Series 604 www.thelancet.com Vol 387 February 6, 2016 Ending preventable stillbirths 3 Stillbirths: economic and psychosocial consequences Alexander E P Heazell, Dimitrios Siassakos, Hannah Blencowe, Christy Burden, Zulfi qar A Bhutta, Joanne Cacciatore, Nghia Dang, Jai Das, Vicki Flenady, Katherine J Gold, Olivia K Mensah, Joseph Millum, Daniel Nuzum, Keelin O'Donoghue, Maggie Redshaw, Arjumand Rizvi, Tracy Roberts, H E Toyin Saraki, Claire Storey, Aleena M Wojcieszek, Soo Downe, for The Lancet Ending Preventable Stillbirths Series study group* with The Lancet Ending Preventable Stillbirths investigator group* Despite the frequency of stillbirths, the subsequent implications are overlooked and underappreciated. We present fi ndings from comprehensive, systematic literature reviews, and new analyses of published and unpublished data, to establish the eff ect of stillbirth on parents, families, health-care providers, and societies worldwide. Data for direct costs of this event are sparse but suggest that a stillbirth needs more resources than a livebirth, both in the perinatal period and in additional surveillance during subsequent pregnancies. Indirect and intangible costs of stillbirth are extensive and are usually met by families alone. This issue is particularly onerous for those with few resources. Negative eff ects, particularly on parental mental health, might be moderated by empathic attitudes of care providers and tailored interventions. The value of the baby, as well as the associated costs for parents, families, care providers, communities, and society, should be considered to prevent stillbirths and reduce associated morbidity. Introduction Despite the 2*6 million stillbirths worldwide,1 the costs of stillbirth are largely unknown and therefore unappreciated by contrast with other adverse pregnancy outcomes.2–5 For the most part, health metrics, such as quality-adjusted life-years (QALYs) and disability-adjusted life-years (DALYs), have neglected stillbirth. No value is generally given for the loss of life or the loss to parents and families. Most economic analyses have focused on the cost of stillbirth prevention.4,6,7 In low-income and middle-income countries (LMICs), costs vary from US$4781 to $10 571 per stillbirth averted (in 2013 prices).4,6 In high-income countries (HICs) with lower stillbirth rates, prevention costs are greater than they are in LMICs-eg, smoking cessation costs $125 961 per stillbirth averted.8 If stillbirths are included in analyses of the eff ect of antenatal and intrapartum care on maternal and newborn deaths, the cost per death averted reduces substantially from $27 551 to $2143 (panel 1).4 However, to accurately assess whether these programmes are cost eff ective, a better appreciation of the costs of stillbirth is needed and so far, no comprehensive estimates have been made. In this Series paper, the costs associated with stillbirths are described as direct (including the cost of medical care) or indirect (such as welfare payments) fi nancial costs. Outcomes are divided into psychological and social eff ects on bereaved parents and families,10 and overall eff ects on health professionals. We identify these costs and outcomes through systematic reviews and new analyses of published and unpublished data (panel 2). We also evaluate interventions to reduce negative eff ects, such as parental support by peers or professionals. To address the cost-eff ectiveness of these interventions and those to prevent stillbirth, we consider the eff ects of diff erent methods used to value the loss of fetal life. Most studies included in the systematic reviews were undertaken in HICs (n=177), with fewer studies in LMICs (n=26; appendix p 68). Direct fi nancial costs of stillbirth Three studies describing direct costs, including investigations into the cause of death, ranged from $1450,19 and £195111 to $8067.20 Care costs for stillbirths were 10–70% greater than with a livebirth.11,20 Direct costs of health-care provision were typically met by government or insurance Lancet 2016; 387: 604–16 Published Online January 18, 2016 http://dx.doi.org/10.1016/ S0140-6736(15)00836-3 See Comment pages 515, 516, e14, and e16 See Series pages 574 and 587 See Online/Series http://dx.doi.org/10.1016/ S0140-6736(15)01020-X and http://dx.doi.org/10.1016/ S0140-6736(15)00954-X This is the third in a Series of five papers about ending preventable stillbirths *Members listed at the end of paper Institute of Human Development, Faculty of Medical and Human Sciences Key messages Cost of stillbirth to families and society Stillbirth is associated with substantial direct, indirect, and intangible costs to