, respectively. In this category, there is no consensus on the effect of quality of services, and less use of contraception (details in Table 4 ). A few of the selected studies also recognized other factors such as life expectancy, need for emergency obstetric care, total calories consumed, and fat residuals as significant factors, which we refused to enter in Table 4 . In terms of economic factors that were mainly studied in ecological studies, lower-income countries levels (GNP or GDP) in 89% of the studies, unemployment rate in all studies, and poverty in 57% of the examined studies were significant threatening factors. However, income inequality and foreign investment are not known as significant factors. In contrast, debt rate, agricultural production indices, and private-sector infrastructure were other contributors in a study, which we refused to enter in Table 4 . In addition, results show that maternal mortality was also caused by social, political, and cultural factors. Thus, levels of education and governance (corruption index, instability, voice and accountability, etc.) were the main contributors in five ecological studies. Also, gender inequality and human development index both in four original studies were known as main factors in this category (details in Table 4 ). Also, other factors such as paved roads, annually published scientific papers, percentage of the indigenous population, and education efficiency were significant factors, which we refused to enter in Table 4 .

Maternal mortality determinants based on income levels of countries

A summary of our findings is shown in Table 4 . As it shows, the attributable risk of maternal death is varied based on income levels and development levels of countries; more than 80% of the examined studies and all four ecological studies found poor income levels of countries (GDP/GNP) and human development index strongly associated with maternal death, respectively.

Among the factors at individual levels, prenatal care, delay in decisions, type of delivery, and hospitalization during pregnancy in most studies (except the type of delivery in lower-middle income group) were significant factors in all different income levels. In six out of seven global studies, skilled birth attendance was a significant contributor in low-income, less developed or developing countries. Also, maternal education as another main factor, was an essential contributor in low and lower-middle-income countries, and more than 80% and 70% of the examined studies have shown a significant relationship, respectively. However, there is no consensus on this factor in upper-middle-income countries. Some factors like history of underlying disease, medical comorbidities, high-risk pregnancy, length of labor, and pregnancy stage were recognized as risk factors in low or lower-middle-income countries (Table 4 ).

Based on this review, most studies statistically accepted that less access to clean water and improved sanitation negatively affected maternal death in low and lower-middle-income countries, respectively. In addition, in poor countries, improvement in the availability or accessibility of health services and health expenditure shares were essential determinants in the reduction of maternal death. Based on our findings, reduction in the share of out-of-pocket and private health expenditures and increase in the share of total health expenditures as a percentage of GDP and public expenditures were most responsible for pregnancy-related deaths, particularly in low-income countries as well as other income groups. Moreover, the risk of pregnancy-related death was highly significant among the population below poverty line in all existing evidence, and the use of contraception in many studies in low-income countries. However, poverty in upper-middle-income countries was not significantly associated with maternal death. Moreover, gender inequality and gender empowerment measures (autonomy, economic empowerment, and attitudes towards violence) in low and lower-middle-income countries were associated contributors in most studies. However, gender empowerment measures did not affect maternal death in two ecological studies, and there is no consensus on the significance of gender inequality in our global studies. Details of the other variables is shown in Table 4 .

This study aimed to review and overview maternal mortality determinants in the world and categorize them based on income levels of countries. Our included studies show that most of these studies were conducted ecologically and were case–control studies and 58% of them were carried out in low and lower-middle-income countries.

According to the result of this study, maternal age, gravidity, type of delivery, education of mothers, pregnancy care, skilled birth attendance, and maternal health status are the main factors at individual level. At family level, factors such as access to improved water and sanitation, region of residence, family income or wealth, and other factors related to living conditions were significantly associated with maternal death. However, the systematic review conducted in Iran [ 156 ] failed to find the place of living as a risk factor because of having more access to primary health care among rural women in Iran. At community or social level, availability of health services, total fertility rate, health expenditures shares, income level, governance, inequality and education were main contributors.

Our review shows that women aged 18–34 years were less likely to die as compared with age groups of 35–49 or under 18 years. A possible reason for the high risk of maternal death among women aged 35–49 years is due to weakened uterus and anemia, and becoming pregnant is too risky for women older than 35 years and studies showed that the majority of women in this age group were not educated. Studies indicate that most maternal deaths occurred in women with no antenatal care and delay in seeking health services. This may be due to lack of awareness about the seriousness of maternal health. The type of delivery can also affect maternal mortality, as most cases of obstetric hemorrhage and emergency postpartum hysterectomy are associated with CS deliveries [ 75 ].

Among original studies, two observational studies in lower-middle income countries [ 93 , 144 ], and an observational study in the west of Iran [ 93 ], as upper middle-income country, demonstrate that skilled birth attendance has no significant relationship with maternal mortality. However, it doesn’t seem that skilled birth attendance has no effects on maternal mortality at this income level as those studies were conducted with a small sample size and limited time span. A longitudinal study in Chile, as a lower middle-income level country, showed this factor as a significant contributor in adjusted models. Some global or regional studies, mainly including less developed or developing countries, accepted this factor [ 76 , 130 , 138 ].

Based on our findings, households’ access to improved water and sanitation especially in low-income countries were negatively associated with maternal death. In line with this, Benova et al. [ 15 ] suggested that women in households with poor sanitation were 3.14 times more likely to die compared to women with better sanitation. The concept is that poor access to sanitation and water can provide the conditions for the prevalence of infectious diseases, which can directly affect maternal death. Studies also show that access to improved sanitation is associated with income level of countries [ 79 , 115 ]; a study shows that clean water and sanitation access improved by 14% due to economic growth in low and middle-income countries from 1990 to 2010 [ 73 ]. Based on WHO data [ 157 ], access to improved sanitation and water was always over 98% among high-income countries since 1990, while in low-income countries, access to clean water fluctuated around 46% to 66%, and access to sanitation fluctuated around 13% to 28% from 1990 to 2015.

Governance is one of the key factors that affects maternal mortality. Governance can be described as a set of traditions and conventions that determines the practice of authority in a particular country. It comprises not only the processes through which governments are selected, held accountable, monitored, and replaced, but also the capacity of governments to efficiently manage resources and formulate, implement, and enforce appropriate policies and regulations. In addition, governance regulates the level of respect received by the citizens and the state for conventions and laws that govern the economic and social interactions in the community. Through better governance, public spending can effectively and efficiently enhance health and education outcomes.

Some economic and health system-related factors such as the Gross National Income per capita and per capita government expenditure on health showed an inverse correlation with MMR. In contrast, private sector and out-of-pocket health expenditure showed a significant direct correlation with MMR: the more private sectors and out-of-pocket health expenditures in a country, the higher the MMR. Since appropriate government financing can ensure better access to some essential maternal health services, greater levels of health expenditure will be required for developing countries to achieve MDG on maternal mortality. Between 1995 and 2014, the average total health expenditure (%GDP) varied from 5% in low-middle income countries, 48% of which was paid by public expenditure, to nearly 11% in high-income countries, 61% of which was paid by public expenditure. Based on WHO statistics, the average out-of-pocket share (% total health expenditure) varied from nearly 41% in low-middle income countries to nearly 15% in high-income countries in the same period [ 158 ].

According to the literature, poverty positively affects maternal mortality as an economic factor, especially in the low-income countries. According to the evidence, poverty is linked to maternal mortality through malnutrition [ 99 ]. Malnutrition has been associated with anemia that is one of the main causes of maternal death [ 159 ]. Malnutrition may lead to chronic iron deficiency and anemia, which can make women prone to hemorrhage and infections [ 160 ]. Furthermore, women who experience malnutrition early in life are usually smaller, which increases the likelihood of obstructed labor [ 160 ].

Also, low human development index (HDI) in all evaluated studies was recognized as a significant contributing factor. HDI is one of the powerful indexes, which is a geometric means with three dimensions of life expectancy at birth, education, and GDP per capita. Much evidence shows that MMR reflects the general health status of a country and its development [ 130 ].

Moreover, education as a social factor is the other main contributor. As seen in Table 4 , among 20 studies that examined the effects of education on maternal mortality at community level, only one did not confirm this. This shows that education is influential in reducing maternal mortality. Educated women are more exposed to having an informed reproductive behavior, and they access reproductive health facilities frequently and timely. When women are educated, they increasingly improve their status, gain autonomy, awareness, responsibility, and control their fertility and reproductive activities such as in the use of contraceptives, prenatal and postnatal care, and health facility delivery [ 13 ]. Literature also shows that education in low and lower-middle-income countries is essential, which may have happened due to the low level of social and economic infrastructures in them.

Other factors including households’ income, employment, urbanization, inequalities, gender empowerment, human resource, availability of services were the other main factors particularly in less developed or developing countries (Table 4 ).

To the best of our knowledge, this is the first systematic review to present a robust summary of individual and ecological determinants of maternal mortality in the world based on income level of the countries. We used a sensitive search strategy in different sources, and finally identified 121 studies (109 original studies and 12 review articles). However, this review has several potential limitations. As the main limitation of this study, we only included studies in English with access to full-text documents. To compensate this, we did a rigorous search on different electronic databases, journals, and paper references, and the abstracts of excluded studies were reviewed to reduce bias. As with any systematic review, we may have missed some studies due to relatively a short list of search terms. Also, some studies did not provide access to the full text and thus they had to be excluded from this review. However, we minimized this by exhausting the search process through key terms, and the search strategy employed multiple academic and grey literature databases. As another limitation, all included studies had analytical observational and review design, so the cause-effect relation between determinants and maternal mortality could not be established. The results of this review can help researchers to understand the main determinants of maternal mortality based on income level of the countries and provide an appropriate space for research. This study can also provide comprehensive views for policymakers to reduce maternal mortality.

Conclusions

This review aims to comprehensively show the determinants of maternal mortality. The results of this study demonstrate the individual-level factors (e.g., age and parity), household-level factors such as region of residence, access to improved water etc., and community-level factors such as socio-economic, cultural, and health care system factors associated with maternal mortality. Based on extracted determinants, providing health services by making them affordable and available, access to quality health care, access to prenatal care, family planning care, and emergency obstetric care, increasing education levels, improving the condition of living and infrastructures, and the use of contraception can play an important role in the reduction of maternal mortality. Further studies are recommended to reach a consensus regarding certain determinants such as age at marriage, postnatal care coverage, etc.

Availability of data and materials

All data generated or analyzed during this study are included in this published article.

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MT, RZ conceived and planned the study. MT, RZ performed the data analysis. RZ, MT wrote the initial draft of the manuscript. MT and AH searched the literature. MT, RZ, and AH applied inclusion and exclusion criteria, extracted data, and appraised study quality. AH commented on this draft and critically revised the draft. All authors read and approved the final draft of the manuscript.

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Tajvar, M., Hajizadeh, A. & Zalvand, R. A systematic review of individual and ecological determinants of maternal mortality in the world based on the income level of countries. BMC Public Health 22 , 2354 (2022). https://doi.org/10.1186/s12889-022-14686-5

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WHO systematic review of maternal mortality and morbidity: methodological issues and challenges

• A Metin Gülmezoglu 1 ,
• Lale Say 1 ,
• Ana P Betrán 1 ,
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Metrics details

Reducing maternal mortality and morbidity are among the key international development goals. A prerequisite for monitoring the progress towards attainment of these goals is accurate assessment of the levels of mortality and morbidity. In order to contribute to mapping the global burden of reproductive ill-health, we are conducting a systematic review of incidence and prevalence of maternal mortality and morbidity.

We followed the standard methodology for systematic reviews. We prepared a protocol and a form for data extraction that identify key characteristics on study and reporting quality. An extensive search was conducted for the years 1997–2002 including electronic and hand searching.

We screened the titles and abstracts of about 65,000 citations identified through 11 electronic databases as well as various other sources. Four thousand six hundred and twenty-six full-text reports were critically appraised and 2443 are included in the review so far. Approximately one third of the studies were conducted in Asia and Africa. The reporting quality was generally low with definitions for conditions and the diagnostic methods often not reported.

Conclusions

There are unique challenges and issues regarding the search, critical appraisal and summarizing epidemiological data in this systematic review of prevalence/incidence studies. More methodological studies and discussion to advance the field will be useful. Considerable efforts including leadership, consensus building and resources are required to improve the standards of monitoring burden of disease.

Peer Review reports

Levels of maternal mortality and morbidity tell us about the risk attributable to pregnancy and childbirth as well as the performance of health systems in terms of access to health care and the quality of care provided. However, accurate assessment of these indicators has been problematic. The World Health Organization (WHO) has developed estimates of maternal mortality [ 1 , 2 ], anaemia during pregnancy, low birth weight and unsafe abortion at national, regional and global levels using modelling techniques. The lack of good quality data for many countries and different methodologies used to estimate levels of mortality complicate monitoring of the trends and comparisons between countries [ 3 , 4 ].

Although considerable amounts of facility-based data on maternal morbidity are generated, these may not reflect the actual health status of women in the whole community or area. Population-based data on the status of women's health are more useful and needed, yet scarce. Even when available, the challenge remains as to how to compile and summarize the data and thus map the burden of reproductive ill-health. A logical approach is to extend the concept of systematic reviews from randomised controlled trials to observational data [ 5 – 7 ].

For more than a decade, systematic reviews of randomised controlled trials (RCTs) have been used increasingly to evaluate the effectiveness of various health care interventions. The Cochrane Library as of 2004 includes more than 3000 systematic reviews [ 8 ]. Considerable experience of methodological issues such as literature search, critical appraisal of identified studies and methods for summarising data has been gained and tools have been developed for the reviews and meta-analysis of RCTs [ 8 ].

However, systematic reviews of observational studies are rather rare and the relevant experience is limited [ 6 ]. Most of the work in this area relates to questions for which RCTs are difficult, impossible or unethical to conduct (e.g. testing aetiological hypothesis, less common adverse effects in drugs) [ 9 ]. Methodological issues with regard to inclusion of studies with different designs, population and setting characteristics, and statistical methods to combine the data are evolving and need to be improved [ 6 , 10 ].

With these considerations, we are conducting a systematic review of prevalence/incidence of maternal mortality and morbidities from 1997 to 2002. The primary objective is to contribute to mapping the global burden of reproductive ill-health. The review will provide a comprehensive, standardized and reliable tabulation of available data on the incidence/prevalence of maternal morbidity and mortality, and case-fatality rates for maternal morbid conditions. The review will also assist us in identifying the most commonly used set of definitions for some pregnancy related conditions, in testing a set of critical appraisal and data-extraction instruments that can be used in future reviews of observational studies, and in guiding future research in this field.

We prepared a protocol [ 11 ] and a form for data extraction (See Additional file 1 ) which were both peer-reviewed. In this manuscript, we present our experience with the methodological, technical and practical challenges encountered in conducting the review.

Criteria for considering studies

Types of studies.

For maternal morbidity, any study type providing prevalence, incidence or case-fatality rates for specified maternal morbid conditions is considered. These include mainly cross-sectional and cohort studies, clinical trials, and incidence/prevalence surveys. Case-control studies are included if the cases selected correspond to all cases in a given population where the denominator is also known. Intervention and control arms of controlled trials are treated separately.

For maternal mortality, studies providing estimates of maternal mortality levels derived from direct counting, or from special surveys are considered for inclusion. Estimates derived from modelling of other variables or extrapolations from other populations are excluded.

Types of participants

Women either pregnant or within one year of termination of pregnancy.

Types of outcomes

Maternal mortality and morbid conditions defined according to the International Classification of Diseases, 10 th revision (ICD-10) [ 12 ].

Studies are ineligible if any of the following apply: (i) data collection dates are not reported, (ii) data are collected only before 1990, (iii) part of the data is collected before 1980 and disaggregation by year is not possible (in order to exclude data before 1990), (iv) number of study participants is less than 200 (this criterion imposed arbitrarily), (v) the study design is case-control and incidence/prevalence estimates from the defined population cannot be calculated, (vi) the methodology is not described.

Search strategy for identification of studies

We searched for published and unpublished studies reported between 1997 and 2002 in any language. The decision to start from 1997 was arbitrary and based on the concept of reviewing recent data.

The sources searched to identify studies included electronic databases (Medline, Popline, EMBASE, LILACS, CAB Abstracts, SocioFile, CINAHL, Econlit, BIOSIS, PAIS International, Index Medicus for the Eastern Mediterranean Region (EMRO) – on-line database of WHO/EMRO); web pages from Ministries of Health for official information and other potentially relevant internet sources (e.g. reproductive health gateway, development gateway, dissertation abstracts, Google). Additional file 2 includes detailed strategy for electronic search.

In addition, we checked reference lists of retrieved articles, proceedings and abstract books of related congresses. We hand searched journals at WHO headquarters' library that are not indexed in electronic databases and countries' statistical reports held at the WHO library. We contacted country focal experts such as WHO representatives and staff from collaborating centres, non-governmental organizations (NGOs), and other organizations known to be active in the field.

A WHO specialised librarian and the trial search coordinator of the Cochrane Collaboration Pregnancy and Childbirth Group developed the search strategy for each of the electronic databases according to their specific subject headings or searching structure in collaboration with the reviewers. We tested the search strategy for citations from 1997, modified the strategy and ran it for the whole period. We used Reference Manager ® software [ 13 ] to keep track of the citations identified. We downloaded the citations identified in electronic searches into Reference Manager ® and entered those retrieved from other sources manually (e.g. hand searching, reference lists). We deleted duplicates and assigned a unique identification number for each citation.

Screening and data-extraction form

Initially, we evaluated all identified citations on the basis of titles and/or abstracts against the eligibility criteria. Those deemed to be irrelevant were excluded and reasons for exclusion noted. A list of excluded reports and the reasons for exclusion are available from the authors upon request. When the information provided by titles/abstracts was insufficient to decide on inclusion/exclusion, or the titles/abstracts were relevant to the project, we retrieved and evaluated the full-text. As of January 2004, we screened titles/abstracts of a total of 64,586 citations from years 1997 to 2002. Among these, 59,960 were excluded and we retrieved full-text reports of the remaining 4626 (Figure 1 ).

Flow diagram of the process of identifying and including references for the systematic review.

We completed a specially designed screening form for each full-text evaluated report. This form was used to collect information on whether the report was included or not and if excluded, the reason for this. For reports meeting more than one exclusion criterion, only one reason – following the order on the screening form – was reported as the reason for exclusion. We extracted data from the included studies using a specifically designed data extraction form (See Additional file 1 ). This form includes 48 questions distributed in five modules. Modules were designed to collect information on (i) the general characteristics of the study such as design, population, setting, (ii) prevalence/incidence of maternal morbid conditions, (iii) maternal mortality, (iv) quality assessment of morbidity reports and (v) quality assessment of studies reporting maternal mortality. We also developed a manual for providing definitions and instructions on how to extract the data (available upon request from the authors). We tested both screening and data-extraction forms for a group of studies of different designs and revised prior to use.

Agreement between the reviewers in screening and data extraction

Two reviewers independently screened titles/abstracts from a sample of citations identified through the electronic search. In order to estimate the level of disagreement between two reviewers when including studies in the systematic review within 2.5% of the true value, a total of 560 studies needed to be classified. This sample size assumes a 95% confidence interval and that the level of disagreement between the two reviewers will not exceed 10% [ 14 ]. The percentage of agreement was 88.9% (95% CI 86.0% to 91.4%). The inter-observer agreement beyond chance was calculated using the Kappa statistics and found to be 0.60 (95% CI 0.52 to 0.69). This value corresponds to moderate to substantial agreement between the reviewers [ 15 ].

Two reviewers independently assessed full-texts of 50 articles for inclusion in the review following the initial screening process and completed the data extraction forms for those that were eligible for inclusion. The reviewers then compared results and disagreements were resolved following discussion. The decision on the number of this sample was agreed on arbitrarily at the beginning and deemed to be sufficient following the discussions on the completed forms.

Data processing

We categorised variables of interest and developed codes for responses to open-ended questions to facilitate data entry and statistical analysis. Initially, we classified morbidities according to the ICD-10 [ 12 ], using the classifications described mainly in chapter XV, which addresses pregnancy, childbirth and postpartum conditions. We assigned unique codes to some conditions (e.g. obstructed labour) that are classified with different codes in ICD-10 according to etiological distinctions. (See Additional file 3 ). These changes were made to facilitate the coding of the conditions during the data extraction process since definitions do not generally include etiological distinctions in incidence/prevalence studies of maternal conditions.

We preferred to extract raw data but where only percentages or rates were available, we also included these. Data presented in graphs and figures were used only if numbers (or percentages) were described in the text or labelled in the graph. Such data were not used if extrapolation was required. Once data extraction was completed, data were reviewed to identify duplicate data, for example the same results published in more than one journal or published papers whose unpublished drafts had been identified previously. Data were manually double entered in a specific database and processed with SAS software ® .

Appraising methodological quality of primary studies

We excluded studies that did not state the methodology used to obtain data. For quality appraisal, we extracted information on (i) study design, sampling method, sources of data and completeness of follow-up or records and, (ii) reported definitions and diagnostic procedures regarding outcome measures.

The evaluation of methodological and reporting quality was used to assess the reliability and accuracy of the data as objectively as possible. For example, the selection criteria and certain characteristics of participants such as economic status, ethnicity, age group or health status allow assessment of the external validity or generalisability of results in addition to presentation of stratified analysis for different categories. Likewise, information on the proportions and characteristics of losses to follow-up, non-responders or those not included in the final analysis after having been initially selected for the study was used to assess the internal validity of a study.

