The economic consequences of obstetric fistula: A systematic search and narrative review

Obstetric fistula develops from obstructed labor and is a devastating condition with significant consequences across several domains of a woman's life. This study presents a narrative review of the evidence on the economic consequences of obstetric fistula.


| INTRODUC TI ON
Obstetric fistula (OF) is a complication caused by obstructed labor and consequent ischemic necrosis of the soft tissues adjacent to the baby's presenting part in the birth canal 1 resulting in the formation of an opening between the vagina and the adjacent urinary tract or the vagina and the rectum, or both.Predominantly, OF results from limited access to quality obstetric care including timely identification of signs of obstructed labor and access to assisted delivery such as cesarean section. 2 The condition has been essentially eradicated in higher income countries but is still prevalent in some low-and lowermiddle income countries (LMICs) where access to affordable and acceptable obstetric care is limited.OF has been found to predominantly affect women who are poorer, live in rural areas and have lower levels of education. 3Treatment of OF typically involves surgical repair; however, these services are poorly accessible in many LMICs and as a result many women live with this debilitating illness for extended periods, sometimes throughout their entire lives. 2 OF leads to many physical symptoms (such as incontinence and recurrent infections), 4 significant psychological consequences (including depression and anxiety), 5 and major social consequences 6 (such as isolation and difficulty maintaining intimate and family relationships).
][9][10][11] In these reviews, the economic consequences of OF feature as a prominent and pervasive issue for women living with the condition.However, economic consequences are often discussed as part of an analysis of the wider psychosocial ramifications of the condition and given limited attention in their own right.There is a noticeable absence of a comprehensive review that specifically examines the economic implications of OF for women, their families and the wider society and economy.This is particularly concerning given that OF disproportionately affects those who are most vulnerable to the impoverishing effects of the condition and have limited access to healthcare.The present study seeks to address this knowledge gap by reviewing the literature to respond to the following question: What are the microeconomic and macroeconomic consequences of obstetric fistula?Addressing this knowledge gap will enable a clearer understanding of the economic challenges faced by women with OF, which in turn, can potentially encourage increased investment from both the global and state health actors.The resulting body of evidence can also pave the way to the development of informed policies aimed at enhancing the overall health and wellbeing of women diagnosed with OF.

| Study design
Our research question was defined using the PEO framework as follows: the population (P) of interest was defined as women in any country with obstetric fistula, either living with the condition or after having their fistula repaired; Exposure (E) is obstetric fistula and outcome (O) of interest are the macroeconomic and microeconomic consequences following the development of OF.Microeconomic consequences were defined as the economic burden on the individual and household levels.We defined macroeconomic consequences as a loss of gross domestic product (GDP).1 with the Boolean operator "OR".All search terms from column 1 were then combined with those in column 2 using the Boolean operator "AND".Database filters were used to restrict search results to papers published after 2003, coinciding with the launch of the UNFPA Campaign to End Fistula UNFPA. 2

| Study selection
Studies were selected by first reviewing titles and abstracts to identify papers with potential relevance to the research question.
The full texts of potentially relevant studies were then reviewed to identify those directly addressing the research question.The inclusion and exclusion criteria used to screen studies at each phase are shown in Table 2.All records were screened by one author (KB), with uncertainties discussed with three other authors (VLO, SA, TM) to determine eligibility for inclusion through consensus.Reference lists of included papers were reviewed to identify additional papers deemed relevant for inclusion.Primary research papers identified from the review articles returned in our search were included if they addressed the research question.Review articles were also included if they provided an analysis or narrative that built upon the information available in the primary studies included.Gray literature was not included.

| Data extraction and analysis
Data were extracted using a template which was developed a priori to capture information on the characteristics of the studies and the TA B L E 1 Search terms used for each database.