women, their partners and families, health-care providers, the government, and the wider society. Appreciation of the costs of stillbirth is essential to evaluate the cost-eff ectiveness of interventions to prevent stillbirth or ameliorate negative eff ects of stillbirth. Data limitations related to costs Data for the cost of stillbirth in high-burden countries are inadequate. In addition to the collection of data for the number of stillbirths, data should also be collected for the resource implications. Challenges faced by parents after a stillbirth Adverse experiences including stigma, social isolation, and disenfranchised grief are widespread among parents whose baby is stillborn and need to be addressed through focused interventions and supportive activities including parents, communities, care providers, and relevant stakeholders. Respectful and supportive care to families following a stillbirth Empathic behaviours during every encounter between bereaved parents and caregivers are essential to minimise additional emotional and psychological burdens in the short, medium, and long term. Adverse eff ects on health workers Caring for families during and after stillbirth places a substantial personal and professional burden on staff . Negative eff ects on staff could be addressed by education, training, and provision of formal and informal support. Series www.thelancet.com Vol 387 February 6, 2016 605 companies, although in some cases this expenditure was passed on to parents; 14% of respondents from HICs and 32% from middle-income countries (MICs) had medical costs to meet during and after the birth. Where reported, parents paid between $197–3093 for investigations to identify the cause of stillbirth and $118–20 000 in hospital fees for additional medical care (appendix pp 75, 76). No direct reports of the cost of care in subsequent pregnancy exist, although three papers,21–23 all from HICs, recommended additional fetal monitoring in pregnancies after a stillbirth. By use of these recommendations to derive models of care, we estimated costs from £3499 after a stillbirth of a non-recurrent cause to £4057 for a stillbirth of unknown cause.11 A pregnancy after stillbirth costs £558–1735 more than if the previous pregnancy ended in an uncomplicated livebirth. Additionally, if care included more intensive surveillance with cardiotocography, costs rose to £4654–5616.24,25 Thus, the costs of subsequent pregnancy care add to the health-care costs associated with stillbirths in HICs; this situation will extend to MICs as these countries scale-up more intensive antenatal monitoring and care. Indirect fi nancial costs of stillbirth The most frequent indirect costs for parents after stillbirth were for the funeral and burial or cremation of their baby (appendix). For some, this cost was mitigated by health insurance, government payments, or grants. Parents' free text responses in the International Stillbirth Alliance (ISA) survey (appendix pp 22, 23) show the substantial fi nancial burden of this group, magnifying the eff ect of these parents' loss (panel 3). Although some parents did not have to pay, others reported costs for funerals ranging from $469–11 719, extending to $1179–11 605 for burial plots and $1410–4605 for memorials (appendix pp 75, 76). The theme that occurred most frequently in the free text responses was the long-term fi nancial eff ect on families. For many parents, stillbirth was associated with reduced earnings from employment or an inability to return to paid employment. Meeting the continuing costs of counselling and medical care in further pregnancies was also mentioned. The experience of stillbirth also aff ected parents' employment, with 10% of bereaved parents remaining off work for 6 months, and 38% of mothers and 21% of partners reducing their working hours (panel 3). Even after parents return to work, productivity was greatly reduced with estimates of 26% of normal work after 30 days, increasing to 63% after 6 months. Searches of the International Labour Organization database showed that only 12 of 170 countries with maternity benefi t policies included specifi c provision for stillbirths; an average of 11 days leave for mothers (range 28–84 days) and an average of 1 day off for fathers (range 1–5 days). Even in the few countries with this leave provision, bereaved parents seem to have little option to delay their return to work. Policies relating to stillbirth or miscarriage were identifi ed from fi ve (10%) of 51 African countries, fi ve (18%) of 28 countries in Asia, three (6%) of 47 countries in Europe, and four (12%) of 34 in the Americas (appendix pp 78–81). Governments might incur costs