Furthermore, we assumed that the presence of definitions of conditions and description of diagnostic methods or procedures could be regarded as an indication of higher quality. For studies which reported maternal mortality, in addition to categorising definitions of maternal mortality, we recorded information about special efforts to capture all maternal deaths and the method to confirm deaths as maternal (e.g. confidential enquiry, verbal autopsy) as indication of higher quality.

We identified and screened about 65,000 different citations for the period 1997–2002. As of January 2004, 4626 citations were identified as potentially eligible for full-text evaluation, 2443 of which were included and 1988 excluded. The remaining 195 are in the process of retrieval and evaluation (Figure 1 ). Citations were mostly excluded because of no relevant data (57%), sample size less than 200 (16%) and no dates of data collection period (11%). The number of included studies for which data extraction and entry is complete is 2204. The distribution of these studies according to their designs, selected characteristics of the population and settings are summarised in Table 1 . Most studies use a cross sectional design (69.5%). The study population is urban in 17% of the studies, rural in 6.7%, mixed in 43.6% and unknown in 32.7%. Nearly two-thirds of the data is facility based while most of the rest is either nationally or regionally representative.

Almost half of the studies are from Europe and North America and one-third are from Asia and Africa (Table 2 ). Similarly, half of the studies are conducted in 43 industrialised countries while the remainder are from 95 less developed and 46 least developed countries (Table 3 ).

The data were disaggregated by study periods, age groups, ethnic groups, settings and interventions used (i.e. different arms of RCTs) where possible and entered in the database as separate data sets. From 2204 included studies, we obtained 3805 data sets most of which include prevalence/incidence data on more than one morbidity. Morbidities of interest in our review were reported 5933 times in these data sets. Table 4 presents the distribution of reported morbidities and shows that hypertensive disorders of pregnancy and stillbirth were most frequently reported (14.9 % and 13.9%, respectively).

A preliminary assessment of the reporting quality of studies shows that the quality is generally low. For example, for hypertensive disorders, about 50% included definitions and only 10% described the diagnostic procedure. More than half of the studies of maternal mortality did not report the definition used for maternal death, and two-thirds did not use any method to confirm the death as maternal. Similarly, less than 20% attempted to capture all maternal deaths among the population studied.

In this paper, we present our initial experience with conducting this large-scale systematic review of observational studies. We discuss methodological challenges as well as barriers encountered at both technical and logistic levels. We present preliminary results on the descriptive characteristics of the data set and expect to generate more discussion and empirical research in this area.

One of the main strengths of this systematic review is the comprehensive search strategy including multiple electronic databases. This search strategy yielded a large number of disparate records. This is partly due to the fact that searching according to study type is possible only for controlled trials. Initial screening by titles and abstracts to select relevant studies reduced the number of potentially relevant reports to a reasonably manageable level. However, it was not always straightforward to judge relevance from abstracts and this has been a tedious and time-consuming process.

Identification of duplicate records has been another time-consuming activity. Different databases use different formats for indexing titles and/or authors. For example, authors of articles written in Spanish tend to present two surnames. The first surname may not be recognised correctly or surname and first name are not always differentiated [ 16 , 17 ]. This lack of uniformity in formatting of citations across databases causes several different recordings of the same article if indexed in more than one database. We experienced an exhaustive process of manual searching and deleting of duplicate records to address this issue.

Although efforts to develop methodologies for searching for and summarising data from observational studies exist, these focus largely on effects of health care interventions that are difficult or impossible to evaluate through RCTs [ 18 ]. The associations between risk factors and disease occurrence, evaluation of diagnostic and screening tests or prognostic variables are other areas that require reviews of observational studies and pose particular methodological challenges [ 9 ].

The specific issues relating to analysis of data from systematic reviews of incidence/prevalence studies need to be systematically explored in order to guide developments in this field. Evaluating the comparability in terms of design, population and setting, and summarizing results pose specific methodological challenges.

A great deal of variation in the incidence/prevalence of maternal conditions between studies has been shown to be related to variations in definitions [ 19 – 21 ]. In addition, for many conditions studied, we identified that a variety of diagnostic tests with different levels of precision were used. We extracted detailed information on such characteristics of the studies in order to explore the contribution of these factors to the heterogeneity of the results. We envisage providing a tabulation of the most commonly used definitions and diagnostic procedures for each condition. In a second step, we will examine why the 'most recommended' or 'official' definition or diagnostic procedure is or is not used and how this affects the outcomes. This could provide a background for initiatives to standardize the definitions and improve the accuracy of measurements.

Another issue of concern is the general poor reporting quality of the studies. Characteristics of the populations and/or settings, definitions of outcomes and diagnostic procedures are not systematically described in the reports. This could limit the comparability of the studies and utility of some data extracted.

An important challenge for systematic reviews on burden of disease is to identify all available data from less developed countries [ 22 ]. Studies from these countries are likely to be published in non-indexed and non-English journals. The amount of research conducted in those countries may also be less considering the difficulties of securing funds for research. Nevertheless, we think that the final data set will include substantial amount of data from developing countries.

The first decade of this new millennium will be a test of our capability and ability to cope with the ever-increasing amounts of information produced. To analyse all available information in a reliable way, we need systematic reviews that include comprehensive searches, critical evaluation of studies and advances in statistical and other (searching, appraisal) methodologies. Considering the fact that it took almost 20 years to achieve satisfactory standards of synthesising the research into the effects of health care interventions, it is clear from our initial work that similar efforts, including leadership, consensus building and resources are required to improve the standards of monitoring the burden of disease.

The conceptualisation of this review, completion of a protocol and actual conduct has taken three years with several staff members involved. Full results will be available at the end of 2004. Although complex and time-consuming this systematic review and others on this issue will contribute to the Millennium Project [ 23 ] in several ways. Firstly, quantifying the burden not only through modelling approaches but also through in depth analyses of empirical studies will improve our understanding of the magnitude of the problem. Secondly, by identifying the gaps in the methodology and reporting, future research could be designed more rigorously.

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Acknowledgements

We thank Tomas Allen and Lynn Hampson for improving the search strategy, Felipe Santana, Genc Kabili and Veronica Lippuner for data extraction, Alexandre Peregoudov for data management, Evelyn Jiguet for coding, Centro Rosarino de Estudios Perinatales (CREP), Argentina for data entry, Agustín Conde-Agudelo for input on the data extraction forms and Dr Paul Van Look for his comments. We also thank Jesse Berlin, Allan Donner and Ken Schulz for their advice on methodology at the protocol development stage.

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UNDP/UNFPA/WHO/World Bank Special Programme of Research, Development and Research Training in Human Reproduction, Department of Reproductive Health and Research, World Health Organization, Geneva, (1211), Switzerland

A Metin Gülmezoglu, Lale Say, Ana P Betrán, Jose Villar & Gilda Piaggio

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Correspondence to A Metin Gülmezoglu .

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Competing interest.

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Authors' contributions

AMG and JV initiated the project. AMG, LS, APB and JV co-ordinated the review. APB, LS, AMG and JV prepared the manuscript. LS, APB and AMG developed the search strategy. LS and APB performed the title/abstract screening of the records. LS and APB, extracted data. GP provided statistical expertise.

Electronic supplementary material

Additional file 1: data-extraction forms used in the systematic review. (pdf 72 kb), additional file 2: final search strategy for each of the searched electronic databases. (doc 46 kb), 12874_2004_74_moesm3_esm.doc.

Additional File 3: ICD-10 codes and those used in the systematic review for maternal mortality and morbidity conditions. (DOC 46 KB)

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Gülmezoglu, A.M., Say, L., Betrán, A.P. et al. WHO systematic review of maternal mortality and morbidity: methodological issues and challenges. BMC Med Res Methodol 4 , 16 (2004). https://doi.org/10.1186/1471-2288-4-16

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Article Contents

Introduction, author biographies, supplementary material, conflict of interest statement.

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Measuring maternal mortality: a systematic review of methods used to obtain estimates of the maternal mortality ratio (MMR) in low- and middle-income countries

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Florence Mgawadere, Terry Kana, Nynke van den Broek, Measuring maternal mortality: a systematic review of methods used to obtain estimates of the maternal mortality ratio (MMR) in low- and middle-income countries, British Medical Bulletin , Volume 121, Issue 1, January 2017, Pages 121–134, https://doi.org/10.1093/bmb/ldw056

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The new global target for maternal mortality ratio (MMR) is a ratio below 70 maternal deaths per 100 000 live births by 2030. We undertook a systematic review of methods used to measure MMR in low- and middle-income countries.

Systematic review of the literature; 59 studies included.

Civil registration (5 studies), census (5) and surveys (16), Reproductive Age Mortality Studies (RAMOS) (4) and the sisterhood methods (11) have been used to measure MMR in a variety of settings.

Middle-income countries have used civil registration data for estimating MMR but it has been a challenge to obtain reliable data from low-income countries with many only using health facility data (18 studies).

Based on the strengths and feasibility of application, RAMOS may provide reliable and contemporaneous estimates of MMR while civil registration systems are being introduced. It will be important to build capacity for this and ensure implementation research to understand what works where and how.

Reducing maternal mortality is one of the priority goals on the international agenda—the new global target is to reduce the maternal mortality ratio (MMR) to <70 maternal deaths per 100 000 live births and country should reduce their MMR by at least two-thirds from the 2010 baseline and no country should have an MMR higher than 140 deaths per 100 000 live births by 2030. 1 , 2 A cross-cutting priority for the post-2015 agenda is to move toward counting every birth, maternal death and perinatal death through the establishment of effective national registration and vital statistics systems in every country, as stated within the recommendations of the Commission for Information and Accountability. 3

Reliable data are needed so that adequate resources can be allocated to maternal health programmes for countries (or regions in countries) that are not yet accelerating the annual reduction in maternal deaths. These data are also needed to monitor progress toward the targets set for the new Sustainable Development Goals (SDG). Assessing progress has been a challenge because <40% of countries currently have complete civil registration (CR) systems in place or other methods to provide accurate and contemporaneous MMR data. Similarly, although Maternal Death Surveillance and Review is promoted and being implemented in many settings, attribution and reporting of cause of maternal death is not yet systematically in place. 4 , 5 Only 2 of the 49 least developed countries have >50% coverage with regard to death registration. 1

The World Health Organization (WHO) publishes global estimates of MMR based on United Nations statistical models, including estimates for countries without reliable data. 6 Most of these estimates are subject to greater or lesser degrees of uncertainty and this is a recognized limitation. There are, in addition to modelling, a variety of methods available to measure MMR including via censuses, household surveys, Reproductive Age Mortality Studies (RAMOS) and using the Sisterhood methods. Each method has strengths and weaknesses. This may include cost of application of method, lack of in-country capacity to use the method and requirement for large sample sizes to be able to estimate MMR with reasonable accuracy. Although some of these methods have been used in a number of developing countries, there is a lack of knowledge and guidance regarding which method(s) are the be most appropriate and feasible to use in which settings (e.g. large or small population, national or sub-national application and type of data required to estimate MMR).

We, therefore, conducted a systematic review of the literature to identify which methods have been used to estimate MMR and reviewed their use and applicability to low- and middle-income countries. The specific research questions included: what are the type of data and data sources required, what are the strengths and weaknesses of each method; and which method(s) would be useful and applicable in low- and middle-income settings and able to provide reasonably accurate and contemporaneous data.

We used the following databases SCOPUS, PUBMED and Institute for Scientific Information (ISI) and MEDLINE to search for studies that measured MMR in low- and middle-income countries. Publications of organizations and programmes such as the United Nations Population Fund (UNFPA), United Nations Children's Fund (UNICEF), World Bank, WHO and the Initiative for Maternal Mortality Programme Assessment (IMMPACT) were included. Internet searches using the Google search engine were conducted to identify relevant literature not published in peer-reviewed journals and the references of all identified, relevant papers were hand-searched.

Search terms used

Medical Subject Headings (MeSH) were searched to identify all relevant terms used to describe maternal mortality and measuring. Boolean operators such as ‘OR’ were used to join keywords and MeSH terms defining the same concepts and different concepts were searched with ‘AND’ to arrive at the final result. We used the search terms ‘maternal mortality OR maternal death OR pregnancy death OR motherhood death OR women deaths’ in combination with ‘measure OR estimate’ OR ‘estimation’. These were then combined using the Boolean operator ‘AND’ with the following search terms: civil registration data, Census, Surveys, health facility data, sisterhood methods, RAMOS and low- and middle-income countries. Star truncation (*) was used where multiple endings of terms were possible.

Inclusion and exclusion criteria

We included papers published between 2000 and October 2016 from low- and middle-income countries as defined by the World Bank income categorization. 7 This time period was selected as many countries undertook to assess the MMR to evaluate the burden of maternal mortality and effect of implementation of interventions to achieve MDG5 at this time. 8

Studies were included for estimates of MMR obtained at either national or sub-national level regardless of method used. We excluded studies assessing impact of one or more interventions on MMR; demographic health surveys (DHS) as they are included in the direct sisterhood methods and global estimates by WHO, UNICEF, UNFPA and the World Bank (published by the WHO and not countries that use statistical models that may have errors, use unreliable data sources and, in some cases, countries do not use them). We also excluded reviews, posters, editorials and discussion papers, which did not include methodologies and estimates of MMR. DHS were excluded as these use the direct sisterhood method which is already included in the review. Global estimates were excluded when they employed statistical modelling.

Data extraction

Two reviewers independently screened all titles and abstracts. When the information provided by title and abstract was insufficient to decide on inclusion or exclusion, full-text versions were retrieved and evaluated. All included papers were reviewed in full. Any discrepancies were resolved through discussion with a third researcher. A summary table was developed and agreed by all authors before full-text review was conducted and all included studies were then summarized. ( Supplementary Table S1 : Summary Table of included studies)

PRISMA diagram for identification of included studies.

CR and vital statistics data

CR is defined as the continuous, permanent, compulsory and universal recording of the occurrence and characteristics of vital events pertaining to the population as provided through decree or regulation in accordance with the legal requirements of a country. 9 The data retrieved from CR systems are referred to as vital registration (VR) data. Complete coverage, accuracy and timeliness of CR are essential for quality vital statistics and are the ideal data to count maternal deaths. CR is carried out primarily for the purpose of establishing the legal documents provided by the law. Additionally, registration of births and deaths generates information that has substantial policy utility, especially when the age of the mother giving birth, age and sex of the decedent and underlying cause of death are correctly specified. 10 Ideally, CR systems with high coverage and good attribution of cause of death provide accurate data on the level of MM and the causes of maternal deaths. The drawback, however, relates primarily to the availability, reliability, completeness and coverage of the CR data. 11 The number of maternal deaths and number of live births recorded are used to calculate MMR (number of MD per 100 000 live births).

Five papers reported using CR data to estimate maternal mortality. These studies were conducted in China, the Dominican Republic, Brazil, Egypt and Guatemala, all middle-income countries. 12 – 16 There were no studies from low-income countries using this method. 12 – 16 In Guatemala, VR data were supplemented with additional information from medical charts and from public healthcare centres which improved the quality of data obtained. The study detected three times the number of maternal deaths compared to using the civil registry data only. 15 However, to establish whether the death of a woman of reproductive age (WRA) is a maternal death, information on pregnancy status at time of death is required. It was noted that information on pregnancy status was often either missing or unclear on the death certificates for studies conducted in China, the Dominican Republic, Brazil and Egypt. The authors note that this resulted in misclassification and possibly an underestimation of the number of maternal deaths. 12 – 14 , 16 Deaths among women living in villages accessible only by foot were not registered in the study in the Dominican Republic. 14

Health facility surveys

Health facility data remain the main, routine source of data on MM for many developing countries. Currently, health facility data are not used by academics and by agencies for compiling global mortality estimates, but they are widely used in many countries as they are locally generated and continuously available. Data sources include routinely reported records in health facilities or sentinel sites, reports from healthcare providers and health facility surveys.

Eighteen papers reviewed used health facility data to estimate MMR. 17 – 34 Most studies were conducted in low- and middle-income African countries such as Nigeria, Cameroon, Malawi and Zambia. However, middle-income countries such as India, Pakistan and Turkey also estimated MMR using health facility data. 18 , 23 , 27 It was noted that 15 studies were conducted in tertiary or teaching hospitals, which are expected to have a significant proportion of high-risk obstetric cases although this proportion was not reported. 17 , 19 – 23 , 27 – 29 , 31 – 34 Maternal deaths were identified from maternity ward records in 16 out of 18 facilities. Only two studies identified cases from other wards including the female or gynaecology ward and from operating theatre registers. 21 , 31 Case notes for women who had died were noted to have been missing in some facilities and there were considerable inaccuracies in routine registers noted in most retrospective studies. 20 , 25

Population census

Was the death due to an accident, violence, homicide or suicide?

If the deceased was a woman aged 15–49, did the death occur while she was pregnant, during childbirth or during the six weeks after the end of pregnancy?

As a result, in the 1990s several countries included questions intended to ascertain if any WRA had died during pregnancy or within a defined period postpartum, usually 6 weeks. In principle, a census allows the identification of deaths in a household in a relatively short reference period (1–2 years) and thereby provides estimates of recent maternal mortality.

Population census data were used to estimate maternal mortality in five studies. 37 – 41 Questions regarding the time of circumstance of death among WRA who died during pregnancy, labour and in the postpartum period (usually 6 weeks after delivery) were included during census data collection. The studies were conducted in Latin America (Honduras, Nicaragua and Paraguay), South Africa, Burkina Faso, Honduras (only) and Indonesia. A study conducted in Burkina Faso used this approach and obtained an estimate of the MMR, the results of which were similar to a previous study which had used the direct sisterhood method. 41 In Latin America, there was a greater number of reported pregnancy-related deaths in census data when compared with the number reported during a household survey which was conducted at the same time. 39 Similarly, in the Republic of South Africa, an increase in maternal death was observed. 40 Narrow confidence intervals were obtained: MMR: 519 per 100 000 (95% CI: 454, 584) in Damage and MMR: 353 per 100 000 (95% CI: 295, 411) in Orangey in Burkina Faso. 41 Similarly, there was a greater number of reported pregnancy-related deaths using census data than obtained via sample surveys in Latin America.

Population or household surveys

Population or household surveys are one of the most important data capturing methods for maternal deaths in settings where routine information systems are weak or non-existent. These surveys are administered at the household level to collect information about maternal deaths. Names and residences are cross-checked to avoid double counting. Sometimes, they are complemented with verbal autopsies where the family members or other people with knowledge about the death could be asked to describe the situation surrounding the death relatives. The WHO has devised a standard verbal autopsy tool to collect information on signs, symptoms, medical history and circumstances preceding death, 42 which countries can adapt according to their situation. In both population or household surveys and verbal autopsies, names and residences are cross-checked to avoid double counting. These methods are only appropriate for settings in which the sampling unit is a complete village and the geographical scope of the study is quite limited. Surveys, however, require a relatively large sample size to obtain statistically significant findings for occurrences that are relatively rare such as maternal deaths. 35 Sixteen studies included in this review estimated the number of maternal deaths using population and/or household surveys. 38 , 43 – 57 Out of the 16 studies, only 5 were conducted in Africa (Ethiopia, Kenya, Malawi, Senegal and Tanzania). 44 , 52 , 56 , 57 Six were conducted prospectively (Colombia, Sri Lanka, Ethiopia, Indonesia, Jamaica and Pakistan). 38 , 43 , 48 , 50 , 51 , 53 For cultural reasons, family members and birth attendants in Cambodia were reported to have felt ashamed of deaths that had occurred and, therefore, did not report all deaths. There were sampling problems in some of the studies and very wide confidence intervals were obtained for the MMR estimates. 55 , 57 For example, in India, only a small area was covered because it was considered too expensive to conduct a household survey covering a bigger, geographical area.

Direct and indirect sisterhood methods

In sisterhood surveys, adult respondents report on the aggregate numbers of surviving sisters and of sisters who have died. 58 There are two types of sisterhood methods, the indirect and the direct method.

How many sisters (born to the same mother) have you ever had who reached age 15 (who were ever-married) including those who are now dead?

How many of these ever-married sisters are alive now?

How many of these are dead?

How many of these dead sisters died while they were pregnant, or during childbirth, or during the six weeks after the end of the pregnancy?

How many children did your mother give birth to?

How many of these births did your mother have before you were born?

What was the name given to your oldest (next oldest) brother or sister?

Is (NAME) male or female?

Is (NAME) still alive?

How old is (NAME)?

In what year did (NAME) die? OR how many years ago did (NAME) die?

How old was (NAME) when she died?

For dead sisters only:

Was (NAME) pregnant when she died?

Did (NAME) die during childbirth?

Did (NAME) die within two months after the end of pregnancy or

Source: World Health Organization (1997). The Sisterhood method for estimating maternal mortality: guidance potential users. Available on http://apps.who.int/iris/bitstream/10665/64007/1/WHO_RHT_97.28.pdf . Accessed on November 21, 2016.

The data requirements for the direct sisterhood method are considerably more demanding than those for the indirect approach. In the direct approach, a respondent is asked to provide the birth history of her mother, including the current age of all living siblings and the age at death and years since death for all deceased siblings. These data allow deaths and births to be placed in calendar time and, therefore, permit the calculation of sex and age-specific death rates for reference periods. 59 Unlike the indirect sisterhood method, the direct sisterhood method targets a more limited reference period for sister deaths: the previous 0–6 years compared with the previous 10–12 years for the indirect sisterhood method. Point estimates for maternal mortality are obtainable. The approach also allows for the calculation of rates/ratios for the reference period of interest and monitor trends. The direct sisterhood method is currently used during DHS. This method requires larger sample sizes than the indirect method. It also requires an additional 8–10 min per interview on average and additional training and supervision in the field.