Search terms for obstetric fistula
Search terms for economic consequences "Obstetric fistula*", "Vesicovaginal fistula*", "Rectovaginal fistula*", "Urogenital fistula*", "Urovaginal fistula*" Economic, Income, "return to work", GDP, "gross domestic product", Microeconomic, Macroeconomic, Finance*, Financial, Productivity, Cost*, Earning* key findings relating to economic impacts.Key findings were organized under three thematic categories: (1) economic consequences of having the condition, (2) economic consequences of seeking care, and (3) macroeconomic consequences.See Table S1 for an overview of the fields of information captured in the data extraction table.
Data were extracted in the form of segments of text representing units of meaning relating to each of the thematic categories.
From these data, individual themes were identified from concepts or issues that were recurrent or prominent within or across studies.

| Overview of studies
The PRISMA diagram outlining the search and screening is shown in Figure 1.
Of the included papers, six were review articles and 43 were primary research papers.A total of 41 of the papers focused on countries in sub-Saharan Africa, with the rest focusing on South Asia, or multiple LMICs.Thirty-seven studies were published within the last decade (after 2012).Themes explored in the papers included physical and/or psychosocial consequences of OF, barriers to accessing care, and challenges women face after surgery.Only one study 12 exclusively examined economic consequences of OF.Forty of the papers involved interviews or surveys of women with OF and 22 papers reported that the majority of their respondents had "low" levels of education (such as primary school level or below).
Twelve studies reported that the majority of their participants lived in rural areas and three papers reported that participants were from a lower socioeconomic strata.Overall, the majority of studies focussed on the types of economic consequences experienced by women and their families; however, few data were found that provided a more quantitative analysis of this economic burden.Table 3 provides a list of the included studies and the main themes identified in each.

| Economic consequences of having the condition
Women losing their jobs due to OF emerged as a major theme across many studies.Asia and Africa and found that 59.88% of women were unable to maintain their prefistula income. 30Some studies found that women often lost their jobs due to a complex interplay of social, psychological and physical consequences from their condition. 24,33For instance, socially, women with OF were described as being subject to harsh stigma, often being seen as "dirty", cursed or believed to be carrying a sexually transmitted infection. 43Due to this stigma, women with OF lose their jobs as their communities do not want to interact with them. 22,39,40For example, Mohamed et al. 26 report the case of a woman who was asked to leave her job washing clothes for a family in her community due to the smell from the leaking fistula.
In some studies, women reported the feeling of fear secondary to the stigma they were subject to as an inhibiting factor for their return to work or reintegration into the community. 25,35,42The physical symptoms of OF such as constant leaking, incontinence, pain, sores and wounds, and foot drop were also reported to directly prevent women from working. 42,44This had economic consequences for women with jobs that required walking 28 or farm work. 23,36This inability to work and loss of job meant that women were unable to secure capital for future employment opportunities, such as starting new businesses. 21,36This perpetuates their financial precarity and TA B L E 2 Inclusion and exclusion criteria used during each phase of screening.