in countries that extend maternity rights to the parents of a stillborn child. Panel 1: Modelled scenario-the eff ect and cost of 90% coverage for quality antenatal and intrapartum care We used Lives Saved Tool (LiST) (version 5.28) to model the results of eff ective proven interventions on stillbirths and maternal and neonatal deaths. We modelled the potential eff ect of introducing selected interventions within health systems of the 75 high-burden Countdown countries (which account for 99% of all deaths).9 For each of the 75 Countdown countries, baseline scenarios were created that represent the most up-to-date details about the health status of these countries, including mortality, cause of death structure, and present coverage of interventions. The base year was set as 2015 and coverage of selected interventions was scaled up linearly to reach 90% by 2030. The modelled interventions were grouped into four packages along the continuum of care. • Preconception nutrition care: balanced energy and protein supplementation, folic acid supplementation or fortifi cation, and micronutrient supplementation (various micronutrients, including iron and folic acid). • Basic antenatal care: prevention of malaria with insecticide-treated bednets or intermittent preventive treatment with antimalarial drugs, syphilis detection and treatment, and tetanus toxoid immunisation. Intermittent preventive treatment was scaled up only in countries where malaria is endemic and the eff ect would apply only to the proportion of women exposed to malaria. • Advanced antenatal care: detection and management of hypertensive disorders of pregnancy, including treatment with magnesium sulphate and hospital care or caesarean section if needed, detection and management of diabetes in pregnancy, detection and management of fetal growth restriction, identifi cation, and induction of mothers at 41 weeks of gestation or more. • Childbirth care: skilled birth attendance, antenatal steroids for preterm labour, antibiotics for preterm premature rupture of the membranes, active management of the third stage of labour, neonatal resuscitation, immediate assessment, and stimulation of the newborn baby. For costing, we used the LiST costing submodule to assess the running costs of the interventions for which we used an ingredients-based approach, identifying and valuing every resource. The costing submodule draws its assumptions about staffi ng, drugs, and need for services from the UN's OneHealth Tool database. We have included only running costs and that was divided in four components: capital costs, drug and supply costs, labour costs, and other recurrent costs. The results suggest that scaling up these proven antenatal and intrapartum interventions in the 75 high-burden countries can prevent 823 000 stillbirths, 1 145 000 neonatal deaths, and 166 000 maternal deaths annually by the year 2030 (appendix p24) at an additional annual running cost of US$4*6 billion or $2143 for each life saved (including stillbirth, maternal, and neonatal deaths; appendix p24). The analysis suggests that interventions in the preconception, basic, and advanced antenatal care packages are crucial, but most of the deaths including stillbirths and neonatal and maternal deaths are prevented by intervening in the intrapartum period alone and with a lower estimated cost of $1370 to save each life compared with $2143 for all interventions. This analysis reaffi rms previous estimates that not only is prevention of stillbirths possible but prevention can be achieved at a reasonable cost of $2143 for each life saved. University of Manchester, Manchester, UK (A E P Heazell PhD); St Mary's Hospital, Central Manchester University Hospitals NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK (A E P Heazell); International Series 606 www.thelancet.com Vol 387 February 6, 2016 Stillbirth Alliance, New York, NY, USA (A E P Heazell, D Siassakos MD, V Flenady PhD, K J Gold MD, C Storey BA, A M Wojcieszek BPsySci); Academic Centre for Women's Health, University of Bristol, Bristol, UK (D Siassakos, C Burden MD); Southmead Hospital, Bristol, UK (D Siassakos, C Burden); Centre for Maternal Reproductive and Child Health, Department of Infectious Disease Epidemiology, London School of Hygiene & Tropical Medicine, London, UK (H Blencowe MRCPCH); Center for Global Child Health, Hospital for Sick Children, Toronto, ON, Canada (Prof Z A Bhutta PhD); Center of Excellence in Women and Child Health, Aga Khan University, Karachi, Pakistan (Prof Z A Bhutta, J Das MBA, A Rizvi MSc); Arizona State Psychological and social eff ects of stillbirth The period after stillbirth has extensive consequences for