Both methods measure the ICD-10 concept of pregnancy-related mortality rather than maternal mortality on the grounds that respondents would not be easily able to distinguish between maternal and pregnancy-related deaths. 39

We did not find any peer-reviewed studies that use the direct sisterhood method apart from the DHS. An analysis of the quality of maternal health indicators for DHS studies is not part of this review and has been described elsewhere. 60 Eleven included studies used the indirect sisterhood method to estimate MMR. Ten studies were conducted in Africa (Liberia, Nigeria (2 studies), Mali, Tanzania (3 studies), Swaziland, Uganda and Ghana). 61 – 69 One study was conducted in India. 70

In Ghana and Uganda, the MMR estimates identified through the indirect sisterhood method were higher than those obtained as national estimates (modelling, UN global estimates) conducted at the same time. 68 , 69 All studies registered pregnancy-related deaths and include death due to accidental or incidental causes (i.e. not maternal deaths). Cause of death is not determined and data collected refers to the previous 10–12 years.

Reproductive age mortality studies

A RAMOS has been identified as a relatively robust method, which uses both active and passive data collection methods to estimate the MMR in countries without VR data and are often considered to be the gold standard. 35 The approach involves retrospective or prospective identification and investigating the causes of all deaths of WRA in a defined area/population by using multiple sources of data such as existing records (CR and health facility data), census, surveys and surveillance. RAMOS are conducted in two phases. The first phase, involves identification of all deaths among WRA in a population. In the second phase, all deaths are investigated (using verbal autopsy, health facility reports or medical record reviews death certificates with medical cause and interview with household members and relatives) to ascertain if there are pregnancy-related or maternal deaths. 71

Four studies conducted in Malawi, Sudan, Jordan and Ghana estimated MMR using the RAMOS method. 72 – 75 Three studies were prospective and one study retrospective. 73 A list of all deaths of WRA was collected using data collated at health facilities (e.g. admission and discharge books, death certificate books, death registers and mortuary logbooks) and individual case notes when necessary, available census data or any other relevant data e.g. the number of births from the most recent DHS and from the Health Management Information System (HMIS). Deaths that occurred in the community were identified by local key informants, traditional birth attendants and community workers. A study in Malawi used the existing health staff (nurses, doctors, medical assistants and community health workers known as health surveillance assistants) at both health facility and community level to identify and report all deaths of WRA. One study in Pakistan included interviews with graveyard caretakers as an additional source of data. In most studies, verbal autopsies where family members or other people with knowledge about the death were interviewed to describe the situation surrounding the death.

All RAMOS studies highlight that this method identifies more maternal deaths than obtained via any one of the existing reporting mechanisms alone (e.g. HMIS and facility death reports). Underreporting of maternal deaths (by 44 and 43%) documented via survey and CR was reported in Ghana and Malawi, respectively. 72 , 75 In Sudan and Jordan, the RAMOS study was conducted at state level, while in Malawi and Ghana the studies were conducted at district and city level, respectively. 73 , 74 In Malawi, verbal autopsy was only done for deaths that were identified as maternal deaths. Maternal deaths were identified using the ICD-10 version 10 definition of MD. 76

Accurate levels of maternal mortality are difficult to measure in a population for it is challenging to identify maternal deaths precisely, particularly in settings where routine recording of deaths is not complete within CR systems. 6 The woman's pregnancy status is usually missed even if such a death was recorded and may not be reported as a maternal death even if the woman was pregnant. Even in countries where routine registration of deaths is in place, maternal deaths may be unidentified due to misclassification of ICD-10 coding and identification of the true numbers of maternal deaths may require special investigations into the causes of deaths. 59 , 77 This review shows that even in high-middle income countries such mechanisms are only now in process of being fully developed. A variety of other methods are used in low- and middle-income settings. Very often the only contemporaneous data available are health facility-based MMR estimates which do not apply to the whole population or estimates obtained via the sisterhood method which are not contemporaneous and report pregnancy-related rather than maternal deaths per se.

Birth and death registration

For birth and death registration systems to provide data on the number of maternal deaths among all deaths of WRA, it is important that pregnancy status is known. Although a tick box has been included on the death notification from, underreporting of the number of maternal deaths and misreporting (misclassification of death of a WRA as a maternal death or not) has been identified as a problem of CR data. 78 , 79 In this review, information on pregnancy status was often either missing or unclear in the identified deaths. 12 – 16 Although countries such as Sweden, the Netherlands, the UK and USA, which have documented reduction in MMR over several decades, have relied on adequate CR systems, misclassification and underreporting exist. 4 , 79 Revision of the death certificate to include information on pregnancy status improves the quality of data and helps to reduce misclassification of maternal deaths. CR data can be compared with data obtained via other systems specific to the evaluation or audit of maternal deaths; countries such as the UK and South Africa have used the Confidential Enquiry into Maternal Deaths (CEMD) to ensure any death missed by the CR system are captured. 2 , 80 , 81 , 82

Health facility data

Valuable information can be obtained when maternal deaths that occur in a health facility are reviewed specifically to identify where the health system needs to improve. 22 , 83 However, in low- and middle-income countries unless >95% of women give birth in a health facility (as opposed to at home), findings from hospital-based studies cannot be generalized to the entire population. However, the majority of the studies from developing country settings reported hospital-based MMR. These only apply to the hospital itself and reflect the type of services provided; large referral hospitals with a large proportion of referred and complicated cases (rather than uncomplicated deliveries) can expect the MMR to be higher than for lower level hospitals (from where patients who are severely ill will be referred out). Thus, facility-based MMR can only be used at the facility level to monitor trends over time and if the proportion of women with potentially life threatening obstetric complications is taken into consideration. It is also crucial that all deaths of WRA are identified and an assessment is made to classify them as maternal deaths or not.

Unless a country has a healthcare system like Saudi Arabia, where almost all maternal deaths take place in the hospital or where all women are brought into hospital soon after death outside the facility, hospital-based data cannot be used to provide accurate estimates of MMR for the population. 84

Census and population or household surveys

The United Nations recommend the use of a population censuses for estimating MMR, without considering this a substitute for VR. 85 Use of census data to calculate MMR is cost-effective as the data can be obtained as part of an already agreed country census. Census data should provide a complete picture of the whole population and results in an estimate with relatively normal confidence indicators due to the large sample sizes. However, a census is usually only conducted every 10 years and cannot be used for routine monitoring. Furthermore, they identify pregnancy-related deaths (not maternal deaths). Early pregnancy deaths may remain under-reported if pregnancy status was not known and maternal mortality can be over-estimated where death was incidental and not due to the pregnancy. This is illustrated in a study conducted in the Republic of South Africa where an increase in maternal deaths was identified when census data were used as compared with a previous MMR estimate which was obtained using a survey method. 40 Despite these limitations, census data still offer the opportunity to measure pregnancy-related mortality as a proxy for maternal mortality in countries with poor or no death registration systems in place.

When specific, planned surveys are used, capturing deaths and births is more complete than with routinely gathered statistics. However, survey methods require prohibitively large sample sizes to obtain statistically significant findings. Such surveys could, however, be used to estimate MMR in resource-limited countries in smaller subsets of populations where the other data sources are not available and/or RAMOS cannot be conducted.

Sisterhood methods

The sisterhood method is cost-effective and easier to perform than prospective population-based methods. Specifically, with the indirect sisterhood method, the number of households that need to be visited in order to obtain information on large numbers of women who have reached reproductive age is relatively small. 58 Given that questions are asked about the deaths of adult sisters, both methods actually measure pregnancy-related deaths rather than maternal deaths, on the grounds that respondents (sisters) would not easily be able to distinguish between maternal and non-maternal deaths and/or usually unable to assign cause of death with certainty. Both methods provide estimates of maternal mortality in orders of magnitude rather than precise ratios since both can have wide margins of error (wide confidence intervals). Neither method provides a current estimate for the year of the survey. For these reasons, sisterhood studies cannot be used to monitor changes in maternal mortality or to assess the impact of safe motherhood programmes in the short term. The sisterhood method has been recommended by the WHO for countries without other reliable source of data and this method is frequently used as part of the 5-year DHS in low- and middle-income countries.

Reproductive Age Mortality Studies (RAMOS)

In the absence of a CR system with/without additional data collection mechanisms such as a CEMD, the RAMOS approach probably provides the most complete and contemporaneous estimation of MMR because information regarding the number of maternal deaths is obtained from a variety of sources and each death among WRA is evaluated to assess whether the death is a maternal death or not. However, the RAMOS approach is difficult in the absence of a reasonably complete initial list of deaths. Inadequate identification of all deaths among WRA results in an underestimation of maternal mortality levels. For example, Surinam had a reliable registration system for deaths which made identification of deaths of WRA relatively easy. 86 Similarly, during the prospective RAMOS in Pakistan, good population-based systems were in place for tracking deaths. 48 This meant that the number of maternal death among deaths of WRA could be assessed. RAMOS studies can be expensive and time consuming when conducted on a larger scale. 14 A RAMOS may, therefore, be considered to provide accurate MMR data for a sub-national population.

To end preventable maternal deaths, it is crucial that countries develop systems and processes to ensure the ability to count every maternal death and identify the cause of death and contributing conditions. This will help identify where and how the availability or coverage as well as quality of care need to be improved. Ideally, MMR estimates should be obtained from CR data, which provide both numerator and denominator data. This would also assist in monitoring any trends in MMR over time. Many low- and middle-income countries are in the process of introducing CR systems for births and deaths. For countries without reliable systems in place, a RAMOS can be an effective method that can be used to obtain recent data and provides better estimates of MMR.

A RAMOS approach can also help illustrate what is needed to support the introduction of a full-scale Maternal Death Surveillance and Response (MDSR) process. The MDSR builds on the principles of public health surveillance and response by collecting accurate information on cause of maternal deaths so lessons can be learnt and actions taken to prevent similar deaths in the future and to improve quality of care.

Florence Mgawadere PhD Florence, a nurse-midwife from Malawi with over 12 years of international experience in teaching, research and technical assistance. Florence's current research interests include quality of care, application of the ICD-MM cause classification for maternal deaths, maternal death audit or review and strengthening of Maternal Death Surveillance and Response (MDSR). Florence has carried out research on maternal health, including at the community and facility levels and programme evaluations. Florence works as a Senior Research Associate at the Centre for Maternal and Newborn Health at the Liverpool School of Tropical Medicine.

Terry Kana MSc Terry, an experienced teacher in midwifery and public health from community to postgraduate level, has worked in a wide variety of hospital and community settings with over 10 years of international programme management and research experience. Her current research interests include assessment of the effectiveness of competency based ‘skills and drills’ training in emergency obstetric care, the role, scope of work and workload of nurse-midwives working in low- and middle-income settings. Terry works as a Senior Research Associate at the Centre for Maternal and Newborn Health at the Liverpool School of Tropical Medicine.

Nynke van den Broek MBBS, DTMH, PhD, FRCOG Professor van den Broek is a recognized international expert in global maternal and newborn health who established and leads the Centre for Maternal and Newborn Health (CMNH), one of the largest academic groups in Europe with an internationally recognized portfolio of work that incorporates priority interventions for reducing maternal and newborn mortality and morbidity. Four key thematic areas include skilled birth attendance, emergency obstetric care, quality of care and maternal morbidity. Professor van den Broek has designed and conducted large population-based randomized controlled trials of single interventions for improved maternal and newborn outcomes. She has used this experience to develop complex packages of interventions and to design and conduct operational research programmes in multi-country settings.

Supplementary material is available at BRIMED Journal online.

The authors have no potential conflicts of interest.

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• developing countries
• maternal mortality
• reproductive physiological process

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• Open access
• Published: 18 May 2021

The influence of social and cultural practices on maternal mortality: a qualitative study from South Punjab, Pakistan

• Sonia Omer 1 ,
• Rubeena Zakar 1 ,
• Muhammad Zakria Zakar 2 &
• Florian Fischer   ORCID: orcid.org/0000-0002-4388-1245 3 , 4  

Reproductive Health volume  18 , Article number:  97 ( 2021 ) Cite this article

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A disproportionately high rate of maternal deaths is reported in developing and underdeveloped regions of the world. Much of this is associated with social and cultural factors, which form barriers to women utilizing appropriate maternal healthcare. A huge body of research is available on maternal mortality in developing countries. Nevertheless, there is a lack of literature on the socio-cultural factors leading to maternal mortality within the context of the Three Delays Model. The current study aims to explore socio-cultural factors leading to a delay in seeking care in maternal healthcare in South Punjab, Pakistan.

We used a qualitative method and performed three types of data collection with different target groups: (1) 60 key informant interviews with gynaecologists, (2) four focus group discussions with Lady Health Workers (LHWs), and (3) ten case studies among family members of deceased mothers. The study was conducted in Dera Ghazi Khan, situated in South Punjab, Pakistan. The data was analysed with the help of thematic analysis.

The study identified that delay in seeking care—and the potentially resulting maternal mortality—is more likely to occur in Pakistan due to certain social and cultural factors. Poor socioeconomic status, limited knowledge about maternal care, and financial constraints among rural people were the main barriers to seeking care. The low status of women and male domination keeps women less empowered. The preference for traditional birth attendants results in maternal deaths. In addition, early marriages and lack of family planning, which are deeply entrenched in cultural values, religion and traditions—e.g., the influence of traditional or spiritual healers—prevented young girls from obtaining maternal healthcare.

The prevalence of high maternal mortality is deeply alarming in Pakistan. The uphill struggle to reduce deaths among pregnant women is firmly rooted in addressing certain socio-cultural practices, which create constraints for women seeking maternal care. The focus on poverty reduction and enhancing decision-making power is essential for supporting women’s right to medical care.

Plain language summary

Round the world, many women are dying because of complications during pregnancy or in childbirth. These deaths are more frequent in developing and underdeveloped countries. Some reasons for this are related to social and cultural factors, which form barriers to women using appropriate maternal healthcare. Therefore, this study aims to explore socio-cultural factors leading to a delay in seeking maternal healthcare in South Punjab, Pakistan.

We interviewed a variety of people to get an overview of this topic: (1) 60 interviews were conducted with gynaecologists, (2) we performed four focus group discussions with eight to ten Lady Health Workers providing maternal healthcare, and (3) we talked with family members of mothers who had died.

The study shows that delays in seeking care are related to poor socioeconomic status, limited knowledge about maternal care, and low incomes of rural people. The low status of women and male domination keeps women less empowered. In addition, early marriages and lack of family planning due to cultural values, religion and traditions stopped young girls from getting maternal healthcare.

The number of new mothers who die is very worrying in Pakistan. One of the important tasks for reducing deaths among pregnant women is to address certain socio-cultural practices. It is very important to reduce poverty and improve decision-making power to make sure women can use their right to medical care.

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Reducing maternal mortality is among the key determinants of development strategies for countries all over the world [ 1 ]. Globally, every two minutes a pregnant woman dies, either due to complications during pregnancy or in childbirth [ 2 ]. Almost 300,000 women died due to pregnancy-related complications in 2017. The great majority of these deaths (94%) occurred in countries with low resources [ 3 ]. With a population of approximately 204.6 million people, Pakistan is the sixth most populous country in the world. In Pakistan, the maternal mortality rate was 140 per 100,000 live births in 2017 [ 4 ]. Although there have been significant improvements in the country’s healthcare system, Pakistan still faces many challenges in relation to its high population growth, infant and maternal mortality, and many infectious and non-infectious diseases [ 5 ]. In terms of development indicators, Pakistan has shown poor performance, specifically with reference to maternal healthcare [ 6 ]. Maternal healthcare-seeking behaviour in Pakistan, particularly in rural areas, is deeply influenced and constrained by a series of religious and cultural factors [ 7 ].

Many studies have documented the association between religious, social and cultural beliefs and the health risks faced by childbearing women [ 7 , 8 ]. For example, some studies on Muslim women show that they usually opt for ‘faith-based’ healthcare services. These services consist, to a large extent, of traditional or spiritual healers without an academic background to support their business. In particular, families who are impoverished and have limited access to education are marginalized from accessing biomedical healthcare services. This in turn creates distrust in the healthcare system and strengthens the preference for traditional healers. For that reason, women disregard proper medical attention during their pregnancy, resulting in negative effects on their reproductive health [ 9 ]. Globally, the development of science and technology has helped to overcome these practices associated with pregnancy. However, in most developing countries, people with low socioeconomic status continue to follow certain beliefs and cultural rituals, despite there being no scientific evidence available in their favour [ 10 ]. It has been significantly revealed that the societal norms, values and culture of any country has a substantial effect on its maternal mortality rate [ 10 , 11 ]. All this is deeply rooted in the role and status of women within the society [ 12 ]. Pakistan is a strongly patriarchal society and thus men largely dominate household decision-making and finances. Women, particularly those living in rural areas and urban slums, are treated as subordinates and have limited or no say in personal and family matters [ 13 ]. The disadvantaged status of women and the male domination of society have adverse impacts on women’s reproductive health [ 14 ].

Worldwide, maternal healthcare systems have used numerous different approaches to improve the understanding of the difficulties faced by women during pregnancy or in childbirth. One of the most noteworthy of these approaches is the Three Delays Model [ 15 ]. According to research conducted by Thaddeus and Maine in 1994, complications during pregnancy can be avoided if adequate and timely treatment is provided. However, if treatment is delayed and/or insufficient, the consequences can be serious. These delays are threefold: (1) delay in making the decision to seek care, (2) delay in reaching a healthcare facility, and (3) delay in receiving the required maternal healthcare. The first delay is about recognizing maternal complications at the earliest possible time and making efforts to seek appropriate medical care immediately [ 15 ]. This type of delay is estimated to contribute to 73% of maternal deaths [ 16 ]. A huge body of research is available on maternal mortality in developing countries. Nevertheless, there is a paucity of literature on socio-cultural factors leading to maternal mortality within the context of the Three Delays Model—specifically the first delay. Thus, this study aims to explore the socio-cultural factors leading to a delay in seeking maternal healthcare, that is, the first delay, in maternal healthcare utilization in South Punjab, Pakistan.

Study design

We used a qualitative method for this study and performed three types of data collection with different target groups: (1) 60 key informant interviews with gynaecologists, (2) four focus group discussions with Lady Health Workers (LHWs), and (3) ten case studies among family members of deceased mothers.

The study was conducted across all four districts of the division of Dera Ghazi Khan (DGK), South Punjab, Pakistan. The reason for selecting this division is that DGK remained lowest in significant socioeconomic indicators among all divisions of Punjab in 2017–18 [ 3 ]. For instance, 56.9% of the population of DGK is living below the poverty line, which is the highest proportion of all the divisions of Punjab province. Furthermore, the literacy rate among young women aged 15–19 years is only 48.9%, which is the lowest in the province. Similarly, the maternal mortality indicators reveal a very alarming situation for women in the area. Only 36.3% of women receive four or more antenatal care visits and only 29.8% of women have deliveries overseen by skilled birth attendants. Many women (43.0%) prefer traditional birth attendants (TBAs) for delivery and consultation during or after pregnancy, which is again the highest proportion of any division in Punjab province [ 3 ].

Data collection

The 60 key informant interviews with gynaecologists at the principal hospitals in all four districts of DGK were performed by the first author. The interview guide (Additional file 1 ) was developed on the basis of a literature review and informal discussions with physicians. This interview guide consisted of questions concerning the socio-cultural factors contributing to a delay in seeking maternal healthcare—and in many cases eventually even leading to maternal death. The interviews lasted between 60 and 90 min. Interviews were conducted until we reached saturation point, where almost all responses were similar to those in previous interviews and no new information was emerging from the data. The interviews with healthcare professionals were conducted at their workplaces in the respective hospitals at a time of their choosing.

Considering the significance of LHWs, who have direct and close interactions with families and women in rural areas, four focus group discussions with LHWs were also conducted, one in each district. The LHW programme is a key part of Pakistan’s national strategy to reduce poverty and improve the health sector [ 17 ]. LHWs bring health services to the doorsteps of under-served and marginalized sections of society [ 18 ]. Therefore, LHWs are the community health workers who are most likely to provide healthcare information, including awareness of maternal health issues [ 19 ]. The list of LHWs within all four districts was obtained from the health department and contacts were made. The LHWs were requested to attend the principal hospital of the relevant district. They were facilitated and given a transport allowance by the research team. Eight to ten LHWs participated in each focus group discussion, which lasted between 60 and 90 min each.

In order to obtain the points of view of families where a maternal death had taken place, ten case studies among family members of deceased mothers were also conducted. The participants in these case studies were selected with the help of LHWs. They identified a person who had remained close to the deceased mother throughout her pregnancy and delivery. The interviews took place in the homes of the deceased mothers after receiving consent from the study participants. The maternal death case was included if the death had taken place during the previous two years in order to minimize recall bias. Maternal death was considered only if the death had been reported as an outcome of complications during pregnancy, in childbirth or during the postpartum period. To ensure valid responses, the interviewee had to be someone who had been close to the mother and was aware of all the details regarding the death. In most cases, the husband was reported as the main family member who had remained with the deceased mother. In a few cases, the mother-in-law or sister-in-law of the deceased mother were identified by LHWs as the main person who had remained with the deceased mother throughout her pregnancy and childbirth.