Inclusion criteria
All types of papers, including editorials, opinion pieces, conference abstracts, systematic reviews, reviews, primary research studies Any studies reporting on microeconomic consequences of OF, such as lost income, expenses related to treatment, expenses related to managing the condition, financial stress-including downstream social and psychological consequences from this.Any studies reporting on microeconomic consequences of OF, such as impact on GDP Any studies reporting on physical, social or psychological symptoms of OF All types of papers, including editorials, opinion pieces, conference abstracts, systematic reviews, reviews, primary research studies Any studies reporting on microeconomic consequences of OF, such as lost income, expenses related to treatment, expenses related to managing the condition, financial stressincluding downstream social and psychological consequences from this.Any studies reporting on macroeconomic consequences of OF (impact on GDP) makes it difficult for women to retain or regain economic independence, thus pushing them further into poverty. 40The literature reveals that the economic consequences of OF can be so severe that in the face of these, some women interviewed in these studies had to turn to begging to make ends meet. 11,16,26,36,44Interestingly, other papers note some women attempted to maintain their financial autonomy and cope with OF by finding new work, such as engaging in casual jobs or beginning to work in markets where they were not known, while others continued with their old jobs. 29,40,4548,50 Women with OF face further economic hardship when they are abandoned by those they depend on for financial support-a theme which further demonstrates the interplay between social and economic consequences of the condition.Abandonment comes in the form of social exclusion and divorce which can be precipitated by stigma and perceived shame surrounding the condition, 25 as well as infertility and inability to engage in sexual relationships leading to women feel themselves to be "useless" which in turn can lead to marital strain. 11,17,28men with OF display resilience and personal agency by utilizing various coping mechanisms to manage and adjust to the symptoms of OF such as changes to hygiene routines, seeking support from family and community groups and adopting optimistic attitudes. 22,35,36][40]48,[50][51][52] This includes money required to buy pads, soaps, and other hygiene products to manage symptoms.Some studies described women having to buy inferior quality products as they were more affordable, although offering poorer relief. 37,39,50These expenses were reported to be particularly economically costly for women with OF who have already experienced loss of income and financial dependence. 40e literature shows that the economic consequences of developing OF extend beyond the woman herself and have potentially long-term ramifications for the health and prosperity of their families.Some studies describe how women have been unable to afford nutritious food for themselves or their families, 20,31,33,42,44 and/or have struggled to afford their children's education. 14,21,31This highlights the potential for the economic consequences of OF to exert intergenerational damage and perpetuate the cycle of poverty and social disadvantage.

| Economic consequences of seeking care
The studies reviewed described the costs of OF repair services; the key finding being that the costs are high and often impoverishing for women and their families.An analysis of 16 demographic and health surveys across 14 sub-Saharan African countries showed that of the women who did not seek repair for their fistulae, 84% did not do so due to concerns about the cost of treatment. 53Studies reported that the cost of treatment ranged from USD 222-359; and highlighted that this was unaffordable for most women with OF. 12,22,54,55 Among the studies reviewed, fistula treatment services were reported to be provided free of charge in Ghana, 20 Uganda, 23,55 Nigeria 23 and Kenya. 45However, despite free treatment, women and their families incur additional expenses when seeking care, such as the cost of food, clothing and medications. 20,23,46Keya et al. 23 reports that while parts of the services may be free of charge, women or their guardians may have to pay an extra fee to be provided timely care.Lyimo et al.
describe that some women turn to traditional healers due to social and cultural beliefs of fistula being the result of a bewitchment or religious indictment. 56Engaging traditional healers has been reported to come at a significant cost, which can be unpredictable and sometimes leave women without funds to access services in the formal sector. 14,23ny papers reported that the cost of repair was unaffordable for women who had lost their jobs or source of financial support. 14,17,26,36,55,56To pay for their treatment, women often had to sell their personal goods such as land/property and livestock, leaving them at a disadvantage for future income generation. 14,20,23,31,34,45 addition to the costs of repair services, travel expenses associated with seeking care were also highlighted as a significant economic consequence of OF. 11,13,[15][16][17]23,32,34,36,45,48,55 One paper reported transportation costs to be up to USD 25 in Uganda, 23 where the average monthly income per capita is only USD 54.75. 57 Access to care ecomes difficult for rural women who face the combined barriers of limited healthcare services in their area as well as long and expensive journeys to a facility with the capacity to provide fistula repair.13,41,42 Some studies highlight how social constructs relating to a woman's standing in her family underly a deeply rooted economic disempowerment which may be experienced by women with OF and serve as a barrier to repair.Several studies described how household finances are controlled by men, and paying for women's personal wellbeing and OF care was deprioritized or disregarded. There are economic consequences associated with OF that persist or emerge post-surgical repair.51 If treatment is unsuccessful, women incur ongoing expenses as they continue to seek successful repair and manage persistent symptoms.20,23,34,45,47,48 Additionally, it has been reported that women were often at a major financial loss from seeking surgical care and must recover from this debt.16,33,42 Follow up from surgery can be expensive and require several referrals, each associated with medical and non-medical expenses.
Chimamise et al. describe that women may abandon seeking fistula repair when it becomes unaffordable. 58 addition to direct expenditure, the indirect costs of seeking care include time away from work for treatment and recovery and were described in several studies.One study reported the average indirect cost of OF treatment in Northern Nigeria to be USD 107.54, nearly twice the monthly minimum wage for people employed in the formal sector. 12Whether employed in the formal or informal sector, women have been shown to experience income and productivity losses during long hospitals stays required for treatment. 14,23,43,51,52sses continue when these women return home and are unable to work during recovery-especially if work is physically laborious. 16,21,47,48,51,52Therefore, women who achieve successful OF repair may be left with economic struggles due to both the direct and indirect costs of seeking care.