parents and their families. Much of the eff ect is nonmonetary, resulting from the negative eff ects of grief, anxiety, fear, and suff ering. These emotional factors have been described as intangible costs.26 Almost all parents report negative psychological symptoms after a stillbirth. In the Listening to Parents study14 in the UK (n=473), 68% of mothers and 44% of partners reported four or more negative psychological symptoms at 10 days, reducing to 35% of mothers and 13% of partners at 9 months. This situation is over three times greater than after a livebirth, when 8–13% of mothers and 3% of fathers report depressive symptoms at about 9 months after the birth of their baby.27–29 Family was the most frequently cited source of support for parents after a stillbirth, although family input was not universally positive (panel 3). This need for support between parents and the wider family could strain relationships. In the Listening to Parents study, 9% of mothers and 5% of partners reported diffi culties in their relationship 9 months after the event, and a similar proportion reported issues with other family members (12% of mothers and 4% of partners).14 In the TEARS cohort30 in the USA (n=216), the mean Family Assessment Device score of respondents was 3*2 (range 0*5–4*0), in which a score of 4 indicates substantial dysfunction in family relationships. Ultimately, this tension might lead to relationship breakdown, which some studies report as more frequent in parents who have a stillborn child compared with a livebirth (odds ratio 1*40, 95% CI 1*10–1*79).31 In another study,32 the proportion of families that divorce is unchanged, but perceived relationship quality changed between married (improved) and single women (deteriorated). Systematic searching located 1082 relevant datapoints from 144 studies of the psychological eff ect of stillbirth (appendix pp 31–50). These data were summarised into 23 themes and thematic sentences of the eff ect on parents with variable frequency eff ect sizes (table). The most frequently reported experiences after stillbirth were negative psychological symptoms, including high rates of depressive symptoms, anxiety, post-traumatic stress, suicidal ideation, panic, and phobias.33,34 Although most studies evaluated these symptoms subjectively rather than with a formal clinical diagnosis, 60–70% of grieving mothers in HICs reported grief-related depressive symptoms that they regarded as clinically signifi cant 1 year after their baby's death.30,65 These symptoms endured for at least 4 years after the loss in about half of cases. If these fi gures are extrapolated to the 2*6 million women who had a stillbirth each year,1 an estimated 4*2 million women are living with depressive symptoms after stillbirth. Many parents reported persistent feelings of remorse or guilt for not being able to save their baby. Nearly 40% of grieving mothers in a convenience-sample survey15 in the USA were prescribed psychiatric drugs despite an absence of evidence for the effi cacy of these drugs in this population. Parents responding to the ISA survey reported accessing internet forums (more than 85%), support groups (about 30%), or consulting with religious leaders (about 30%) or health-care professionals (about 55%) to address their psychological symptoms. Little diff erence was noted in the sources and frequency sought by parents from HICs and MICs (appendix p 69). Psychological distress persisted into subsequent pregnancies when parents reported diff ering emotions (eg, relief and worry, hopeful optimism, and panic attacks or depressive symptoms).66 Women tended to report volatile emotional states, whereas fathers tended to report suppression of their feelings. Parents were afraid to prepare for the birth of their subsequent baby and avoided general antenatal classes because they felt, as parents, they were outside the boundaries of normality. Some women struggled to diff erentiate their dead baby's identity from their subsequently-born live baby. The capacity to express and integrate grief reactions was a crucial part of parents' psychological responses. Many studies described disenfranchised grief, when Panel 2: Methods To extend the knowledge base with respect to direct, indirect, and intangible costs of stillbirth on parents, families, and health-care providers, a series of systematic reviews were implemented and meta-syntheses were undertaken with established methods.11–13 A further systematic review and meta-synthesis identifi ed interventions or systems that might reduce the negative eff ects of stillbirth. Detailed methods of search strategies and PRISMA diagrams are included (appendix). The search