Data analysis

All the interviews and focus group discussions were audio recorded and notes were taken during data collection. As interviews progressed, some participants used their native language of Punjabi (the regional language of Punjab province) and others spoke in Urdu (Pakistan’s national language). All the interviews were transcribed in Urdu and afterwards translated into English. Data analysis was performed manually using thematic analysis. All initial codes related to the research questions were joined together and transferred into a theme. The study used both a deductive and inductive approach for data analysis. During the analysis, a few themes were assessed based on previous literature related to the first delay in seeking care within the Three Delays Model developed by Thaddeus and Maine (deductive) [ 15 ]. As we continued the analysis process, inductive themes emerged from the data. Analytical induction and constant comparison of the categories were applied. Sub-themes and categories were themes that primarily emerged during notetaking, transcribing, translating and interpreting the data [ 20 ].

Table 1 presents the socio-demographic profile of the LHWs who participated in the study. Almost three quarters of the participants were aged between 30 and 40 years. The majority (77.8%) was married and about half (42.9%) had four or more children. Most of the LHWs (58.3%) were earning between 50,000 and 75,000 Pakistani rupees. One third (33.3%) had work experience of between five and ten years and a further 44% had work experiences of more than 15 years.

Table 2 presents the socio-demographic profile of the physicians—all of them women—working on gynaecology and obstetrics wards within the study area. The largest group (46.6%) of physicians was aged 30–35 years and 31.6% were aged 36 years or above. A great majority (70.0%) had an MBBS with specialization in gynaecology. About half of the sample (53.3%) had experience of working on gynaecology wards for 10 to 15 years. A significant proportion (70.0%) of the physicians was married. Among the married physicians, 66.6% had between one and three children. Additionally, 43.3% reported a monthly income between 100,000 and 125,000 Pakistani rupees per month.

We investigated the social and cultural factors contributing towards delayed decision-making in seeking healthcare for women during pregnancy. The themes derived from the data analysis are presented below.

Low status of women

Despite many governmental efforts at gender mainstreaming, Pakistan is far behind most nations in achieving gender equality in health, education, and economic and political participation for women. Women are still subjected to different forms of discrimination and have little or no say on personal or family matters [ 21 ]. The present study found that illiterate and socially isolated women were more vulnerable to poor reproductive and general health. There were certain social and cultural practices, such as purdah (the veil system), dependency on a male guardian, and other social restrictions on the independent mobility of women that deprived them of the ability to seek timely medical care during pregnancy and childbirth. A majority of LHWs (32 of 36) were of the view that, in rural areas, many women could not read or write because education was not considered necessary for them. One mother-in-law had a clear stance against women’s education, saying:

Why bother with school or college education for girls? The ability to read and write is good but the most important thing is that they should be able to read the Holy Quran. And she should have the skills of cooking and home making. This is what every Muslim woman must learn.

While discussing the healthcare needs of women, especially during pregnancy, the wife of a retired military spy said:

During pregnancy, the family—especially the husband and mother-in-law—must be careful and considerate. If there’s a problem, it’s the duty of the husband to arrange a visit to a qualified care provider.

Lack of autonomy and mobility

In the local culture of Pakistan, women generally lack autonomy to seek care when they need it. It was noted that there were many “stakeholders” whose consent was necessary before a pregnant woman could embark upon seeking care from a health facility. One LHW explained:

It’s not the decision of the pregnant woman when, why and from whom to seek healthcare. Usually, it’s the joint decision of many players, including the mother-in-law, father-in-law, husband and sometimes the husband’s brother. They make the decision according to the perceived severity of the illness, the cost, the nature of the threat, the availability and competence of care providers, and other conditions.

A majority of physicians (50 of 60) reported that sometimes the decision to travel to a healthcare facility was not based on women’s health condition but on the availability of transport as well as the availability and willingness of their husband, father or brother to travel with them. A gynaecologist pointed out the restrictions on the mobility of women:

Most of the time, mothers with pregnancy complications are brought to us [referring to gynaecologists] only when they’re near to death. Yesterday, I received a pregnant mother for delivery. She was in a critical condition with profuse bleeding. When I asked the family about this delay, they told me that they were waiting for her husband to bring her to hospital because culturally it’s forbidden for a woman to go alone.

Another dimension of the lack of decision-making power is illustrated by one of the LHWs:

Sometimes, women don’t want to make their decisions independently because it has serious consequences if something goes wrong. For example, if a woman selects a particular doctor for treatment and if the pregnancy is terminated by this treatment, the woman will be in trouble. So, she needs to take other family members into her confidence while making the decision.

Low nutritional status

Due to the low social status of women, they also experience discrimination at home. Sometimes, they are not provided with the proper food which is required during pregnancy, and their dietary needs are frequently ignored. A healthy and balanced diet during pregnancy and after childbirth is one of the significant factors affecting women’s health. The absence of good nutrition can have adverse effects on a mother’s health and can lead to maternal complications [ 22 ]. A gynaecologist in her early 30 s expressed her view on women’s poor nutritional status:

I become horrified to see the pale ghostly faces of women who come to us with maternal complications. Giving food to the men within the house first, and then the children, is the cultural thing here. Women’s dietary needs are not their priority.

The LHWs, being close to rural households, had their own observations to make about the obstacles causing the first delay in seeking healthcare for pregnant women. Some of the LHWs (15 of 36) were of the view that one of the main obstacles was poverty and powerlessness within the family power structure. A senior LHW said:

In poor families, pregnant women can’t even get two decent meals; not to mention timely and proper medical care.

Another middle-aged woman, who had retired from being an office attendant at the local middle school, said:

Here, the main issue is not poverty but priority and preference. Women from a poor background—whose parents are poor and not influential—are not properly cared for in their in-laws’ homes. They are simply ignored; the issues of their health and illness are taken for granted.

While probing families about any special focus on the pregnant mother’s diet, one mother-in-law commented:

I can’t feed the pregnant mother first, if the children are crying out of hunger. My mother-in-law didn’t pay special attention to me nor did I with my daughter-in-law. What’s so special about giving birth?

Early marriages

In Pakistan, especially in poor rural families, early marriage, forced marriage and cousin marriage are common and considered normative cultural practices. A growing body of literature demonstrates the negative physical consequences of early marriages on young girls [ 23 ]. A few of the physicians (15 of 60) reported that child marriage was one of the main reasons for maternal mortality. According to them, a girl married at a young age was not mature enough to decide about her own healthcare and she was more dependent on her in-laws and husband for her healthcare needs. In the local culture, child marriage is justified by providing many reasons. One of the mothers-in-law expressed her strong belief in girls’ early marriage:

Poor and powerless people are not safe here—so is the case with their daughters. We can’t afford to keep daughters unmarried for long at home.

One of the LHWs said:

This is a general perception here: The younger the girl is, the brighter are the chances of producing more children. Therefore, many people think that it’s a cultural thing, and they follow it.

A female physician shared her views in this regard:

In this area, girls are married at an early age and they have multiple pregnancies before reaching the age of 25 years. Sometimes I refuse to believe the age of the pregnant women when I’m told it. The fact is, they usually look 10 or 20 years older than their actual age. They’ve been producing children every year and the chances of maternal mortality with such health conditions are always higher.

One LHW who had been working in a village community for the past ten years added:

The poor parents are always in a hurry to marry off their daughters to lessen their burden. Culturally, people think marrying daughters early prevents them from becoming characterless. If girls remain unmarried after attaining puberty, there is a risk of creating affairs or sex scandals.

One female physician emphasized the dependency which goes along with early marriage:

I’ve assisted in the deliveries of many young mothers in this community. Frequently, they’re brought to us with pregnancy complications when it’s difficult to save their lives. They [referring to the young mothers] are not prepared to decide for themselves, about their family planning. And they don’t know anything about their reproductive health. They’re totally dependent on their in-law families for such decision-making.

Lack of reproductive autonomy

Due to their social exclusion and economic non-participation, women are not fully aware of their reproductive rights. In some areas, the birth of a baby girl is not welcomed; rather, it disempowers the mother who gave birth to a daughter. Therefore, a woman who is pregnant with a baby girl is less likely to seek appropriate and timely care during pregnancy. The present study showed a lack of family planning among married couples and that the average family size in rural households is large. A few of the case study participants (4 of 10) shared their beliefs that family planning is considered a sin in their religion. The majority of LHWs (30 of 36) were of the opinion that the desire for a son was the primary factor in a large family size. One husband with very poor socioeconomic status admitted:

My wife died during the birth of our seventh child and I admit I never followed any family planning. I know very well it’s a conspiracy against Muslims.

One LHW shared her experience and added:

I was once physically abused by a mother-in-law and husband when a young married girl – who died in childbirth later – asked for a contraceptive pill, but tried to hide it from her family. Here, culturally and religiously, people think it’s a sin to follow family planning methods.

Within the household power structure, mothers-in-law have more power. They can influence decisions related to the reproductive life of their daughters-in-law, including their health-seeking behaviour during pregnancy. When reproductive decisions are made by someone else and not by the mother, timely decisions about healthcare are difficult to achieve. The same observation was made by many participants (76 of 96) in terms of the role of mothers-in-law in the lives of their daughters-in-law. A female physician on a gynaecology ward revealed:

The mothers-in-law are the ones who decide the next course of action once a pregnant mother is brought to us. I’ve even seen them insisting on saving the life of a baby in place of its mother, specifically if it’s a baby boy.

When commenting on the situation, one LHW noted:

When things are decided by the mother-in-law regarding seeking care, she has her own ‘agenda’. She may delay the visit to save money, to avoid travel or simply to settle a score with the pregnant mother. It’s unfair and doesn’t make sense; but this is how it is.

Poor understanding of pregnancy complications and risk factors

The present study found that women and their families were not very aware of pregnancy complications or the related risk factors. It is evident that a timely diagnosis of complications during pregnancy is possible, if antenatal visits are available for pregnant mothers [ 24 ]. However, the local culture has its own understanding of pregnancy and its associated processes. More than half of the LHWs (20 of 36) reported that many pregnant women had no opportunity to visit medical facilities for antenatal care in their local areas. Many of the LHWs (26 of 36) and gynaecologists (52 of 60) blamed the families for this situation. It was also noted that there is a lack of trust in certain diagnostic medical procedures performed at healthcare facilities for pregnant women. One female physician observed the following:

Sometimes, there’s a serious lack of trust between doctor and patient. Some mothers are suspicious of ultrasound and think it’s a family planning device. Lack of trust is also a factor in delays to seeking formal care.

While explaining the condition of rural women, one LHW stated:

Poor women—who are the majority in this village—have no concept of prenatal care. They’re taken to healthcare facilities when something visibly serious happens to them such as bleeding, fits or they simply lose consciousness. For minor ailments, they’re treated at home.

While describing the need to seek healthcare during pregnancy, one mother-in-law stated:

Problems in pregnancy are normal and natural. Why rush to doctors for a natural process? For thousands of years, women have been delivering children at home. Doctors just complicate things to make money.

One gynaecologist reported:

Who cares about their treatment or antenatal check-ups? In the local culture, pregnancy is kept secret. A web of superstitions regulates the lives of pregnant women. They only come to us at a very critical stage.

One of the sisters-in-law of a deceased mother revealed:

My mother-in-law believes in keeping pregnant women inside the home. Therefore, she couldn’t be exposed to sunlight during the first three months of pregnancy. She did the same with my sister-in-law, who died in the fifth month of pregnancy due to some complication, because she was not allowed to go to a doctor due to her [referring to her mother-in-law] superstitious beliefs.

Seeking care within a pluralistic medical system

Like other developing countries, Pakistan has a complex pluralistic medical system in which the biomedical system coexists and competes with a host of indigenous medical systems, such as traditional healers ( hakeems , folk healers, or spiritual healers). Depending on a patient’s social class, level of education, income and the perceived nature of their aliment, the patient selects a particular care provider, or multiple care providers, at any given time. A majority of the physicians (50 of 60) reported that getting multiple sets of advice and treatment from multiple care providers could cause a delay in seeking treatment from a qualified care provider. One physician noted:

Women come to us with long-term complications such as high blood pressure or gestational diabetes. Poor women fail to understand the long-term treatment and ask for a quick remedy. Here come the traditional and spiritual healers: They promise quick relief.

South Asian communities still attribute many physical and mental illnesses to the presence of supernatural powers and seldom hesitate to consult spiritual healers [ 8 ]. The role of spiritual religious leaders, even in providing consultations for medical care during pregnancy, is common. One of the respondents, who was a sister-in-law of a deceased mother, and who was also pregnant said:

I’ve been advised by peer sahib [referring to spiritual healers] to keep a knife under my pillow and avoid sunlight throughout my pregnancy. My mother-in-law believes that it could save me from future complications.

While explaining the mechanism of decisions being delayed by spiritual healers, one LHW added:

I was once called upon to see a woman who had profuse bleeding in her fifth month of pregnancy. While probing, I learned that she was stopped by a local peer [referring to a spiritual healer] from travelling outside her home or consulting a doctor as she ran the risk of being attacked by evil forces. She died the same evening.

The mother of a deceased women shared:

Here, we have a strong belief in nazar lagna [the evil eye]—especially during pregnancy. My daughter’s mother-in-law did not allow her to go outside the home or consult a doctor. Always the Dai [a TBA] came to her home to provide treatment. But she [referring to the Dai] did a very bad job with my daughter. She took my daughter’s life. She and my daughter’s in-law’s family are responsible for her death.

A physician on a gynaecology ward expressed great disappointment in the large adverse impact of cultural and religious beliefs on pregnant women:

These spiritual healers are part of the religious and cultural beliefs of rural people. Sometimes, a husband comes or not. But when a pregnant mother is close to death and is brought to us, the local traditional healer accompanies the family and even intervenes in our treatment methods.

Spiritual healers of various types influence the health belief systems of women during pregnancy, which in turn regulates their care-seeking behaviours [ 25 ]. Frequently, pregnant women and their families in villages are dependent on TBAs for healthcare. The families in our study greatly preferred TBAs and sought help from them for women during pregnancy. A mother-in-law in her late 70 s showed her great trust in these TBAs:

Let us not break with tradition. In the four walls of our house we get great help from Dai [referring to TBAs] during pregnancy and for delivery. The poor Dai is happy to receive a few kilos of Atta [wheat flour] as her fee, even after delivering the baby. I’m happy they’re always available for us.

An experienced LHW, however, expressed her anger about the role of TBAs:

I’m helpless when I see these Dais treating cases of preeclampsia and eclampsia [referring to high blood pressure during pregnancy] with herbal medicine. They don’t hesitate to cut the baby’s umbilical cord with a knife used for vegetables. Families blindly trust in them and no force can stop them.

The gynaecologists shared their own experiences of dealing with complicated cases brought to them after treatment from untrained TBAs. One senior gynaecologist shared her views by commenting:

The half-dead pregnant mothers are sometimes brought to us with serious complications. Most of the time, they are brought to us after inappropriate interventions by untrained Dais, and Dais are unable to handle the delivery. In my eyes it’s a killing, it’s a murder.

It is a globally accepted phenomenon that the factors determining health behaviours are found in different contexts. These can be physical, social, economic or cultural in nature [ 26 ]. The same goes for maternal healthcare in Pakistan, particularly in rural areas. This study found that various socio-cultural factors contribute towards the first delay in decision-making (delay in seeking care) about appropriate maternal care, which has an impact on maternal mortality in South Punjab [ 15 ]. Many studies have found that the first delay is the most significant contributor to maternal deaths [ 16 , 27 ]. The qualitative data gathered in this study shows that healthcare-seeking behaviours during pregnancy are extremely complex and embedded in a cultural and indigenous belief system. Additionally, prevailing economic conditions, patriarchal ideology, and the role and status of women within the family power structure also influence women’s healthcare-related decision-making [ 28 ].

We argue that the low socioeconomic status of women is one of the major obstacles affecting women’s decision-making in seeking healthcare during pregnancy and childbirth in rural areas and urban slums [ 28 ]. This low social status of women exposes them to multiple negative social and cultural practices, such as early marriage, multiple and closely spaced pregnancies and domestic violence [ 29 ]. We observed that women’s strong socioeconomic dependency on men throughout their lives hinders them from deciding about their own health and wellbeing. The limited autonomy of women, their lack of education, and their dependency on men leaves them at the mercy of their husband’s family to look after them during or following pregnancy [ 30 ]. We found that women were not provided with sufficient nutrition during pregnancy. This indicates that poverty or lack of resources is a prime factor affecting women’s health. A healthy and balanced diet during pregnancy and after childbirth is one of the most important factors for women’s health. The absence of an adequate and balanced diet can have adverse effects on a mother’s health and can lead to maternal complications [ 22 ].

Cultural restrictions on women’s mobility are another barrier noted in our study. Restrictions on women’s mobility during obstetric emergencies may lead to a delay in seeking timely care [ 31 ]. The women were found to observe purdah (the veil) under all circumstances and were not allowed to travel outside the home without men even during a health emergency. The consent of “stakeholders”—i.e., the husband’s and mother-in-law’s permission—was revealed as necessary for a woman in relation to significant events in her life—ranging from family planning issues to her choice about consulting an appropriate maternal caregiver [ 32 ]. Women going out alone due to a medical emergency are thought to bring great dishonour to the family. Making a timely decision about selecting the right kind of healthcare provider is critical for avoiding maternal and child deaths. Unfortunately, for some poor rural women, this decision is not simple [ 33 ]. Our study highlights that delays in seeking care for obstetric complications are influenced by traditional ‘wait and see’ tactics. The action of seeking medical care is usually undertaken only when the situation is already out of control or the condition of the pregnant woman worsens [ 27 ].

It was also noted that the practice of deciding family size was also in the hands of the mother-in-law or the husband, for cultural reasons. Many studies have highlighted the influential role of mothers-in-law and husbands in women’s lives regarding decisions about family planning and giving birth to children [ 33 , 34 ]. Among others, the lack of family planning among rural couples was found to be a pivotal factor resulting in high maternal deaths [ 33 ]. The participants in the case studies reported multiple socio-cultural factors behind the low use of family planning methods and short birth spacing amongst married couples. The religious interpretation regarding family planning among rural couples was an important element, as has already been explained in previous research [ 34 ]. Rural people believe that the use of family planning methods and restricting family size is against the preaching of Islam. A few reported the use of family planning as a conspiracy against Muslims to limit their population growth [ 35 ]. A large majority of religious groups in developing countries do not favour birth control methods and call them un-Islamic and unnatural [ 36 ]. Another cultural factor noted for leading to a large family size was families’ desire for a son [ 37 ]. The present data reveals that this particular desire pushed women to give birth almost every year. Multiple pregnancies in turn lead to higher risk.

This study also shows that Pakistan, with its patriarchal society, has visibly delineated roles related to gender. The household structures in villages, where mothers-in-law and husbands hold major positions, hardly allow women any power to make decisions regarding their own lives [ 38 ]. A similar influence was found in relation to the use of antenatal care visits. Our results indicate very low use of antenatal care, which greatly contributes to the first delay. A growing body of knowledge has revealed a lack of awareness amongst women and their families about the delivery and continuity of antenatal care [ 39 ]. Some of the results that have emerged from this study, including the low use of antenatal care, are similar to studies conducted in other South East Asian and South Asian countries [ 40 , 41 , 42 , 43 ]. The data from this study also indicates the strong cultural trait of early marriage. Early marriages were seen to be the root cause of multiple pregnancies. These are the notable social and cultural conditions that increase the lifetime risk of maternal death [ 44 ]. The primary cause noted for early marriage in rural areas was the perception of daughters as a burden. There is suspicion about the character of girls if they remain unmarried for too long. The fact that girls were young and under the influence of their mothers-in-law and husbands was found to be an important factor in their inability to seek the desired maternal care [ 44 ]. Previous studies have shown that bearing a child at an early age increases the risk of several medical complications and, therefore, leads to an increased risk of complications during pregnancy or immediately after childbirth [ 44 , 45 ].

In addition to that, the culture of relying on TBAs is a common social and cultural practice in rural areas of Pakistan [ 46 ]. Dependency on TBAs is found to be a great barrier to seeking appropriate maternal care. The majority of TBAs are largely untrained and without clinical expertise to handle complicated situations during pregnancy or childbirth [ 46 ]. This study documents that rural communities are inclined towards their centuries-old traditions of trusting and utilizing the services of TBAs. They are the preferred choice, and families are not ready to compromise on that. Families find them easier to afford and it is convenient to call them when needed. In addition, they are available 24 h a day and provide services on the doorstep [ 47 ]. The issue of purdah (the veil) is also not compromised in such cases, because it saves women from going out to consult physicians, who might in some cases be male. However, a previous study attributed complications during pregnancy or after childbirth to uneducated and untrained TBAs [ 48 ].