| Macroeconomic impacts
There were no studies identified that reported on lost GDP due to OF-a key indicator of the macroeconomic impact of the condition.However, one paper quantified the economic cost of fistula from a health system perspective.Epiu et al. 59 conducted a costeffectiveness analysis of fistula repair in Uganda which showed each procedure costs the health system USD 378, equating to a cost of USD 54 per disability-adjusted life year (DALY) averted.As the estimate is well below Ugandan per capita GDP (USD 62), authors argued that fistula repair is a highly cost-effective intervention in the Ugandan context, warranting allocation of sufficient resources.

| DISCUSS ION
This narrative review reports on the economic consequences of obstetric fistula and describes how these affect a woman and her family.Our work shows that economic consequences span the spectrum of women's experiences of OF, from managing the symptoms of the condition, through to seeking care, and can persist after women access surgical repair for the condition.Economic ramifications of OF are often compounding and exacerbate existing vulnerabilities, given that the condition disproportionately affects women from lower socioeconomic strata who are most at risk of falling deeper into poverty. 13,40e economic consequences that result from OF are not only driven by the physical symptoms of the disease but are often intertwined with the complex psychosocial consequences of the condition.For example, the inability to work can be due to physical symptoms such as incontinence, or social factors such as the stigma attached to the condition.When OF leaves women unable to meet cultural expectations around child-bearing, it can lead to abandonment and the severing of financial support. 15Addressing the economic impacts of OF requires a holistic approach, with due consideration for how the complex interplay of physical, psychological, and social consequences drive women into economic distress.
This review has revealed several gaps and limitations in the literature surrounding OF.It is notable that most studies recruited women from fistula centres or similar healthcare facilities.Findings may therefore only be representative of patients who can afford or have access to care.This may limit the understanding of the broad range of experiences of OF and the economic consequences that exist for women under different circumstances.Furthermore, few of the studies sought to specifically explore the economic consequences of OF, with most focused on the broader social or psychological consequences.Thus, the nuances of the economic consequences may not have been fully captured in these reports.
For example, primary research focused specifically on the economic impacts of OF may shed more light on downstream consequences of income losses on the family, such as access to education and nutrition.Further studies into the mechanisms by which women and their families cope with the economic shocks due to OF would also be valuable, as evidence on this point was scarce in the literature reviewed.Research such as this would enable a deeper understanding of the true nature and complexity of the economic consequences that are associated with OF.
Little evidence was found on the macroeconomic impact of OF, leaving an incomplete understanding of the larger economic consequences that extend beyond the individuals and households affected.Further research in this area is warranted and may explore the impact of OF on a range of macroeconomic indicators beyond GDP, such as the effects on unemployment and public spending on maternal health services.This would be critical to enable a greater understanding of the systemic impact OF poses and its potential burden on a country's health system spending or economic growth.
For example, studies that explore the additional cost on the health systems due to prolonged and unaddressed complications of OF would inform prioritization of OF management in LMICs.Another focus may be on the macroeconomic impacts due to productivity loss due to high burden of OF among women of reproductive age.
Such understanding would be crucial evidence to strengthen advocacy for the elimination of OF in LMICs.
Finally, there is a need for more studies that comprehensively characterize and quantify the economic loss suffered by women who have OF.For instance, studies that comprehensively assessed the impact of OF on health care utilization, out-of-pocket expenditures, catastrophic health expenditures, and productivity loss.Such studies are widely available for other conditions including noncommunicable diseases, 60,61 tuberculosis, 62,63 HIV/AIDS, 64,65 and other illnesses, but not on OF.Similar assessments focusing on OF are critical to better define the financial and economic loss women and their families face due to OF and thus would stand to fortify the existing qualitative data on economic consequences.