strategy was designed to capture the whole fi eld of studies worldwide; no language restrictions were imposed and searches were carried out in CINAHL, AJOL, LILACS, MEDLINE, PsycINFO, Cochrane, and PubMed. After screening, studies were identifi ed by whether they met the inclusion criteria and reported relevant information, three studies reported information on direct costs, 144 studies reported on the psychological and social eff ect on parents, 20 studies reported psychological eff ect on professionals, and 42 studies were included in the analysis of interventions to maximise wellbeing for bereaved parents. To supplement data from published medical literature, we extracted data from three questionnaire studies (the Listening to Parents study,14 the TEARS study,15 and the International Stillbirth Alliance survey) including a total of 5358 parents from high-income and middle-income countries. We searched for data to match ten themes to identify intangible costs of stillbirth identifi ed by systematic review and meta-ethnography.16 Data addressed eight of these themes, including: negative psychological impacts after stillbirth; eff ects on relationships with others; the duration of these eff ects; how soon after the stillbirth parents returned to their previous routine and on returning to work how soon parents returned to a full productive capacity; the nature, adequacy, and eff ectiveness of any supportive measures; and whether parents sought medical treatment or counselling for any negative eff ects associated with the stillbirth. These data were supplemented with data extracted from a questionnaire survey of the experiences of care providers in high-income countries and low-income and middle-income countries distributed by the International Stillbirth Alliance. For the surveys, quantitative data were analysed with descriptive statistics, and free text responses were analysed by thematic analysis. Where fi nancial costs were reported by parents, the costs were converted to US$ and shown in 2013 prices. Published cost estimates are reported in their original currency but shown in 2013 prices.17,18 Series www.thelancet.com Vol 387 February 6, 2016 607 University, Tempe, AZ, USA (J Cacciatore PhD); Institute for Reproductive and Family Health, Hanoi Vinmec International General Hospital, Hanoi, Vietnam (N Dang RM); Mater Research Institute, University of Queensland Brisbane, QLD, Australia (V Flenady, A M Wojcieszek); Department of Family Medicine and Department of Obstetrics, University of Michigan, Ann Arbor, MI, USA (K J Gold); Krachi Midwifery Training School, Kete-Krach, Ghana (O K Mensah BSc); parents felt their grief was not legitimised or accepted by health professionals, family, or society.35–37 This issue was particularly evident in LMICs, in cultures where talking about death is taboo, and where the dead baby was not yet deemed to be a person.50,51,64 In these contexts, mothers' accounts suggested that they suppressed grief in public, instead choosing to deal with the emotions privately and alone.47,48 These accounts are supported by responses to the ISA survey of care providers (LMIC n=117, HIC n=2020). Fewer care providers from LMICs agreed that a death before birth is the same as the death of a child (19% LMIC vs 33% HIC) and more care providers attributed stillbirth to a mother's fault (4% LMIC vs 0*5% HIC) compared with HICs. Respondents from LMICs more frequently agreed that parents should forget about their stillborn baby and have another child (26% LMIC vs 3% HIC) and parents should not talk about their stillborn baby (12% LMIC vs 4% HIC; appendix p 71). Fathers reported feeling unacknowledged as a legitimately grieving parent. The burden of these men keeping feelings to themselves increased the risk of chronic grief.46 Diff erences in the grieving process between parents can lead to incongruent grief,38,39 which was reported to cause serious relationship issues, from confl icts about sexual intercourse to marital breakdown.31,45 Although family and friends were often essential for eff ective support,67 respondents to some studies reported that family members had unrealistic, unhelpful expectations of recovery after stillbirth. Many studies described the adverse eff ects of stillbirth on siblings, a surviving twin, and subsequent children, including issues with parent–child relationships, which could aff ect siblings' physical and mental health in the longer term.43,44 Some parents described anxiety with respect to the wellbeing of children of other parents.54 Stillbirth was reported to have adversely aff ected the emotional wellbeing of grandparents and other family members.59 For some