This study has also shed light on traditional and customary beliefs about spiritual healers. From medication to advice on the daily matters of life, the advice of spiritual healers is considered highly important. As a matter of fact, this is known to be a social issue in rural areas [ 49 ]. Visiting these spiritual healers in rural communities is another ritualistic practice in rural areas that prevents women from seeking maternal care from medical practitioners. In addition, a strong belief in witchcraft or the evil eye directed against pregnant women is a major component of many cultures [ 50 , 51 , 52 , 53 ]. In order to ward off evil spirits and the influence of supernatural powers on pregnant women, the elderly of the family, most frequently the mothers-in-law, take the decision to consult spiritual healers [ 54 , 55 ]. Rural people believe in particular rituals and practices, such as the chanting of certain verses, a spiritual healer’s blow to the face or body of a woman, or simple water as a great medication for maternal complications [ 55 ]. These spiritual healers were found to be influential figures in rural women’s lives in decisions ranging from the place of birth to permission for her mobility during pregnancy and family planning. This practice is very common in Pakistan and families prefer spiritual healers over professional and trained physicians [ 55 , 56 ]. Therefore, it might be advisable to include TBAs more in the medical health system by providing them with adequate training. Since TBAs have access to a large part of the population, they may be able to transfer knowledge about the impact of appropriate maternal healthcare. This may change sociocultural beliefs in the long term.

Limitations

One of the major strengths of our study is the large sample size for a qualitative study and the use of three different types of data collection (key informant interviews, focus group discussions, and case studies) that included diverse groups of interviewees. The synthesis of these results allows for an in-depth examination of the social and cultural factors associated with maternal mortality. However, one needs to keep in mind that these results describe the specific situation in a rural and impoverished region of Pakistan. A further limitation is associated with the data analysis. The thematic analysis is based on what was spoken during the interviews and focus groups. However, attitudes that were not expressed aloud and non-verbal information are not included in the data analysis.

Maternal health in Pakistan is strongly influenced by socio-demographic elements, societal structures, cultural practices and religious beliefs, gender discrimination and the lack of autonomy among women. The situation of maternal mortality is very alarming in the country. The uphill task of reducing deaths among pregnant women is deeply embedded in addressing certain sociocultural practices which are constraints for women seeking maternal care. Despite governmental efforts to provide maternal care to rural women in Pakistan, social practices and cultural beliefs play important roles in deciding which women will survive and which will not. It is absolutely pivotal to identify the causes of maternal deaths as early as possible. Maternal deaths can be easily prevented if women are saved from putting off seeking care. The important key to reducing maternal mortality is to address the poor economic and social status of rural families. A strong emphasis is required on raising the status of women in their communities through education and economic empowerment. Without addressing the social and cultural practices in the broad integrated strategies aimed at improving maternal health in Pakistan, the average Pakistani mother will continue face a high risk of maternal mortality and will leave behind tales of misery, discrimination and vulnerability.

Availability of data and materials

Pseudonymised transcripts are available from the corresponding author upon reasonable request.

Abbreviations

Dera Ghazi Khan

Lady Health Worker

Pakistani Rupee

Traditional birth attendant

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Acknowledgements

We acknowledge support from the German Research Foundation (DFG) and the Open Access Publication Fund of Charité—Universitätsmedizin Berlin.

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Sonia Omer & Rubeena Zakar

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Muhammad Zakria Zakar

Institute of Public Health, Charité – Universitätsmedizin Berlin, Berlin, Germany

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The interviews were part of the PhD thesis conducted by SO. SO conducted data collection and analysis, RZ supervised this process. SO analysed the data in collaboration with RZ and MZZ. SO drafted the manuscript, RZ, MZZ and FF revised it critically for important intellectual content. All authors read and approved the final manuscript.

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Ethical approval was obtained from the University of the Punjab, Lahore, Pakistan. Furthermore, the Health Department, Government of Punjab, Pakistan, also granted permission to conduct the study. Written informed consent was obtained from each respondent after explaining the objectives of the study and assurance of confidentiality of their identity.

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Omer, S., Zakar, R., Zakar, M.Z. et al. The influence of social and cultural practices on maternal mortality: a qualitative study from South Punjab, Pakistan. Reprod Health 18 , 97 (2021). https://doi.org/10.1186/s12978-021-01151-6

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Knowledge gaps in scientific literature on maternal mortality: a systematic review

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• 1 Preventive Medicine and Public Health, University of Alicante, San Vicente del Raspeig (Alicante), Spain. [email protected]
• PMID: 17143465
• PMCID: PMC2627556

Issues related to maternal mortality have generated a lot of empirical and theoretical information. However, despite the amount of work published on the topic, maternal mortality continues to occur at high rates and solutions to the problem are still not clear. Scientific research on maternal mortality is focused mainly on clinical factors. However, this approach may not be the most useful if we are to understand the problem of maternal mortality as a whole and appreciate the importance of economical, political and social macrostructural factors. In this paper, we report the number of scientific studies published between 2000 and 2004 about the main causes of maternal death, as identified by WHO, and compare the proportion of papers on each cause with the corresponding burden of each cause. Secondly, we systematically review the characteristics and quality of the papers on the macrostructural determinants of maternal mortality. In view of their burden, obstructed labour, unsafe abortion and haemorrhage are proportionally underrepresented in the scientific literature. In our review, most studies analysed were cross-sectional, and were carried out by developed countries without the participation of researchers in the developing countries where maternal mortality was studied. The main macrostructural factors mentioned were socioeconomic variables. Overall, there is a lack of published information about the cultural and political determinants of maternal mortality. We believe that a high-quality scientific approach must be taken in studies of maternal mortality in order to obtain robust comparative data and that study design should be improved to allow causality between macrostructural determinants and maternal mortality to be shown.

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Maternal and Child Health Training of Traditional Birth Attendants and Pregnancy Outcomes: A Systematic Review and Meta-analysis

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• Rakhi Dwivedi 1   na1 ,
• Muhammad Aaqib Shamim 2   na1 ,
• Pradeep Dwivedi 1 , 2 ,
• Anannya Ray Banerjee 1 ,
• Akhil Dhanesh Goel 3 ,
• Varuna Vyas 4 ,
• Pratibha Singh 5 ,
• Shilpi Gupta Dixit 6 ,
• Kriti Mohan 7 &
• Kuldeep Singh 1 , 4  

1 Altmetric

Introduction

In remote communities, maternal and child health is often compromised due to limited access to healthcare. Simultaneously, these communities historically rely greatly on traditional birth attendants (TBAs). However, optimal integration of these traditional methods with modern healthcare practices remains a topic of debate. We assessed the effect of maternal and child health training of traditional birth attendants on adverse pregnancy outcomes.

We conducted a systematic review and meta-analysis to answer the above research question. We independently screened studies using databases like PubMed, Scopus, and CENTRAL, extracted data, and assessed the study quality. Due to fewer original studies in this field, we considered both pre-post and between-group differences to assess the effect of differences. These were synthesised separately, assessed against a p-value function, and subjected to sensitivity analyses.

We included six interventional studies. Training TBAs reduced the risk of perinatal mortality [0.69, 0.61–0.78] and 7-day neonatal mortality [0.65, 0.53–0.80] but not stillbirth [0.70, 0.39–1.26]. In randomized controlled trials, there is a lower risk of perinatal mortality [0.73, 0.67–0.79] and neonatal mortality [0.70, 0.62–0.80] but not stillbirth [0.81, 0.56–1.18] with trained traditional birth attendants. There are methodological concerns with most existing studies, including domains like allocation concealment.

There is some evidence of the benefit of training TBAs, though of a low to very low certainty. Due to fewer studies, inconsistent estimates for different critical outcomes, and concerns with the existing studies, further well-designed studies can give more insights. They can also help optimize the contents of TBA training interventions.

CRD42023412935 (PROSPERO).

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1 Introduction

Maternal health among tribals is a concern, as they often lack access to proper antenatal care, skilled birth attendants, and emergency obstetric services. This lack can lead to higher maternal mortality rates and complications during childbirth. Malnutrition and anaemia are also prevalent among tribal women, further exacerbating their health risks [ 1 ]. In the case of maternal and infant health care, these challenges can be particularly pronounced. The tribal population often resides in remote areas with limited access to healthcare facilities. This lack of accessibility can delay proper prenatal care, skilled birth attendance, and postnatal care [ 2 ]. Furthermore, cultural beliefs and practices within these communities may influence the utilization of healthcare services. Traditional methods and superstitions sometimes precede evidence-based medical practices, adversely affecting maternal and infant health outcomes [ 3 ]. In tribal communities, traditional birth attendants, often called Dais, are crucial in supporting women during pregnancy and childbirth. These skilled individuals have been serving their communities for generations, providing care and assistance to women during this transformative time [ 4 ]. During childbirth, a local traditional birth attendant assists the mother. They use their knowledge and experience to provide comfort measures, such as massage, positioning, and breathing techniques, to help ease the process of labour. They also ensure a clean and safe environment for delivery, using traditional practices and remedies to promote a smooth birth. In addition to their practical skills, traditional birth attendants also hold a significant cultural role within the community. They are respected members who carry the wisdom and traditions of their ancestors. Their presence during childbirth is seen to connect with the community's heritage and maintain cultural practices [ 5 , 6 ]. However, it is important to note that the role of traditional birth attendants is evolving in many tribal communities. The traditional birth attendant's role has faced challenges and changes with the introduction of modern healthcare systems and the emphasis on skilled birth attendance. Some communities have transitioned to utilizing formal healthcare services for childbirth, while others continue to value the traditional birth attendant's expertise [ 7 ]. Traditional birth attendants, or TBAs, have long played a role in providing care during childbirth in many communities worldwide. However, the recognition and training of TBAs have been a subject of debate and evolution in maternal and child health [ 8 ]. In recent years, there has been a shift in global health policy towards promoting skilled birth attendance by trained healthcare professionals, such as midwives and doctors. This shift has led to a decrease in the formal training and recognition of TBAs in some regions. However, it is important to note that TBAs still play a significant role in providing care, particularly in areas with limited access to skilled birth attendants. In some countries, efforts were made to integrate TBAs into the formal healthcare system through training programs [ 9 ]. These programs aim to enhance the knowledge and skills of TBAs, ensuring that they can provide safe and effective care during childbirth. The training typically covers topics such as maternal and child health towards hygiene, infection prevention, recognizing complications, and making timely referrals to skilled healthcare providers [ 10 , 11 ]. The training of TBAs varies across different regions and countries. Government health departments lead some training programs, while non-governmental organizations or community-based initiatives implement others. Training status for traditional birth attendants in maternal and child health care varies across regions and countries [ 12 ]. Efforts have been made to integrate TBAs into the formal healthcare system through training programs, but it is important to ensure that skilled birth attendance by trained healthcare professionals remains the primary focus for ensuring safe and effective childbirth care [ 13 ].

The requirement for an updated systematic review and meta-analysis (SRMA) on the impact of training Traditional Birth Attendants on adverse pregnancy outcomes stems from the importance of evidence-based practice in healthcare. As new studies are conducted and new evidence emerges, it is crucial to periodically review and synthesize the existing literature to ensure that our knowledge remains current and reliable.

In the case of training Traditional Birth Attendants, there has been ongoing interest in understanding the effectiveness of such interventions in improving pregnancy outcomes and reducing adverse events. By conducting an updated SRMA, researchers can systematically analyse the available evidence, including older and more recent studies, to assess the overall impact of training Traditional Birth Attendants on adverse pregnancy outcomes.

This process allows for a comprehensive evaluation of the benefits, limitations, and potential risks of training Traditional Birth Attendants. It helps to determine whether such interventions significantly reduce adverse events such as maternal and neonatal mortality, complications during childbirth, or other adverse outcomes. Furthermore, an updated SRMA can help identify gaps or inconsistencies in the existing literature, highlight areas requiring further research, and inform healthcare policies and practices. It provides a valuable tool for healthcare professionals, policymakers, and researchers to make informed decisions based on the most current and robust evidence.

2.1 Research Question and Selection Criteria

This SRMA answers the following research question: ‘What is the effect of training traditional birth attendants on adverse pregnancy outcomes?’ following the 2020 Preferred Reporting Items for Systematic Reviews and Meta-analyses(PRISMA) guidelines (Table S1 ). To answer this question, we systematically searched the literature and identified eligible articles based on the selection criteria defined using a PICO format (Table S2). We included all interventional studies assessing the effect of training traditional birth attendants on the risk ratios of all adverse pregnancy outcomes. For formal data synthesis, we only included the outcomes reported in two or more studies i.e., stillbirth, 7-day neonatal mortality, neonatal mortality, and perinatal mortality. We included both study designs—randomised controlled trials comparing training to no training, and pre-post single group interventional studies assessing training. Our primary outcome is risk ratio of stillbirth assessed before and after training. We registered the study at a publicly visible registry (PROSPERO), with the record ID CRD42023412935.

2.2 Systematic Search and Data Extraction

We systematically searched across three databases of published literature: PubMed, Scopus, and Cochrane Central Register of Controlled Trials, on 09.08.2023. RD prepared the search strategy for PubMed using MeSH terms, keywords, truncated terms and Boolean operators according to this extension to the PRISMA statement [ 14 ]. Another author(MAS) reviewed this as per the Peer Review of Electronic Search Strategies checklist [ 15 ]. Additionally, we included the grey literature via several preprint servers like Medrxiv, Biorxiv, Arxiv, and SSRN. A search in Google and Google Scholar supplemented this. We also reviewed clinical trial registries like ClinicalTrials.gov, International Clinical Trials Registry Platform(World Health Organisation), and Clinical Trials Registry–India. After this, we checked the references of eligible articles. Following this, we further searched by forward citation matching of the included articles. We sought the opinion of an expert in the field to identify other eligible articles. To achieve reproducibility, we have reported the search strategy across all three databases in Table S3.

After the systematic search and deduplication, two authors (RD & AR) reviewed the title and abstracts for eligibility. In case of any disagreement, the co-authors conversed to build consensus and decided on eligibility. A third independent reviewer's (KS) opinion was considered binding if conflict persisted. Potentially eligible studies went to the next step for full-text screening. Two authors (RD & AR) performed this. They followed a similar process with disagreements, and a third independent reviewer (PD) was the adjudicator.

The included reports were taken up for data extraction using a datasheet containing columns on bibliographic information, study characteristics, baseline data, information on intervention, control, outcome parameters, and other key details. Two authors (RD & AR) extracted data and followed a similar procedure. In case of conflict, they followed the opinion of an independent reviewer (PD).

2.3 Risk of Bias Assessment

Two independent authors (RD & AR) evaluated the quality of the selected studies for individual eligible outcomes. After answering signalling questions and assessing the risk of bias under five distinct domains, as recommended by the Cochrane Risk of Bias v2.0, the two assessors compared their findings [ 16 ]. In case of an inconsistency between them, they discussed it. If it persisted, they complied with the opinion of a third assessor (KS). This yielded an overall risk of bias for the outcomes in all these studies. The pre-post studies were assessed using an appropriate tool by National Institute of Health [ 17 ].

2.4 Statistical Analysis

The efficacy parameters are binary outcomes and are expressed as risk ratios. These were log-transformed and synthesised using the Mantel–Haenszel method [ 18 ]. These were then back-transformed in the visualisation for ease of interpretation.

For the assessment of heterogeneity, we used tau and tau-squared values. We used the restricted maximum likelihood estimator [ 19 , 20 ] for estimating tau-squared and the Q-profile method [ 21 ] for determining the confidence interval of tau-squared. In addition, we used the I-squared [ 22 ], Q-test [ 23 ], and prediction interval [ 24 ] for estimating heterogeneity. These were considered for the assessment of heterogeneity.

For synthesis, we employed a fixed-effects or a random-effects model based upon the between-study heterogeneity using a threshold of 50% for I-squared. Along with this, we also demonstrated the prediction interval for the true outcomes. Given the current evidence, this gives us the expected range for results from future studies, and is more important for real-world application by epidemiologists and clinicians. We computed the prediction interval based on a t-distribution [ 25 ]. We constructed it only for outcomes with more than two studies.

The pooled estimate has been expressed using forest and drapery plots. Forest plots are the conventional and more commonly interpreted method of summarising the results from a meta-analysis. We have also included the details of the outcome parameter and meta-analytical methods in the plots for comprehensiveness and quicker interpretation. Drapery plots complement and add to the inference drawn from a forest plot [ 26 ]. The limitation of the former is that they only display the result at a single fixed threshold for the level of significance, usually p < 0.05. We can avoid this controversial sole reliance on a single level [ 27 ] and instead use p-value functions. This helps us visualise the results across a wide range of confidence intervals corresponding to diverse p-values. We can visualise the results across a range of significance values. Moreover, the studies are presented in a logical sequence, and the overall trend can be easily observed, along with assessing small-study effects and heterogeneity. The prediction interval could be computed only for those outcomes where more than two studies were present.

We could not use formal tools like the funnel plot and the corresponding Egger’s regression to assess small-study effects and publication bias because of less studies (< 10). The sensitivity analysis omitted each study once for outcomes with more than two studies. We then assessed the change in the pooled estimate. Sensitivity analysis was done in the form of leave-one-out meta-analyses omitting each study one by one and assessing the robustness of the results. We explored the moderation of pooled estimate by a quantitative predictor by performing meta-regression, and visualised it using a bubble plot.

We considered a p-value of 0.05 significant for the primary analysis. Analyses were done using meta [ 28 ], and metafor [ 29 ] packages in R(v4.2.1) [ 30 ]. We assessed evidence certainty using the grading of recommendations, assessment, development, and evaluation (GRADE) framework [ 31 ].

2.5 Ethical Statement

Ethical review is not applicable here since this is an SRMA of publicly available data.

3.1 Study Selection

We can visualise the screening process via the PRISMA flowchart (Fig.  1 ). After a systematic search across three databases, following this search strategy (Table S3), we identified 1630 records. This study assessed the effect of training intervention on several professionals like physicians, nurses, TBAs, and others. TBAs only constituted 30%—40% of the study group, and the outcome data could not be retrieved separately for TBAs. So, we excluded this study [ 32 ]. This study assessed the effects of two different interventions using a factorial design. The effect of a community intervention with women’s groups has been studied here, and the effect of training the TBAs was planned to be released in a separate publication [ 33 ]. However, this could not be retrieved even after a detailed search and mailing the authors (see Figs. 2 , 3 and 4 ).

PRISMA flow chart detailing the literature search, and providing reasons for exclusion of studies

Forest plot showing the risk ratio of perinatal mortality with training of traditional birth attendants

Drapery plot showing the ‘level of significance’–dependent variation in the risk ratio of perinatal mortality with training of traditional birth attendants

Leave-one-out meta-analysis for the risk ratio of perinatal mortality with training of traditional birth attendants

We finally included six studies for systematic review and quantitative synthesis [ 34 , 35 , 36 , 37 , 38 , 39 ] [Table 1 ]. The studies were conducted in several countries in Asia, Africa, and South America. We have summarized the detailed information in Table S4.

3.2 Pooled Estimate

Training traditional birth attendants reduced the risk of perinatal mortality [RR: 0.69, 95% CI 0.61–0.78] post-training [Fig. 2 ]. The 95% prediction interval (95% PI) states the expected range into which the results of 95% of the future studies may lie, and is a more practical estimate. It is expected to give an effect size with the range of 0.32–1.50 in future studies.

Training traditional birth attendants also reduced the risk of 7-day neonatal mortality [RR: 0.65, 95% CI 0.53–0.80; 95% PI 0.08–5.81] post-training. However, the risk of stillbirth did not reduce significantly post-training [RR: 0.70, 95% CI 0.39–1.26; 95% PI 0.00–1036.89].

Trained traditional birth attendants compared to standard care showed a reduced risk of perinatal mortality [RR: 0.73, 95% CI 0.67–0.79] and neonatal mortality [RR: 0.70, 95% CI 0.62–0.80]. However, the risk of stillbirth [RR: 0.81, 95% CI 0.56–1.18] did not reduce significantly compared to usual care.

The drapery plots depict the individual study estimates and the pooled estimate alongside the shaded prediction interval [Fig. 3 ]. This is depicted against a range of significance thresholds. The thick blue lines shows that the risk reduction in neonatal mortality (after training), perinatal mortality (after training), and neonatal mortality (versus untrained) are all statistically significant even at a p-value as less as 0.01.

3.3 Heterogeneity and Small-Study Effects Assessment and Exploration

The pooled estimates had differing between-study heterogeneities as assessed by I 2 and prediction interval and depicted in the respective forest plots. Since there is high between-study heterogeneity for risk of stillbirth [I 2  = 84%, τ 2  = 0.24 (0.03–12.03), Cochran’s Q: p < 0.01], we explored the relation with sample size. This was done using meta-regression, and visualised as a bubble plot. However, it did not moderate the pooled risk ratio (beta = 0, p = 0.16). We could not conduct a formal test for publication bias or small-study effects because of the low number of studies. The detailed outcome assessment for individual studies and heterogeneity estimates for each outcome can be checked in the respective forest plots.

3.4 Sensitivity Analysis

We conducted sensitivity analyses using leave-one-out meta-analyses for the outcomes with more than two studies [Fig. 4 ]. The effect of omitting each study one-by-one is depicted in Figs. S9–S11.

3.5 Risk of Bias

Amongst the randomised controlled trials, two studies were at a high risk of bias, while one had some concerns. In this study [ 37 ], the randomisation for the latter part of the control group is not clearly given. In another study [ 35 ], there is no information on allocation concealment. The detailed domain-wise quality assessment is given in Fig. S13. Amongst the pre-post studies, the study quality varied from moderate to low risk of bias. The question-wise detailed assessment is given in Table S5. The evidence certainty is provided in Table S6, with most outcomes providing effects of a low to very low certainty.

4 Discussion

We assessed the effect of maternal and child health training on traditional birth attendants. There is a decrease in the risk of early neonatal mortality, neonatal mortality, and perinatal mortality. However, there is no difference in the risk of stillbirth. This change indirectly suggests that these training modules help mitigate the reduction in the outcomes following delivery i.e., neonatal mortality. Although stillbirth is not reduced, the summary estimate of perinatal mortality shows a decline because of the neonatal parameter.