A limitation associated with this review is that the studies included were screened only for relevance to the research question and not for quality.As a further limitation, our search did not include the identification and screening of gray literature.Therefore, it is possible that a rich source of information on the economic consequences of OF has been missed.However, reporting on the evidence available specifically in the peer-reviewed literature allows for the identification of where further research of this nature is warranted to address gaps and bolster the evidence base for driving advocacy efforts and policy development.
Understanding the nature of the economic hardships faced by women with OF provides insights important for improving interventions and strategies that may help to mitigate these consequences.
Strategies targeted toward prevention and treatment of OF form the cornerstone of existing approaches to reduce the burden of the condition worldwide. 2These include investing in obstetric capacities to reduce obstructed labor in LMICs and strengthening surgical systems to expand capacities for OF repair.The economic consequences of OF identified here can help shape and expand these approaches to drive a robust and holistic response to this condition and all its consequences.Funding schemes providing free surgical repair services should be expanded to include non-medical costs of seeking care such as food, transport, and clothing.Similarly, programs to assist women with financial security as part of social reintegration post-surgery are critical as financial insecurity was identified as a main factor in the continual burden of OF. 17,18,45,47,48,66 Importantly, some studies noted the value of reintegration programs which teach women income generating skills to help achieve financial stability and reintegration. 27,47,49l interventions and strategies to address fistula will require careful consideration on how they will impact women who are poorer and living in rural areas, as findings from this review show these groups of women face a disproportionate burden of OF and its economic consequences.This should include, but not be limited to, consideration of comparatively higher costs of seeking care experienced by women living in rural areas, who typically face long and complex journeys to reach quality fistula repair services.In this respect, outreach fistula repair campaigns which bring fistula repair services to more rural areas are beneficial.However, the transient nature of service availability associated with these "campaign-style" models illustrates the importance of striving toward more sustainable solutions to addressing the barriers and economic consequences of seeking fistula repair services.Ensuring equitable access to prevention programs, fistula repair services, and reintegration programs for those in need will require addressing the affordability, physical accessibility, and social acceptability of these programs.

| CON CLUS ION
This review shows that economic consequences that arise from OF are multifaceted, pervasive and are intertwined with the physical and psychosocial impacts of the condition.Understanding these consequences will be critical for a tailored response to ensure it addresses the financial burden that OF has on a woman and her family.
The existing literature extensively covers the microeconomic consequences of OF, but there is a notable lack of research addressing its macroeconomic impact.To generate stronger political commitment and attract the attention of global health actors, it is imperative to conduct further research on the effects of obstetric fistula on national economies, particularly in LMICs where this condition is prevalent.A better understanding of the macroeconomic impact will drive further investment in this area to contribute to the development of multisectoral strategies, harnessing more sustainable health system responses, which are better integrated with appropriate social services, aimed at effectively addressing the challenges posed by this debilitating health condition.

3
Overview of papers included in the review and main themes identified from each paper.

Table 1
presents the keywords used to search the literature across two major public health databases-Medline OVID and Global Health-on May 15-16, 2022.These same search terms were used across these databases again on November 21, 2022 with the addition of the Scopus database to update and expand our search.
11,Roa et al. surveyed 130 women with OF across