mothers, stillbirth aff ected their approach to life and death, self-esteem, and their own identity.36,41,42 Some mothers reported losing their sense of control, including during subsequent pregnancies, and their confi dence in parenthood and child-rearing. Some women avoided contact with babies, creating social isolation and worsening depressive symptoms.34 Some mothers were hesitant to meet neighbours or those who had known them when they were pregnant. Many women stopped going out, leading to voluntary social isolation. Social isolation could also be involuntary, with parents reporting stigmatisation, resulting in them feeling less valued as members of society.49 In reports from some LMICs,49,50,51,68,69 women reported being substantially less valued by partners, families, and society. In extreme circumstances, this situation has led to spousal abuse, enforced divorce, and rejection by family and society, partly based on beliefs that women who have stillbirths are possessed by evil spirits or have procured abortions. In the period shortly after the stillbirth, changed body image was important.45,58 Some women reported being embarrassed by their body after pregnancy. Others wanted to keep a pregnant body shape, maintaining a connection with their baby. Some women linked the grief to their physical body through physical symptoms, such as pain and by developing an image of themselves as unattractive and ugly. Such negative self-perceptions decreased sexual activity and pleasure. Women reported pressures to delay or prioritise conception originating from themselves or from family and society.34,53 Chronic pain and fatigue, increased substance use, employment Panel 3: Parents' experiences of the direct, indirect, and intangible cost of stillbirth in high-income countries Direct and indirect costs "It's diffi cult as I had already purchased all the baby items and then had additional tests to pay for. I wouldn't have minded [the tests] if my child lived, but having to pay for them after he died was diffi cult and a constant reminder as the bills kept coming" (participant number 3903, Australia). "I could not properly bury my child because I lacked the fi nancial means; that hurts today, because I have no grave" (#19 342, Germany). "The higher cost, in fi nancial terms, was the long process of psychotherapy that I followed in the next three years and more examinations that I had privately before and throughout the course of the next pregnancy" (#11 707, Italy). Employment "The loss of income when you can't bring yourself to go back to work is substantial and many work places don't understand the pain" (#7358, Australia). "Because neither I nor my husband was able to start work after the birth, we had no income. We could not get compensation from the social insurance because we were not sick we were just grieving" (#26 496, Sweden). Financial support from family, friends, and others "I never thought anything like this would happen, so I was unprepared! Glad I had family and friends to help give her a beautiful burial service which I could not aff ord on my own" (#5582, UK). "The funeral home did not charge us for our daughter's cremation or vessel for her ashes. They told us that "we had already paid too much". We will always remember their kindness and compassion" (#2295, USA). Support from family "My family and my friends were a great help to us. They were always there to listen and off er support when I needed it. They got me through a lot of the time" (#4583, Australia). "My family was supportive at fi rst. After a while they seemed to think I should get over the death of my twin; that I had grieved long enough" (#3159, USA). Financial support from government "[The costs of stillbirth were] all paid for by the state. I am very happy for this. It is devastating enough losing your baby, without getting debt because of it, or having to consider if you can aff ord the help you need, or can aff ord a funeral, an autopsy, etc" (#8516, Norway). "All medical expenses were covered by social security and burial expenses by insurance. The only expense was the grave" (#19 795, Spain). Series 608 www.thelancet.com Vol 387 February 6, 2016 Frequency eff ect size* Example quotes (country) Psychological eff ect of stillbirth on parents Stillbirth has been associated with a number of emotional and psychological symptoms 77% "I am depressed, saddened, hurt, empty, guilty and lonely. I cry every day. I will mourn him forever."33 (Australia) "A number of mothers recalled suicidal thoughts because of their desire to be with their baby"34 (USA) Parental grief following stillbirth might not be legitimised by health professionals, family, and