Several studies have explored the impact of maternal and child health training on traditional birth attendants (TBAs) on reducing the risk of perinatal mortality. TBAs are locally available individuals who provide childbirth and maternal care in many communities, particularly in areas with limited access to formal healthcare facilities [ 37 ]. Similarly, our findings show that training traditional birth attendants reduced the risk of perinatal mortality [RR: 0.69, 95% CI 0.61–0.78]. Post-training traditional birth attendants (TBAs) have proven effective in reducing perinatal and 7-day neonatal mortality risk. However, it appears that the training did not significantly impact stillbirth rates. Therefore, there is a need for TBAs' knowledge and abilities to be developed within acceptable limits by ongoing collaboration between TBAs and health systems [ 40 ]. As governments progress towards providing SBA to all women, the possible role of TBAs should be carefully considered. Although the data has limitations due to the research design, TBA training can enhance some newborn outcomes but does not affect mother mortality [ 41 ]. In India, because access to public healthcare is limited, the tribal health system faces challenges. Tribal health challenges differ due to the tribal populations' distinctive socio-cultural practices, rituals, customs, and languages [ 42 ] .  Traditional birth attendants are very popular and highly influential in their tribal community. People prefer to take their help in maternal and child health. The TBA's psychological and socio-cultural support and community-wide access to mothers are believed to have contributed significantly to maternity and newborn health care. TBAs have an important role in improving maternal and newborn health, particularly in rural regions where they continue to be a vital force in delivering health and nutrition initiatives. One study shows that utilization of traditional birth attendant (TBA) services in a rural Local Government area with proper training of TBA can provide conventional and traditional maternity care that will translate into improved maternal and newborn health outcomes in relevant contexts [ 40 ]. The trained TBA adoption could positively influence the community members to handle and manage obstetric and neonatal complications [ 41 ].

There are several strengths to this manuscript. We have comprehensively covered a variety of evidence on the training of traditional birth attendants. We have followed it up with robust methodological and statistical steps to get accurate estimates. The limitation of this manuscript is tied down to the lack of enough well-designed interventional studies in this field. We have addressed this by covering several different research questions and synthesising them separately to get overall evidence. Training traditional birth attendants improved outcomes. However, there is a considerable scope for future research. There are very few studies, and further well-designed studies especially in the lower and middle- income countries can shed more light on the effectiveness of these training modules. The overall course content in these training packages can be further optimised to improve their effectiveness. As highlighted by the differential change in neonatal mortality and stillbirths, special attention should be paid to the neonatal components of training modules apart from the labour-related components. These can build an evidence base for good health and well-being with reduced inequality in the primary care for maternal and child health.

Data Sharing Statement

All data has been made available here and in the annexures.

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Acknowledgements

The authors are thankful to the research team of the Centre of Excellence for Tribal Health, AIIMS, Jodhpur. The authors are extremely thankful to Dr. Naval Jit Kapoor, Additional Secretary, and the entire team of the Ministry of Tribal Affairs, Government of India, for their constant guidance and financial support. The authors acknowledge the National Workshop on Systematic Review and Meta-Analysis conducted by the Department of Pharmacology and Centre of Excellence for Tribal Health, AIIMS, Jodhpur.

Author information

Rakhi Dwivedi and Muhammad Aaqib Shamim joint first authors and contributed equally.

Authors and Affiliations

Centre of Excellence for Tribal Health, All India Institute of Medical Sciences, Jodhpur, 342005, India

Rakhi Dwivedi, Pradeep Dwivedi, Anannya Ray Banerjee & Kuldeep Singh

Department of Pharmacology, All India Institute of Medical Sciences Jodhpur, Jodhpur, India

Muhammad Aaqib Shamim & Pradeep Dwivedi

Department of Community Medicine and Family Medicine, All India Institute of Medical Sciences, Jodhpur, 342005, India

Akhil Dhanesh Goel

Department of Pediatrics, All India Institute of Medical Sciences, Jodhpur, 342005, India

Varuna Vyas & Kuldeep Singh

Department of Obstetrics & Gynaecology, All India Institute of Medical Sciences, Jodhpur, 342005, India

Pratibha Singh

Department of Anatomy, All India Institute of Medical Sciences, Jodhpur, 342005, India

Shilpi Gupta Dixit

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Study design: RD, MAS, PD, AR, PS, VV, KS. Data collection: RD, AR, MAS, PD. Data analysis: RD, MAS, ADG. Figures and tables: RD, PD, AR, ADG. Data interpretation: ADG, PS, VV, KS, SGD, KM. Initial manuscript writing: RD, AR, PD, SGD, KM. Critical inputs and final revision of the manuscript: All the authors Availability of the entire study Availability of the entire study data: All the authors.

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Dwivedi, R., Shamim, M.A., Dwivedi, P. et al. Maternal and Child Health Training of Traditional Birth Attendants and Pregnancy Outcomes: A Systematic Review and Meta-analysis. J Epidemiol Glob Health (2024). https://doi.org/10.1007/s44197-024-00300-x

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A Systematic Policy Review of Black Maternal Health-Related Policies Proposed Federally and in Massachusetts: 2010–2020

Keri carvalho.

1 Department of Community Health, Tufts University, Medford, MA, United States

Anna Kheyfets

2 Department of Public Health & Community Medicine, Tufts University School of Medicine, Boston, MA, United States

Pegah Maleki

3 School of Social Policy & Practice, University of Pennsylvania, Philadelphia, PA, United States

Brenna Miller

Siwaar abouhala, eimaan anwar, ndidiamaka amutah-onukagha, associated data.

The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.

Background: Black women in the United States experience maternal mortality three to four times more often than white women ( 1 , 2 ). States vary in degree of disparity, partially due to programs and policies available to pregnant people. In Massachusetts, Black women were approximately twice as likely as white women to experience pregnancy-associated mortality, with a large percentage of these deaths reported to be preventable ( 3 ).

Methods: Using Massachusetts as a state-level comparison to national policies, we searched the US Congress and Massachusetts legislative databases for maternal health policies from 2010 to 2020. We screened 1,421 national and 360 Massachusetts bills, following set inclusion/exclusion criteria. Data analysis included (1) assessment of bill characteristics, (2) thematic analysis, and a (3) quality appraisal following an adapted model of the analytical framework for evaluating public health policy proposed by the National Collaborating Centre for Healthy Public Policy. Additionally, our data analysis identified the level of racism (internalized, interpersonal or institutional) that each policy addressed.

Results: From 2010 to 2020, 31 national and 16 state-level policies were proposed that address maternal health and racial disparities. The majority of policies addressed racism at the institutional level alone (National: N = 19, 61.3%, Massachusetts: N = 14, 87.5%). Two national and two Massachusetts-level policies became law, while two national policies passed only the House of Representatives. Our critical appraisal revealed that the majority of unintended effects would be neutral or positive, however, some potential negative unintended effects were identified. The appraisal also identified 54.8% ( n = 17) of national policies and 68.8% ( n = 11) of Massachusetts with positive impact on health equity.

Conclusions: There has been an increase in policies proposed addressing racial disparities and health equity in maternal health over the last 10 years. Although half of national policies proposed showed positive impact on health equity, shedding light on the work the U.S. is doing on a federal level to confront the Black maternal health crisis, only two policies made it to law, only one of which addressed racial disparities directly and had a positive impact on health equity.

Non-Hispanic Black women have historically been underrepresented in both the United States legislature and in legislation, and thus their maternal health needs and priorities have not been properly addressed or prioritized ( 4 ). Non-Hispanic Black women in the United States experience maternal mortality at a three-fold higher rate than non-Hispanic white women, hereafter referred to as Black and white, respectively ( 1 ). While pregnancy-associated mortality ratios are three-four times higher in Black women in comparison to white women, this disparity is further widened for specific mortality causes (e.g., ectopic pregnancy) ( 2 ). Across 13 state Maternal Mortality Review Committees, reports state 60% of pregnancy-associated deaths were preventable ( 5 ). States vary in degree with this disparity, partially due to varying programs and policies available to pregnant people. As of 2014 in Massachusetts, Black women were 1.9 times as likely as white women to experience pregnancy-associated mortality and 24% of these pregnancy-associated deaths between 2000 and 2007 in Massachusetts were determined to be preventable ( 3 ).

From 2011 to 2015 specifically, the majority of pregnancy-related deaths in the United States (35%) were caused by cardiovascular conditions, followed by 12.5% of deaths by infection, and 11.2% of deaths due to obstetric hemorrhages ( 5 ). However, the leading clinical causes of pregnancy-related deaths differ for Black women in comparison to white women. Black women experience more pregnancy-related deaths due to cardiomyopathy, thrombotic pulmonary embolism, and hypertensive disorders than their white counterparts ( 6 ).

Racial disparities in pregnancy-related deaths persist for Black women, regardless of seemingly protective factors. Black women with college degrees are more likely to die from pregnancy-related causes than pregnant white, Hispanic, and Asian/Pacific islander women without high school diplomas ( 7 ). Black women have a pregnancy-related mortality rate that is approximately 5.2 times that of white college-educated women ( 6 ). A study hypothesizing differences in maternal mortality across race/ethnicity evaluated whether risk factors could be related to differing medical insurances, but conclusively found no association of higher maternal mortality rates among Black and Hispanic women with differing insurance types ( 8 ). Alternatively, one factor exacerbating disparities could be increased likelihood for Black and Hispanic women to deliver babies at hospitals with poorer outcomes for maternal morbidity and preterm morbidity and mortality ( 9 ). Another factor could be quality resources and hospitals allocated by ZIP code, as adverse birth outcomes can be seen from hospitals located spatially close to or within racially segregated ZIP codes ( 10 ). However, the reality is that there is much unknown: during 2000–2007 in Massachusetts, the preventability of 33% of pregnancy-associated deaths were categorized as undetermined. There is a clear need to identify and subsequently address preventable factors in order to prevent maternal deaths, particularly for Black women, with one integral risk factor begging to be addressed: that of racism, directly leading to disproportionate Black maternal deaths.

The three levels of racism—internalized, interpersonal, and institutional— branch and intertwine across sectors and disciplines including public health and medicine ( 11 ). Internalized and interpersonal racism are defined as subconscious or conscious forms of discrimination that are driven by racial bias and that are reflected among relationships with others and one's self. Institutional racism, in contrast, encompasses the inequitable policies, attitudes, and organizations that are driven by power imbalances, and furthered by a lack of comprehensive and productive representation in all facets of society ( 11 , 12 ). Policymakers, public health researchers, healthcare providers, and associated stakeholders are not exempt from the perpetuation of institutionalized racism. Numerous studies and projects often report alarming rates of both implicit and explicit bias among health professionals ( 13 – 16 ). The presence of such discriminatory attitudes toward vulnerable populations in medicine and public health is not only a mere reflection of the institutionalized racism that exists in society, but an amplification of societal imbalances.

An effective means to make sustainable change and break through barriers of institutional racism is through legislative action. The intertwined factors that contribute to this disparity include the quality of prenatal and postpartum care and health-seeking behaviors and overall satisfaction with care ( 17 ). However, policies at the state and federal level have been presented, with some passed, to address the effects of institutional racism on Black maternal health. This includes a compilation of policies, collectively titled the Black Maternal Health Momnibus Act of 2020 1 ( 18 )). The overwhelming lack of legislation to reduce maternal mortality and severe morbidity in Black women must be addressed in order to reduce the poor maternal health outcomes women of color face to ultimately reduce pregnancy-associated deaths. The goal of the proposed study is to analyze the interventions highlighted in current policies and their potential health impacts on Black women. These policy analyses will help to determine the opportunities needed to increase equity to prenatal and postpartum care.

Previous researchers have conducted analyses comparing local and national policies related to maternal health in England ( 19 ). However, to our knowledge, there is a paucity of systematic reviews of maternal health-related policies conducted in the United States, including local and national policy comparisons. Because there are differences by state in health insurance regulations, Medicaid guidelines, and racial health disparities, it is particularly important to examine local policies compared to national policies in relation to Black maternal health.

We chose to focus on comparing maternal health policies on the national level and in Massachusetts as a case study. Although Massachusetts and national maternal mortality rates are the same (17.4 per 100,000 births), racial disparities in maternal mortality are significantly lower in Massachusetts than at the national level ( 3 ). Given the difference of maternal mortality racial disparities in Massachusetts compared to the nation, we aim to understand whether there are differences in policy geared toward Black maternal health that may contribute to this difference.

Research Question

This review investigated legislation proposed and passed to address Black maternal health in Massachusetts and at the national level from 2010 to 2020. The period examined was selected to capture recent legislative action, as racial disparities in maternal mortality have gained public and political attention.

Search Strategy

We conducted a systematic analysis of national and Massachusetts state-level proposed legislation and legislation passed between 2010 and 2020 addressing Black maternal health. Databases searched included the Massachusetts online legislative database and the United States Congress online legislative database. Key search terms included were maternal health, maternal mortality, maternal morbidity, perinatal, prenatal, and postpartum. Each term was searched independently on the databases, and duplicate results were removed from analysis.

Search Outcomes

The initial search resulted in 1,421 national bills and 360 Massachusetts state bills. Bills that did not pertain to maternal health and maternal health outcomes and duplicates were removed. Ninety national bills and 158 Massachusetts state level bills were included for in-depth full text review. Forty-five further national bills and 115 Massachusetts state level bills were excluded as they did not mention race, ethnicity, underserved communities, minority communities, health equity, or racial disparities. Prior to performing data extraction, 14 national and 28 Massachusetts state level bills were identified as duplicates or repeated drafts of bills and were excluded. We included the most recent version of each bill proposed, if multiple drafts were proposed. As shown in Figure 1 , 31 national bills and 16 Massachusetts state bills were included in the final sample for this review. Bills were independently reviewed by four researchers.

Flow chart of inclusion of bills in review. Panel (A) shows the national bills inclusion process and panel (B) shows the Massachusetts state-level bill inclusion process.

Inclusion/Exclusion Criteria

The eligibility criteria for this review included: ( 1 ) bills introduced, passed, or enacted by the U.S. Congress or Massachusetts Congress, ( 2 ) bill related to maternal health, ( 3 ) bills that explicitly referenced race, ethnicity, racial disparities, health equity, underserved communities, or minority communities, ( 4 ) bills introduced, passed or enacted into law between 2010 and 2020. We excluded sources that were not considered bills (e.g., reports, journal articles, amendments, etc.), and bills that did not include the designated key terms.

Data Extraction and Thematic Analysis

We developed a data extraction tool to document the following information from each bill: bill name, year of introduction, status in each respective congress, and content for thematic analysis and quality appraisal. We use qualitative methodology for data extraction to allow the most significant extraction of legislation content intricacies, for our thematic analysis, similar to methodology in a policy analysis of maternal health policies in Malawi ( 20 ). We used inductive qualitative analysis to determine themes of the content of the bills.

Bills were categorized into “category 1” including bills that mention terms regarding race, racial disparities, people of color, discrimination, health equity infrequently and discuss race on an epidemiological level and into “category 2” including bills that are predominantly concerning Black maternal health, racial disparities, and health equity. Additionally, our analysis of the factor of “health equity” is inclusive of which level a policy addresses racism (i.e., internalized, interpersonal, institutional) ( 11 ).

Thematic analysis for all of the bills was conducted in two rounds by independent reviewers to ensure accuracy. Interrater reliability was determined by agreement of categories addressed in the bill, and by matching quality appraisal (i.e., negative, neutral, positive). There was 91.2% agreement across reviewers. Conflicts were resolved by a senior reviewer.

Quality Appraisal

To complete the quality appraisal we followed an adapted model of the evidence-informed analytical framework for evaluating public health policy proposed by the National Collaborating Centre for Healthy Public Policy ( 21 ). This framework follows a two-pronged method of assessing three factors to determine effects of a policy, and three factors to assess a policy's implementation. For our analysis, we adapted the framework to focus on determining the first prong, or a policy's effects by assessing ( 1 ) effectiveness (how effective the policy is at achieving its named objective), ( 2 ) unintended effects (what positive or negative effects a policy may create unrelated to the objective), and ( 3 ) health equity (if a policy creates varying effects for varying populations, and if it maintains, increases or decreases inequities of health.) Guiding questions used in the data analysis are shown in Table 1 .

Guiding Questions from the adapted model of the evidence-informed analytical framework for evaluating public health policy by the National Collaborating Centre for Healthy Public Policy.

From 2010 to 2020, 31 national and 16 state-level policies were proposed that addressed maternal health and racial disparities and included in our analysis. Two national policies became law and two passed the House and two Massachusetts-level policies became law, while the majority of the policies included in the systematic analysis were only introduced in the respective congresses. On the national level, there has been an increase in proposed policies regarding maternal health and racial disparities in 2019–2020, while in Massachusetts there was less of a temporal trend.

The majority of policies addressed racism at the institutional level alone [National: 61.3% ( n = 19), Massachusetts: 87.5% ( n = 14)], while some also addressed interpersonal racism in addition to institutional racism [National: 38.7% ( n = 12), Massachusetts: 5.9% ( n = 1)]. The majority ( n = 18, 58.1%) of included national policies were categorized as “category 2,” meaning they were predominantly concerning Black maternal health, racial disparities, and health equity. However, the majority ( n = 14, 87.5%) of Massachusetts-level policies were categorized as “category 1,” meaning that the bills mention terms regarding race, racial disparities, people of color, discrimination, health equity infrequently and discuss race on an epidemiological level rather than being centered on racial and ethnic maternal health disparities. Detailed breakdown can be seen in Table 2 .

Characteristics and quality appraisal of included national and Massachusetts state-level bills.

In the category of “Scoring on Addressing Black Maternal Health,” a score of 1 indicates the bill mention terms regarding race, racial disparities, people of color, discrimination, health equity infrequently, a score of 2 indicates the bill directly concerns Black maternal health, racial disparities, and health equity .

Characteristics

From 2010 to 2020, US Congress Legislative handled 103 bills containing the key terms identified in this review. Of those 103 bills, 31 national policies met the inclusion criteria. After data extraction, the national policies selected varied in frequency during the chosen 10-year period. Of the 31 national bills, there was one in 2010, none in 2011, one in 2012, one in 2013, none in 2014, one in 2015, none in 2016, one in 2017, three in 2018, nine in 2019 and 14 in 2020. Out of the 31 national policies selected, 27 were introduced, two were Passed by the House and two national policies Became Law. The two national bills that passed were: Preventing Maternal Deaths Act of 2018 2 and PREEMIE Reauthorization Act of 2018 3 ( 22 , 23 ). There was also a distribution in the levels of racism addressed amongst the selected national bills. Out of the 31 national policies selected, no policy addressed internalized ( n = 0) or interpersonal racism only ( n = 0). The two levels of racism addressed were institutional only (61.3%; n = 19) and interpersonal and institutional (38.7%; n = 12). No bill addressed all three levels of racism. Eighteen bills were identified as “category 2,” predominantly focused on Black maternal health, racial disparities, or health equity. Thirteen bills were identified as “category 1”. Full details of included national bills can be seen in Table 3 .

Summary of quality appraisal, status, and category ranking of included national bills.

Thematic Findings

After data collection and distillation of national policies concerning maternal health from 2010 to 2020, 31 national policies were identified meeting the inclusion criteria. Thematic findings include 38 themes (seen in Table 4 ), with most frequent themes including: Expanding funding for maternal health research ( n = 16), Diversifying Healthcare Workforce/Committees ( n = 9), Increasing insurance coverage, training and inclusion of doulas, community birth workers, childbirth educators, lactation consultants and midwives ( n = 9), Expanding Medicaid coverage to 1 year postpartum ( n = 6) and Improving Medical Training for Maternal Health Workers ( n = 6).

Summary of themes in national and Massachusetts state-level bills included in the systematic review.

Five policies total included in this review were proposed prior to 2018. Three policies were proposed in 2018, 9 policies in 2019, and 14 policies in 2020. Three themes identified in policies were related to the current pandemic, including: Inclusion of Pregnant People in Vaccine Development for COVID19 ( n = 2), Funding for Research in Maternal Health & COVID19 ( n = 2), and Occupation Risk for Pregnant People & COVID-19 ( n = 2). Several themes were related to racial disparities and discrimination, but one theme was identified specific to Black women who give birth: Programs on education, civil rights, and maternal health for Black women and girls ( n = 1).

The themes of the two bills that became law included (1) expanding funding for research on maternal and infant health, (2) improving data collection, and (3) implementing and expanding state Maternal Mortality Review Committees (MMRCs). The themes included in the two bills that only passed the house included: (1) expanding Medicaid coverage to 1 year postpartum, (2) improving coverage of doula services, (3) implementing a bundled payment model, (4) expanding telemedicine, and (5) expanding funding for research on maternal health, specifically in rural communities.

In utilizing a modified version of the analytical framework for evaluating public health policy described in the Methods section, data was extracted from each policy related to effectiveness of the policy, unintended effects of the policy, and health equity . For each of these components, a value of positive, neutral, negative, neutral/positive or neutral/negative was assigned. For evaluation of effectiveness of policies, 19 were identified as having a positive effect (e.g., supporting community-level research or encouraging diversity in committee/ task force bodies), seven identified as neutral/positive (e.g., lacking specificity of insurance expansion, but increasing services generally), and five as having neutral effect (e.g., mentioning grant programs without specifically outlining steps/directives).

For the component of unintended effects, 13 were identified as having neutral unintended effects (e.g., adding various grant programs that may or may not be effective), 13 as positive unintended effects (e.g., insurance expansion & increased access to quality care long-term), and three as having potential negative unintended effects (e.g., policy attempts at increasing diversification of task force membership asking members to serve without additional pay). One policy was classified as neutral/positive unintended effect, and one policy classified as having a neutral/negative unintended effect.

For the component of health equity, 17 policies were identified as having positive impact on health equity (e.g., policy issuing a call for increased research and initiatives to address social determinants of health, and how they affect racial disparities), eight having neutral impact on health equity (e.g., policy briefly mentioning existence of racial and ethnic disparities, but not offering solutions to racial disparities in legislation), and none were identified as having a negative impact on health equity. Four policies were classified as having a neutral/positive impact on health equity, with one policy classified as having a neutral/negative impact on health equity.

Massachusetts

From 2010 to 2020, the Massachusetts State Legislature handled 158 bills containing the key terms identified for this review. Of those 158 bills, 16 met inclusion criteria. Massachusetts varied in the number of bills proposed per year across this 10-year period, seen in detail in Table 5 . During this 10-year period, two of those bills passed, while the other 14 remain with the status of being introduced. The bills that passed are (1) An Act Improving the Quality of Health Care and Reducing Costs through Increased Transparency, Efficiency and Innovation, which was signed by the Governor in 2012, and (2) An Act to Reduce Racial Inequities in Maternal Health 4 , which was signed by the Governor on January 13, 2021 ( 24 , 25 ). The distribution in the levels of racism addressed amongst the selected Massachusetts bills are as follows: out of the total 16, one bill addressed only interpersonal racism (5.88%), one bill addressed both interpersonal and institutional racism (5.88%) and the rest addressed only institutional racism ( n = 14, 87.5%). Two bills were identified as “category 2,” predominantly focused on Black maternal health, racial disparities, or health equity. Fourteen bills were identified as “category 1.”

Summary of quality appraisal, status, and category ranking of included Massachusetts state-level bills.

Sixteen Massachusetts policies were identified meeting the inclusion criteria. Of these, 14 policies were introduced, and two policies became law. Thematic findings include four themes, including: (1) Proposing Medicare for all (ex., mentions covering cost of maternity care/ family planning through perinatal), (2) Supporting midwife involvement on maternity care teams, and (3) Proposing Medicaid coverage for doula services (4) Formation of a committee to reduce maternal racial disparities. Of the two bills that became law the themes included: (1) Forming a committee to reduce maternal racial disparities and (2) expanding access to healthcare.

For evaluation of effectiveness of Massachusetts policies, 12 were identified as having a positive effect (e.g., comprehensively standardizing abortion access and sexual/reproductive health service access for vulnerable populations), two identified as neutral/positive (e.g., legislation containing a myriad of non-maternal health parts, but additionally containing some language around expanding provider accessibility during pregnancy), and two as having neutral effect (e.g., actual implementation of policy impacted by health care context/culture.) For the component of unintended effects, five were identified as having unintended potential negative effects (e.g., allowance of disenrolled providers to continue being able to treat pregnant individuals, potentially allowing providers disenrolled for harmful reasons to work with patients), seven as positive unintended effects (e.g., broad language around maternity and fertility care allowing ability to insert abortion care and access underneath umbrella of coverage), and four as having neutral unintended effects (e.g., attempt to incentivize physicians to leave private practice in favor of CHCs.) For the component of health equity, 11 policies were identified as having positive impact on health equity (e.g., correcting social inequities through increasing representation in legislation), three having neutral impact on health equity (e.g., simple reference to culturally competent providers), and two as having a neutral positive impact on health equity (e.g., containing language noting performance benchmarks for hospitals shall include reduction of racial and ethnic disparities without clarifying disparities and consequences to hospitals that don't abide.) None were identified as having a negative or neutral negative impact on health equity.

This systematic policy review investigated legislation proposed and passed to address Black maternal health in Massachusetts and at the national level from 2010 to 2020. Specifically, we analyzed and compared policies between Massachusetts and the federal government regarding health equity in maternal care and improving Black maternal health to determine what could be implemented on a national level that Massachusetts has proposed. Overall, we found that there is a lack of legislation that would address maternal racial disparities that is passed at both the federal level and in Massachusetts. At the federal level, only two bills were passed out of 31 proposed, and in Massachusetts, only two bills were passed of the 16 proposed. Even still, the bills that have passed (National: PREEMIE Reauthorization Act of 2018 and Preventing Maternal Deaths Act of 2018, Massachusetts: An Act to Reduce Racial Inequities in Maternal Health and An Act Improving the Quality of Health Care and Reducing Costs through Increased Transparency, Efficiency and Innovation 5 ) represent only initial movement toward improving maternal outcomes over time ( 22 – 25 ).

The bills that became law on the national levels sought to address Black maternal health by identifying disparities through improved data collection, including the establishment of maternal mortality review committees (MMRC) across the country and a standardized form, the Maternal Mortality Review Information Application (MMRIA 6 ), through the Centers of Disease Control and Prevention (CDC)'s “Review to Action” program ( Maternal Mortality Review Information Application, MMRIA ). This seeks to standardize the process of reporting and analyzing maternal death across the country to determine preventability, factors that contribute to the deaths, and what areas in particular need to be addressed in a more holistic manner, through the interprofessional MMRC team. Another aspect of the bills that have become law nationally is to allocate funding for research on preterm birth. Expanding funding for research on the topic has been a predominant theme amongst the included national bills.

Additionally, two bills nationally passed the House of Representatives but have not been voted upon in the Senate: The Maternal Health Quality Improvement Act of 2020 7 and The Helping MOMS Act of 2020 8 ( 26 , 27 ). While it is unclear whether these two bills will become law, they address several focuses that have been commonly proposed in other bills that remained stagnant in the House of Representative or Senate.

In Massachusetts, one bill that became law, An Act to Reduce Racial Inequities in Maternal Health, established a diverse commission specifically to examine and make recommendations to reduce racial inequities in maternal health ( 25 ). The other bill that passed into law, An Act Improving the Quality of Health Care and Reducing Costs through Increased Transparency, Efficiency and Innovation, aimed to improve the quality of healthcare and reduce healthcare costs through increased transparency, efficiency, and innovation within the healthcare system ( 24 ). Lastly, the bill that passed the House but has not been passed in the Senate, An Act Establishing Improved Medicare For All in Massachusetts, 9 would enact Medicare for all residents in the state and establish a statewide healthcare trust to disburse funds for medical treatment ( 28 ).

Two of the most salient themes noted across policies were similar at the federal level and in Massachusetts. First, bills identified the need for increased roles and support for doulas and midwives. Although these bills did not directly address Black maternal health, heightening the availability, compensation, and services that doulas and midwives can offer will likely result in healthier outcomes for Black mothers. Previous research demonstrated that Black women experience lower risk for cesarean birth and intrapartum analgesia with the presence of a doula who can provide continual emotional and relational support through birth ( 29 ). Given the substantial association between doula services and labor and delivery outcomes, some states such as Oregon and Minnesota have already expanded Medicaid to support doula services ( 30 ). However, this is yet to be supported on a national level or in Massachusetts. These bills also propose Medicaid expansion for other birth and breastfeeding support roles, such as childbirth educators and lactation consultants. Studies demonstrate that attending childbirth classes reduce labor interventions and risk for cesarean birth ( 31 ). Access to lactation consultants and lactation education has been shown to increase initiation of breastfeeding and likelihood of exclusive breastfeeding, which improves health outcomes such as disease burden for both mother and child ( 32 , 33 ). The bills that were included under this theme addressed two aspects of expanding the roles of doulas, midwives, lactation consultants, and community health workers. The bills both called for greater inclusion of these professions into the birth setting, policy development, MMRCs, and research while also proposing increased access to these professions by expanding insurance coverage, including Medicaid, to cover their services and establishing community-based training and recruitment efforts to increase the workforce numbers and diversity.

Another similar theme for bills at the federal level and in Massachusetts was diversification of committees to address maternal health disparities. Diversifying committees may lead to greater health equity by providing a platform for communities of color to have representation in discussions of maternal health. Federal level bills also proposed further diversifying the healthcare workforce. Studies have shown that a shared racial identity between Black mothers and providers may halve the mortality rate of Black infants ( 34 ).

One other core theme that emerged at the federal level was funding for research expansion. With the recent recognition of Black maternal health as a public health crisis, there may be a temporary surge in funding to investigate racial disparities in healthcare settings. However, for the multipronged societal intervention required to alter the underlying institutional racism that has resulted in maternal racial disparities, long-term funding will be required across many different settings and areas of study. This will include funding to create effective community-level interventions for social determinants of health, such as housing, employment, and other environmental barriers to health equity.

In Massachusetts, there was particular emphasis to enact Medicare for all, which is a single payer health care financing system for all residents. This includes coverage for prenatal, pregnancy, and postpartum services, and would allow for care beyond the current coverage of 60 days postpartum through Medicaid for women who meet poverty level financial thresholds. Medicaid prioritizes coverage for pregnant women and finances over 4 in 10 births in the United States ( 35 ). However, nearly one-third of maternal mortalities occur between 1 week and 1 year postpartum, the majority of whom are Black women ( 5 ). This therefore suggests the need for expansion of Medicaid coverage to 1 year postpartum ( 36 ). States can apply for a waiver to extend postpartum coverage, but only the federal government can alter the length of postpartum coverage. Recently, the Helping MOMS Act of 2020, a bill that proposes lengthening postpartum coverage to 1 year, has passed the House of Representatives and is one of the most common themes identified in 19.4% of bills at the federal level, but has yet to be enacted as law (citepbib700).

Massachusetts vs. National (Quality Appraisal)

Across analyses, both federal and Massachusetts level legislature were determined to have a neutral or positive effect, with most also having positive or neutral unintended effects and a positive impact on health equity. However, it is important to note that because these analyses included legislation that specifically addressed terms related to race and health equity, there may be a difference in unintended effects and health equity between bills included here and others that only mention race in an epidemiological sense. For instance, previous research demonstrates that state-level policies that impact clinic closures and impose restrictions on gestational age for abortion increase mortality rates ( 37 ).

Institutional and/or interpersonal racism were most commonly addressed across federal and Massachusetts policies. Federal policies may be particularly effective in addressing issues of health equity and racism, as federal policies provide a minimum standard to which all states must adhere. For example, if Medicaid were extended to 1 year postpartum at the federal level, racial disparities in maternal mortality may decrease across the population ( 36 ). Although these analyses focus particularly on legislation related to maternal racial disparities, it is essential to also recognize that policies that relate to other aspects of institutional racism (i.e., housing, employment, education) also have the potential to make significant improvement in the health of Black women.

Gaps Identified

At both the federal level and in Massachusetts, there are still many gaps to be addressed. In particular, postpartum depression within Black, Indigenous and People of Color (BIPOC) populations has yet to be identified as a focus within policy. However, studies suggest that there are great disparities between Black and white women in both diagnosis and treatment for postpartum depression ( 38 ). Black women are less likely than their white counterparts both to attend postpartum appointments that might result in diagnosis of postpartum depression and to attend follow-up appointments to treat postpartum depressive symptoms ( 39 ). Researchers speculate that many factors could be contributing to this outcome, including fear of racial discrimination from healthcare providers, mental health stigmas, and issues of access and practicality related to attending another appointment ( 40 , 41 ). Policies are needed to ensure equitable opportunity and access to diagnostics and follow-up treatment for Black women.

Another important gap to address is funding for hospitals with poor maternal health outcomes. This is especially important in hospitals that serve communities of color and low-income communities. One study showed that location of delivery accounted for 47.7% of racial disparities in severe maternal morbidity rates between Black and white mothers, with Black mothers being more likely to deliver at high-risk hospitals for severe maternal morbidity ( 42 ). By implementing policies that allow for additional funding and resources in hospitals that primarily serve communities of color, we may begin to create more equitable systems for Black women and improve maternal outcomes.

Importantly, most policies aim to address the symptoms of institutional racism rather than the root causes. For instance, institutional racism has made it such that healthcare workers in decision-making roles are predominantly white males ( 43 ). This in turn has resulted in several bills calling for diversity within the healthcare workforce at the national level. Certainly, diversity of healthcare providers may begin to improve patient-provider relationships for Black women, but it does not heal the deep wound and impact of institutional racism entirely. A single bill does not address the inherent racial bias woven into every part of the fabric of the healthcare system and American society that has ultimately manifested in the preventable deaths of Black mothers. Rather, it is clear that many of the symptoms of institutional racism will need to be addressed at the policy level in order to begin to alter the course of action required to reverse trends in maternal racial disparities.

Conclusions

In this systematic review, we compare state-level legislation in Massachusetts to national level legislation addressing maternal health and racial disparities between 2010 and 2020. After accounting for all inclusion criteria, we found that only 31 national and 16 state-level bills were proposed. Of the proposed bills, two federal and two state-level bills were enacted into law. Although there is initial movement toward increasing bills that can focus on and address maternal health disparities, tremendous progress still remains to be seen at both the state and federal level to improve outcomes for Black women.

Data Availability Statement

Author contributions.

The study idea was formulated and written by KC, AK, PM, BM, SA, and EA. Initial analyses were performed by PM, BM, SA, and EA. Final analyses were performed and reported by KC and AK. Final writing and preparation of the manuscript, including edits, were done by KC, AK, and NA-O. All authors contributed to the article and approved the submitted version.

Conflict of Interest

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

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1 Black Maternal Health Momnibus Act of 2020, no. H.R.6142, 116th Congress (2020).

2 Preventing Maternal Deaths Act of 2018, H.R.1318, 115th Congress (2017-2018), Public Law No: 115-344 (2018). Available online at: https://www.congress.gov/bill/115th-congress/house-bill/1318/text .

3 PREEMIE Reauthorization Act of 2018, S.3029, 115th Congress (2017-2018), Law No: 115-328 (2018). https://www.congress.gov/bill/115th-congress/senate-bill/3029 .

4 An Act to Reduce Racial Inequities in Maternal Health, no. H.4818, 191st Congress of MA (2019-2020) (2021). Available online at: https://malegislature.gov/Bills/191/H4818 .

5 An Act Improving the Quality of Health Care and Reducing Costs through Increased Transparency, Efficiency and Innovation, no. S. 2400, 187th Congress of Ma (2011-2012) (2012). Available online at: https://malegislature.gov/Bills/187/S2400 .

6 Maternal Mortality Review Information Application (MMRIA). Review to Action . Available online at: http://www.mmria.org/ .

7 Maternal Health Quality Improvement Act of 2020, no. H.R. 4995, 116th Congress (2019-2020) (2020). Available online at: https://www.congress.gov/bill/116th-congress/house-bill/4995/text .

8 Helping MOMS Act of 2020, no. H.R. 4996, 116th Congress (2019-2020) (2020). Available online at: https://www.congress.gov/bill/116th-congress/house-bill/4996 .

9 An Act Establishing Improved Medicare For All in Massachusetts, no. H. 2987, 190th Congress of MA (2017-2018) (2018). Available online at: https://malegislature.gov/Bills/190/h2987 .

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The CDC says maternal mortality rates in the U.S. got better, after a pandemic spike

Selena Simmons-Duffin

After an alarming spike in 2021, maternal mortality numbers the next year went back down, according to a report released Thursday. CDC Director Mandy Cohen says the rates are still too high. Rich Legg/Getty Images hide caption

After an alarming spike in 2021, maternal mortality numbers the next year went back down, according to a report released Thursday. CDC Director Mandy Cohen says the rates are still too high.

After spiking in 2021, the maternal mortality rate in the U.S. improved significantly the following year, according to a new report from the Centers for Disease Control and Prevention.

The data shows that 817 women died of maternal causes in the U.S. in 2022, compared to 1,205 in 2021. These are deaths that take place during pregnancy or within 42 days following delivery, according to the World Health Organization , "from any cause related to or aggravated by the pregnancy or its management, but not from accidental or incidental causes."

"I think that the bump [in 2021] reflects the pandemic and we're returning to pre-pandemic levels," says study author Donna Hoyert, who a health scientist at the CDC's National Center for Health Statistics.

The maternal mortality rate in 2022 was 22.3 deaths per 100,000 live births. That's a significant decrease from the 2021 rate of 32.9, but it's still much higher than the rate in other wealthy countries.

There continue to be enormous racial disparities in the U.S. maternal mortality rate as well – the rate for Black women was 49.5 deaths per 100,000 births in 2022, compared to a rate of 19 deaths for white women. Research shows the vast majority of these deaths are preventable .

Dr. Veronica Gillispie-Bell is an OB-GYN in New Orleans who was not involved in the CDC report. She agrees that COVID-19 was likely the reason for the major spike in maternal mortality.

"I really think that 2021 was actually an outlier because of the circumstances," Gillispie-Bell says. "We know that because of COVID-19, there were disruptions to care that obviously impacted our ability to care for pregnant individuals, plus there were pregnant individuals who were dying from COVID." It's hard to know for certain since the CDC report did not include cause of death, she adds.

She's encouraged that the 2022 numbers are slightly lower than 2020 – 817 in 2022 versus 861 in 2020. "It could mean that we're moving in the right direction – I think we need more years of data to know," she says.

CDC's newest data comes several weeks after an academic study cast doubt on the agency's methodology, suggesting that a pregnancy checkbox on death certificates was causing the numbers to be much higher than they are in reality. CDC strongly rejected the study's findings.

Hoyert also defends CDC's methodology. "There was plenty of literature before we made the changes that we were underestimating [maternal deaths] without a checkbox, and so we did add the checkbox," she says, explaining that they have continued to do evaluations and issue guidance to ensure it's being used correctly.

"I think CDC is doing great work in collecting the data and sharing that back," CDC Director Mandy Cohen told NPR last month. "We disagree with how that study was looking at it, and think it's unacceptable for moms to be dying at that rate here in the United States."

The stakes for getting these numbers right are high in a post- Roe America. Reproductive health advocates warn that abortion bans threaten women's lives, and if CDC's data is not viewed as reliable by the public, that could make it hard to evaluate the impact of these restrictions.

In a statement about CDC's latest report, Dr. Verda Hicks, president of the American College of Obstetricians and Gynecologists, connected the maternal mortality figures to "the worsening state of reproductive health care since the Dobbs decision."

"When treating pregnancy complications, abortion care can be lifesaving, and withholding that care unquestionably compromises patient lives and outcomes," Hicks wrote.

Despite the challenges with the data, Dr. Gillispie-Bell says the public should still put a "great bit of stock" into CDC's analysis. She also pointed to the work of state maternal mortality review committees around the country – she is the medical director of the committee in Louisiana. They are supported and funded by CDC.

"The first step for our maternal mortality review committee – once we get the death certificate with that pregnancy checkbox – is to then start extracting data to confirm ... so our numbers are very accurate," she says.

Not all states have these committees validating maternal deaths and making recommendations to reduce their numbers. CDC Director Cohen pointed out the agency now has funding available for each state. She also pointed out that CDC's data has already led to policy changes to reduce maternal deaths, including allowing Medicaid coverage to continue for a year postpartum .

"I think we're making strides, which is great," Cohen added. "We have more work to do."

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Report of the ninth meeting of the WHO Strategic and Technical Advisory Group of Experts for Maternal, Newborn, Child and Adolescent Health and Nutrition, 12–14 May 2024

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• Published: 12 September 2024

Predictors of mortality and severe illness from Escherichia coli sepsis in neonates

• Adriana Hoffman 1   na1 ,
• Sriram Satyavolu 2   na1 ,
• Danah Muhanna 3 ,
• Sindhoosha Malay 1 ,
• Thomas Raffay 1 ,
• Anne Windau 4 ,
• Eric M. Ransom 4 , 5 &
• Devashis Mukherjee   ORCID: orcid.org/0000-0002-6812-8970 1  

Journal of Perinatology ( 2024 ) Cite this article

Metrics details

• Bacterial infection
• Risk factors

Neonatal Escherichia coli ( E. coli ) sepsis is increasing. There is limited data on the factors contributing to increased mortality and severity of illness in neonatal E. coli sepsis. A retrospective review of neonates (<30 days) admitted to a Level IV NICU in the United States from 2008 to 2022 diagnosed with E. coli bloodstream or cerebrospinal fluid infection was conducted. Primary outcome was defined as mortality from or severe illness during E. coli infection (defined as a need for inotropic support or metabolic acidosis). E. coli neonatal sepsis rate increased from 2008 to 2022 (average of 1.12 per 1000 live births). The primary outcome, which occurred in 57.4% of cases, was independently associated with prematurity, neutropenia, and thrombocytopenia. Ampicillin resistance was not associated with the primary outcome. GA, neutropenia, and thrombocytopenia but not ampicillin resistance, are associated with mortality or severe illness from E. coli sepsis.