society (disenfranchised grief) 31% "Women shared their distress that their motherhood of their dead babies was denied by others. One participant recounted that when she told her sister she was not sure she was ready for Mother's Day rituals, her sister replied "Well, you're not a mother- you have to have your baby fi rst."35 (USA) "This perceived lack of social understanding left these mothers alone and uncomforted. Added to this, the silence was aggravated by the failure of friends and family to acknowledge the loss and grief as real. They experienced people avoiding them, or treating them as though they had never been a mother"36 (Australia) "Women who have not gone through stillbirth don't want to hear about my birth, or what my daughter looked like, or anything about my experience."37 (USA) Stillbirth might have a positive or negative eff ect on relationships-eg, through diff erent grief reactions (incongruent) 29% "Some women felt their husbands did not show any sadness and were impatient with them, they felt their relationship had changed; stillbirth had created a distance between them"38 (Taiwan) "Mothers and fathers stated that they became closer after the loss, and that the feeling deepened over the course of the following year. They had something in common; going through the loss together-a sense of experiencing a special unifying bond"39 (Sweden) In subsequent pregnancy some parents might experience psychological distress 27% "Fathers exhibited great emotion as they shared the burden of worry over what was going on at home. They had diffi culty concentrating at work and called home frequently, asking the mother to validate fetal movements."40 (USA) "You're happy that you are [pregnant] but you can't be that innocent...Am I confi dent? No. Will I relax? No. There is not a point that I will relax until they are out and breathing..."35 (USA) Stillbirth might change parents' approach to life and death, self-esteem, own identity, and sense of control in subsequent pregnancy, parenthood, and childrearing 26% "The thoughts expressed by parents in our study consisted of being more humble and more grateful toward life itself and taking nothing for granted"41(Sweden) "The men in the study also questioned their identity as fathers, uncertain as to their right to the term father"42 (UK) "Each woman struggled with her sense of identity. Although each felt she was a mother, she was a mother without a child, and did not have tangible evidence of her motherhood"37 (Australia) Stillbirth can have an adverse eff ect on siblings, including the surviving twin, and subsequent children 24% "Older siblings from the ages of 7–12 years were described as being worried, nervous, tense, and silent. They were worried about life and their parents' health"43 (Sweden) "Infants next-born after a stillbirth were signifi cantly more likely to be classifi ed as disorganised in their attachment behaviour with their mothers than controls, this was strongly predicted by unresolved mourning in the mothers"44 (UK) After stillbirth some parents might seek isolation, can change their uptake of religious practice, approach to sexual intercourse, engagement with health promoting activities, work, and social media and this behaviour might continue into subsequent pregnancies 20% "The fathers in this study were exhausted, physically and emotionally. When asked to say more about how they managed, a common response was 'I keep myself busy'"40 (USA) "Men looked at sex as a tension reliever and attributed a therapeutic value to it"45 (Norway) "Many parents relied on their spirituality to deal with their loss. For some parents this was in the form of praying; for others, it was going to church"34 (USA) "I cry when I talk to a real person so it was easier to talk to someone online, less emotional"36 (USA) Some parents feel the need to suppress outward grief, including during subsequent pregnancy 18% "Fathers felt they denied their own emotional reactions in order to protect and support and care for their partners"46 (USA) "According to Taiwan's culture talking about death is a taboo subject and these mothers often dealt with their grief privately and alone"47 (Taiwan) "I think I genuinely suppressed a lot of my anxiety because of my [desire to protect my] family. Yes, I wanted to stay strong for my husband and myself. Outward I was strong but inside I was a mess"48 (USA) Stillbirth might lead to avoidance of activities that remind them of the pregnancy and the baby 13% "Most mothers found it very diffi cult to be in situations that reminded them of 'what could have been.' Examples of these situations were being around pregnant women or