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Neonatal bacterial infections have significant morbidity and mortality, with an estimated annual 6.3 million cases and 230,000 deaths globally [ 1 ]. In the United States, the incidence of neonatal bacterial sepsis varies between one and four per 1000 live births (LBs) [ 2 ]. Neonatal sepsis is categorized into early-onset (EOS, infants ≤72 h old) and late-onset sepsis (LOS, infants >72 h). EOS classically represents maternal-to-fetal transmission, while most LOS are nosocomial or community-acquired [ 3 ]. As antenatal Group B Streptococcus (GBS) screening and intrapartum antibiotic prophylaxis (IAP) have led to significantly decreased GBS EOS, Escherichia coli ( E. coli ) is now one of the predominant organisms responsible for EOS [ 4 , 5 , 6 , 7 ]. Within the National Institute of Child Health and Development (NICHD) Neonatal Research Network (NRN) neonatal intensive care units (NICUs), 36.6% of EOS cases are due to E. coli (0.4 per 1000 live births), compared to 30.2% from GBS [ 4 ]. The Centers for Disease Control and Prevention (CDC) Active Bacterial Core surveillance data from 2005 to 2014 reported that 24.8% of EOS (0.2 per 1000 live births) in the US were E. coli infections [ 8 ]. The E. coli fraction of EOS cases increases to 58% (NRN) and 44% (CDC) for very low birth weight (VLBW, <1500 g BW) infants. E. coli is also the leading Gram-negative organism in LOS (5–12%) in preterm infants in the US [ 6 , 9 , 10 ]. E. coli sepsis in neonates is associated with substantial mortality, which is influenced by the gestational age (GA), birth weight (BW), and immune status of the neonate [ 11 , 12 , 13 ].

Although the evaluation of suspected EOS has changed over time, the first-line drugs still remain ampicillin and gentamicin [ 14 ]. There has been growing concern regarding the emergence of multidrug-resistant E. coli strains and reports of increasing resistance to ampicillin and gentamicin [ 15 , 16 , 17 ]. The rates of ampicillin and gentamicin resistance in E. coli isolates from US NICUs were between 66–80% and 8–17%, respectively [ 4 , 18 ]. The use of ineffective empiric antibiotics due to resistance results in inadequate infection control and disease progression to severe illness or death

There are inconsistent reports of an association between the risk of death among infants with E. coli EOS and ampicillin resistance [ 19 ]. It is critical to identify the risk factors associated with mortality and severe illness from E. coli sepsis so that antibiotic stewardship programs and aggressive antibiotic regimens for neonates at risk can coexist in the NICU. We determined specific risk factors associated with mortality or severe illness in E. coli neonatal sepsis.

We conducted a retrospective review of all neonates ≤30 days old with E. coli positive blood or cerebrospinal fluid (CSF) culture admitted to Rainbow Babies and Children’s Hospital, a Level IV NICU in Cleveland, Ohio, from 01/01/2008 through 12/31/2022. This study was approved by the Institutional Review Board, and informed consent was waived due to its retrospective nature. Information was collected on infant and maternal clinical and demographic variables. Infants with congenital anomalies and conditions that would predispose to infection, such as immunodeficiency syndromes, were excluded from statistical analysis. Microbiological information included hours of life at culture collection, source (blood vs CSF), time to positivity, and antibiotic resistance profile. Infant clinical characteristics included outcome (discharged home vs death due to E. coli infection), complete blood counts (CBC) during sepsis, duration of antibiotic therapy, presence of metabolic acidosis (pH <7.20 or base deficit >10 mEq/L without respiratory acidosis) and need for inotropic medication during the E. coli sepsis episode. Maternal characteristics included presence of intra-amniotic infection (IAI, defined as maternal peripartum fever 38–38.9 °C with signs of purulent cervical drainage, maternal leukocytosis, or fetal tachycardia, or isolated maternal fever ≥39 °C), E. coli infection in the mother at any time during pregnancy, and maternal intrapartum antibiotic therapy.

We defined our primary outcome as either death or severe illness from E. coli infection. Death was attributed to E. coli infection if it happened at any point from collection of the blood or CSF culture till the end of antibiotic treatment for that particular episode of infection. Severe illness was defined as the new need for inotropic medication or new development of metabolic acidosis, as described above, during the episode of E. coli infection. We specifically included only those infants in our primary outcome who had a new need for inotropic medication and had worsening or new metabolic acidosis at the time of blood or CSF culture at any time during the treatment of the infection as these clinical findings can exist in preterm neonates for reasons other than bacterial sepsis. The time of detection of sepsis, which was used to differentiate between EOS and LOS, was based on the time of collection of blood or CSF culture relative to the time of birth.

Statistical analysis

Charts that met inclusion and exclusion criteria were checked for completeness and consistency and coded into Rstudio Version 2023.06.1+524. Descriptive data were obtained, and summary statistics were created. These summary tables included the associated p -value stratified for the variable of interest. The primary outcome variable was death or severe illness, as defined above. For all analyses, this composite outcome was used for descriptive statistics and logistic regression modeling. Two-way ANOVA was used to evaluate the interaction and main effects of two factors on the dataset. The analysis included multiple comparisons to assess differences between both columns and rows. Bivariate analysis was performed to identify co-linearity between variables of interest. Those variables that were strongly associated ( p  < 0.05) with the outcome variable and relevant to our study were used. Multivariable logistic regression was performed using a backward stepwise method to identify key variables to improve the model’s overall fit to the dataset. The model fitness was assessed using the goodness of fit test to estimate if the model fits the data appropriately. Odds ratios were identified and represented in an odds ratio plot with a 95% confidence interval. This odds ratio plot was used to identify and assess the direction of association between the predictor variables and the outcome variable. P -values were reported for each of the given variables in the multivariate logistic regression model. The generalized variance inflation factor (GVIF) was used to identify co-linearity between predictor variables numerically. Those variables with GVIF greater than five were excluded from the model despite these parameters adding significance to the model.

Demographics and incidence of E. coli sepsis (Table  1 )

In the 59,984 live-born infants from 2008 to 2022, there were 68 unique cases of monomicrobial E. coli bloodstream or CSF infection in neonates under 30 days old (1.12 cases per 1000 LBs). E. coli sepsis incidence per 1000 LBs has steadily increased at our center from 0.35 (7/19,716) in 2008–2012 to 1.03 (21/20 331) in 2013–2017 and to 2.01 (40/19 932) in the last five years (2018–2022) respectively. The incidence of E. coli sepsis in VLBW infants was 12.4 per 1000 LBs. There were 38 cases of EOS and 30 cases of LOS. Median GA and BW of infants with E. coli sepsis were 31 2/7 wks (27 4/7 – 38 0/7 wks) and 1620 g (970–2835 g) respectively. There were three cases of E. coli meningitis; two grew E. coli from their blood cultures simultaneously. The median age of diagnosis of E. coli sepsis was 31.5 h (1.9–173.8 h).

Maternal IAI

Twenty-one infants (30.8%) were born to mothers with a documented IAI. EOS was significantly higher in these infants vs. LOS ( p  < 0.001). Only two E. coli LOS cases (without a previous EOS) occurred in infants born to mothers with IAI. There were no differences between rates of mortality, severity of illness, or the primary outcome between infants who were born to mothers with IAI vs those who were not.

Early and late-onset E. coli sepsis

There were 38 EOS and 30 LOS cases during the 15-year study period (Table  3 ). Males were more likely to have LOS than females, who were more likely to have EOS. The median GA or BW was not different between EOS and LOS infants. Although more VLBW infants were in the EOS group than the LOS group (55% vs 33%, p  = 0.071), this did not reach statistical significance. There was no difference between ampicillin or gentamicin resistance between EOS and LOS E. coli isolates. Mortality was not different between EOS and LOS cases, and neither was a need for inotropic medication or severe metabolic acidosis.

Primary outcome of mortality or severe illness from E. coli sepsis

Thirty-nine (57.4%) infants out of the 68 diagnosed with E. coli sepsis met the primary outcome (Table  2 ). 24/39 (61.5%) of these infants had a BW < 1500 g (VLBW). Out of all VLBW infants diagnosed with E. coli sepsis, 77.4% experienced death or severe illness in comparison to 40.5% in infants with BW ≥ 1500 g who were diagnosed with E. coli sepsis (OR 5.029, 95% CI 1.729–14.624, p  = 0.003). The median age (IQR) at which E. coli infection was diagnosed for infants with the primary outcome was 37 h (2–168), with 22/39 (56.4%) of these infants having EOS. Twenty-nine of the 39 infants (74.4%) had ampicillin-resistant E. coli , which was not statistically different from the degree of ampicillin-resistance in the entire cohort (69.1%), or in the infants who survived to discharge without severe E. coli sepsis (62.1%, p  = 0.28). Apart from GA (and its surrogate marker, BW), the lowest platelet count and the lowest ANC during the episode of sepsis were the only other variables significantly different between those infants who died or had severe illness from E. coli sepsis vs those who did not in our univariate analysis (Table  3 ). In addition, infants who died or had a severe illness were also more likely to be thrombocytopenic or neutropenic during the episode of sepsis. There was no association between the primary outcome and sex, race, ampicillin or gentamicin resistance, mode of delivery, or maternal IAI. None of the three cases of E. coli meningitis died or had severe illness. The median antibiotic use duration for E. coli sepsis in those infants who survived (with or without severe illness) was 21 days.

In our multivariate logistic regression model, the combination of GA < 37 weeks, neutropenia, and thrombocytopenia had four times greater odds of an infant experiencing mortality or severe illness related to E. coli sepsis. All three of these variables were individually associated with an increased risk of mortality or severe illness (Fig.  1 ). GA and BW were co-linear variables, so BW was not included in the multivariate model. Note that sex was used as a variable to improve the overall degrees of freedom, which increased the r-squared statistic.

OR for platelet count <150,000/µL = 1.95 (1.68–3.46), GA < 37 weeks = 9.11 (4.23–10.75), ANC < 1500/µL = 1.21 (1.13–3.22). GA gestational age.

Platelet counts and absolute neutrophil counts (ANCs)

Platelet counts and ANCs were available on 64/68 infants with E. coli sepsis, out of which 26 infants were thrombocytopenic (<150,000/µL). The lowest platelet count and ANC at any point of time during the entire duration of the sepsis episode was used for analysis. Those who died or had a severe illness were significantly more likely to be thrombocytopenic as compared to those who did not (64% vs. 14%, p  < 0.0001), with a lower median platelet count during the episode of sepsis (97 (70–164) vs. 262 (213–294), p  < 0.0001, values in 1000/µL).

Those who died or had a severe illness were also significantly more likely to be neutropenic (<1500/µL) as compared to those who did not (33% vs. 14%, p  = 0.04). They also had a lower median (IQR) ANC during the episode of sepsis (2.9 (0.9–5.9) vs. 4.0 (2.4–6.2), p  = 0.8, values in 1000/µL), although this did not reach statistical significance.

Empiric antibiotic resistance

Antibiotic susceptibility data were available for 67 of the 68 E. coli isolates. Ampicillin resistance was 70% overall and similar when including only VLBW infants. Twenty-eight out of 38 (74%) E. coli EOS isolates and 19/29 (66%) LOS isolates were ampicillin-resistant. Three out of 38 (8%) E. coli EOS cases and 2/29 (7%) LOS cases were gentamicin resistant. Ampicillin and gentamicin resistance did not vary over time. There was no significant difference in our primary outcome between ampicillin-resistant and susceptible cases (29/47, 61.7% vs 10/20, 50.0%, p  = 0.37). Ampicillin resistance did not differ between VLBW infants and those with BW ≥ 1500 g (22/30, 73% vs. 25/37, 68%, p  = 0.91), EOS and LOS cases (28/38, 74% vs. 19/29, 66%, p  = 0.36), infants who met primary outcome vs. those who did not (29/39, 74% vs. 18/28, 64%, p  = 0.37), and those who died vs. those who survived (12/17, 71% vs 35/50, 70%, p  = 0.96).

E. coli neonatal sepsis accounts for a more significant proportion of cases since the widespread adoption of GBS IAP prophylaxis and subsequent reduction in GBS EOS cases [ 4 , 20 ]. This is reflected in our study, where the incidence rate of E. coli sepsis has increased almost three-fold from 2008–2012 to 2018–2022, despite a similar number of births throughout the entire period. We hypothesize this is predominantly attributable to a global increase in E. coli infection in the community, particularly resistant strains [ 21 , 22 ]. This could potentially lead to increased maternal colonization, leading to more EOS cases, and increased nosocomial transmission, leading to more LOS cases. Although the mortality from E. coli disease did not increase in our cohort during this period, the increase in the number of cases is still of concern as the median duration of antibiotic use in infants who survived after E. coli sepsis was 21 days. On further chart review, a significant number of the cases where CSF studies could not be obtained due to infant instability were subjected to 21 days of antibiotics for possible meningitis. This is particularly concerning as recent epidemiologic studies have shown that every day of antibiotic exposure in neonates increases mortality and risk of bronchopulmonary dysplasia [ 23 , 24 ]. In addition, neonatal EOS and LOS may lead to long-term neurodevelopmental impairments in survivors, both in humans and in animal models of disease [ 25 , 26 , 27 ]. This is one of the few studies to report the factors associated with the composite outcome of mortality or severe illness from E. coli sepsis.

In our univariate analysis, the outcomes of mortality, severe illness, or a combination of either were not associated with sex, maternal race, maternal IAI, maternal intrapartum antibiotic use, delivery type, timing of disease onset, and ampicillin resistance. GA, or BW as a surrogate of GA, thrombocytopenia, and neutropenia, were the only variables significantly different in infants who died or had severe illness, both in the univariate analysis and the multivariate logistic regression model. GA and BW are two variables that have been consistently associated with mortality in neonatal sepsis [ 28 , 29 ]. Thrombocytopenia is more common in Gram-negative and fungal neonatal sepsis than Gram-positive sepsis. It is associated with increased odds of mortality, especially in Gram-negative sepsis in VLBW neonates [ 30 , 31 ]. Thrombocytopenia in neonates has been associated with other morbidities, such as IVH and NEC [ 30 , 32 ]. Thrombocytopenia and neutropenia were both significantly associated with mortality or severe illness in our cohort, both in the univariate analysis and the multivariate logistic regression. Intubation and the need for mechanical ventilation have also often been associated with increased mortality in neonatal sepsis. However, we did not include this variable in our analysis as a majority of these were preterm neonates who were on mechanical ventilation due to their lung disease. We did not have information on the episode-related need for intubation or escalation of respiratory support. There were 15 outborn infants in our cohort, and there were no significant differences in any outcomes when excluding them from analyses. These infants were all transferred from hospitals within our hospital’s health system network.

Antibiotic resistance to E. coli strains has been increasing. Although a substantial number of the isolates in our cohort were resistant to ampicillin, our data were similar to recent cohort studies from the NICHD NRN and the CDC, which point to a more than 80% resistance to ampicillin within E. coli strains isolated from neonates [ 4 , 8 ]. Ampicillin resistance did not change over the 15 years we studied. This starkly contrasts with adult studies, where E. coli is the most commonly isolated bacterial pathogen in culture-positive community-onset sepsis, but the rates of ampicillin resistance are lower and in the 40–50% range [ 33 , 34 ]. Based on microbial susceptibility, unit protocols, and drug availability, either cefotaxime, cefepime, or ceftazidime was added to the antibiotic regimens at our center once an isolate was positive for ampicillin-resistant E. coli . Although there was no difference in the time to positivity between E. coli strains isolated from neonates who met the composite primary outcome vs. those who did not, the prompt switching to a targeted antibiotic could reduce the strength of the finding that ampicillin resistance is not associated with mortality or severity of illness.

Antimicrobial resistance was also similar in VLBW and non-VLBW infants, and we did not find any relationship between ampicillin resistance and death, severe illness, or the composite outcome of either in our study. These data are reassuring, especially in the era of multi-drug resistant E. coli , drug shortages, and concern regarding the continued efficacy of ampicillin and gentamicin as first-line empiric antibiotic therapy for EOS in neonates [ 35 ]. Neonates with antibiotic-resistant E. coli sepsis may also require prolonged hospitalization, which increases the risk of exposure to healthcare-associated infections, including other antibiotic-resistant pathogens. Antimicrobial resistance also leads to longer antibiotic duration and the use of multiple antibiotics, which are independently associated with mortality and morbidity in neonates. Our data point toward the hypothesis that antimicrobial resistance and host-pathogen interaction are possibly exclusive of each other. Mortality and severity of illness are a function of bacterial pathogenicity and host susceptibility. Preterm neonates are susceptible hosts in this context due to their immature developing immune system, which has a dysregulated immune response. Bacterial pathogenicity, which is one of the key drivers of the exaggerated immune response in preterm neonates, leads to endotoxin-mediated tissue damage and hypoxia, metabolic acidosis, cardiorespiratory compromise, organ dysfunction, and ultimately death. Our data lends credibility to the recent AAP consensus statement on the continued use of ampicillin and gentamicin for EOS and warrants caution when attributing causes of sepsis-related death to resistant bacteria [ 36 , 37 ]. We did find a trend towards increased ampicillin resistance in the neonates who met the primary outcome; however, we cannot comment on whether this would have reached statistical significance with a larger sample size.

A major strength of this study is the extended study period. The incidence of E. coli neonatal sepsis is relatively low at any major US NICU. Most studies include NRN, CDC, or VON datasets, which do not account for geographic variations in E. coli pathogenicity patterns. We also use a composite outcome of death or severe illness to identify infant and maternal risk factors. As neonatal care becomes better at reducing the outcome of death, there is an increased burden of morbidities in the survivors, and it is critical to include this in studies on sepsis [ 38 , 39 ]. It is reassuring that ampicillin or gentamicin resistance was not associated with the primary outcome in our study. The rates of ampicillin and gentamicin resistance seen in our population are similar to the rates reported by the recent NRN study, and this rate was similar across EOS and LOS cases as well in VLBW infants who were at the highest risk of death or severe illness [ 4 ]. Although this is from a single center, these data suggest that ampicillin and gentamicin remain effective empiric antibiotics for neonatal sepsis.

The main drawback of this study is that this is a single-center experience; thus, the number of E. coli cases is insufficient to draw conclusions that can be applied to other centers. We were also limited as to which clinical, demographic, and microbiological factors we could use for our analyses due to the retrospective nature of this study. The completeness of the documentation of these factors in the medical record primarily determined the factors we chose to study. We acknowledge that there are possibly multiple other complex mechanisms and factors that are associated with increased mortality or severity of illness in neonatal E. coli sepsis. Maternal factors that have been linked to neonatal death from sepsis are prolonged rupture of membranes, chorioamnionitis, preterm delivery, and cesarean delivery [ 40 ]. Neonatal factors that have been consistently shown to be associated with increased odds of mortality are prematurity and low birth weight [ 41 , 42 ]. Other clinical factors such as respiratory compromise, septic shock, Gram-negative infection, lactic acidosis, and thrombocytopenia have been frequently but not consistently implicated in neonatal sepsis-related mortality [ 42 , 43 , 44 , 45 ].

The laboratory system at our institution instituted new microbiological techniques with enhanced antimicrobial inactivation to improve the isolation of microorganisms in 2016. This upgrade might partially account for the increased E. coli incidence in the last five years. We also do not have follow-up data on the surviving infants, so we cannot speculate on their long-term neurodevelopmental outcomes. However, studies show that hypotension and the need for inotropes are associated with neurodevelopmental impairment (NDI) in preterm infants [ 23 , 46 ]. We also do not have data on other Gram-negative pathogens, such as Klebsiella and Pseudomonas , and hence cannot comment on whether there was a similar increase in their incidence during the time period of our study. We also do not have information on the pathogenic strain or phage typing of the E. coli species isolated, as this is not routinely reported in our laboratory system.

This study adds to the current wealth of knowledge of E. coli sepsis in the neonatal population and the relationship between various host and pathogen factors contributing to poor outcomes.

Data availability

Original data is available upon request from the corresponding author.

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These authors contributed equally: Adriana Hoffman, Sriram Satyavolu

Authors and Affiliations

Rainbow Babies and Children’s Hospital, Case Western Reserve University School of Medicine, Cleveland, OH, USA

Adriana Hoffman, Sindhoosha Malay, Thomas Raffay & Devashis Mukherjee

Case Cardiovascular Research Institute, Cleveland, OH, USA

Sriram Satyavolu

Case Western Reserve University School of Medicine, Cleveland, OH, USA

Danah Muhanna

Department of Pathology, University Hospitals Cleveland Medical Center, Cleveland, OH, USA

Anne Windau & Eric M. Ransom

Department of Pathology, Case Western Reserve University School of Medicine, Cleveland, OH, USA

Eric M. Ransom

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AH conducted chart review, data gathering, writing, and manuscript editing. SS conducted statistical analysis and wrote and edited the manuscript. D Muhanna conducted a preliminary literature review and wrote and edited the manuscript. SM conducted statistical analysis. TR performed data gathering and edited the manuscript. AW and ER conducted GA gathering, reviewing, and editing the manuscript. D Mukherjee conducted chart review, statistical analysis, writing, and editing and provided a conceptual framework for the manuscript.

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Correspondence to Devashis Mukherjee .

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This study was performed in accordance with the Declaration of Helsinki. The Institutional Review Board at University Hospitals Cleveland Medical Center approved this study (20221171) and waived the need to obtain informed consent from study participants due to its retrospective nature. All methods were performed according to institutional guidelines and regulations.

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Hoffman, A., Satyavolu, S., Muhanna, D. et al. Predictors of mortality and severe illness from Escherichia coli sepsis in neonates. J Perinatol (2024). https://doi.org/10.1038/s41372-024-02117-9

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Received : 20 February 2024

Revised : 03 September 2024

Accepted : 06 September 2024

Published : 12 September 2024

DOI : https://doi.org/10.1038/s41372-024-02117-9

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