infants, attending baby showers, and celebrating holidays"34 (USA) Parents report stigmatisation, rejection, and spousal abuse 13% "There were a few people at work who just never spoke to me again...I mean I defi nitely got the feeling...like I was bad luck"49 (UK) "Every time I walked into the living room, my in-laws lowered their voices. Mostly, they stopped talking. I disappointed them because I didn't give them a descendent like every daughter-in-law should do. I felt unwomanly, since I failed to have a baby."50 (Taiwan) "I know a girl who was in school and married off by her parents. After the marriage, she repeatedly lost her new-borns and was divorced. Not to face the humiliation in the village she ran away to a city and now she is a commercial sex worker."51 (Ethiopia) Parents might have mixed feelings towards the decisions they made-eg, post mortem or seeing and holding their baby 13% "In the limited time available for mothers to meet the child, mothers did not know how to spend time with their child, and had multiple hesitations due to their child being dead, and regretted this later on"52 (Japan) Parents might have external or internal pressures to prioritise or delay conception 9% "Some mothers did not plan on a subsequent pregnancy because of their concern about their ability to deal with another perinatal loss"34 (USA) "Perinatal loss signalled a potential underlying health problem, which in turn accentuated anxieties relating to both future reproductive abilities and investment of limited resources on another potentially unsuccessful pregnancy. Such women described feeling pressure to prove their reproductive capabilities as soon as possible"53 (Benin) Bereaved parents might become hypervigilant with siblings and subsequent children, and anxious about other people's children 8% "All mothers shared stories of feeling out of control, especially when faced with normal or common childhood events, such as tonsillitis, middle ear infection, or being stung by a bee. These events were enough to cause them to feel hysteria and intense fear they were about to lose another child"54 (Australia) (Table continues on next page) Series www.thelancet.com Vol 387 February 6, 2016 609 Frequency eff ect size* Example quotes (country) (Continued from previous page) Bereaved parents might increase or decrease their use of health-care services; and in subsequent pregnancy, fathers might express a desire to be more included in care 7% "Mothers with a history of prior perinatal loss may attempt to cope with their anxiety in pregnancy and depression in early post partum with requests for additional health-care resources"55 (USA) "Fathers felt the need to take more interest or active involvement in the subsequent pregnancies"46 (USA) Chronic pain and fatigue can occur after stillbirth 7% "3 months after the loss both mothers and fathers responded to grief most usually with tears; men also reacted with anger, irritation, silence, and one mother reacted most frequently with physical pain"39 (Sweden) Some parents described parental pride after the birth of their stillborn baby 6% "Even though it wasn't the outcome I wanted, I loved giving birth to my son. It was a beautiful experience and how I wanted it."33 (Australia) "Virtually every mother in this study felt tenderness and warmth when they held their baby...this supports the belief that the mother attaches to her new-born even if the baby is dead"56 (Sweden) "There were parents who described a surging feeling of love from the moment they saw their child"52 (Japan) Employment diffi culties and fi nancial debt are potential eff ects of stillbirth 6% "Together with sustained diffi culties in paying off hospital bills, this strained relations with family members from whom funds had been borrowed"53 (Benin) Stillbirth can motivate parents to engage with health-care improvement, including public awareness 4% "I deal with it in a way that you know, to crusade, to campaign, to make sure things change, to try and take the positives as much as you can out of the whole situation"49 (UK) Increased substance use has been reported for some parents 4% "They always were social drinkers but after Ricky died Mom increased her drinking"57 (USA) Women might develop a complex emotional response to body image 4% "Some women also found that their own body reminded them of their loss...their body was a bearer of both pain and memories. They could feel intense pain in their body, feel physically exhausted and sense that their body was against them"45 (Norway) "Women were embarrassed/guilty of their post-pregnant body as they did not have a baby, conversely some women wanted to keep their body in a pregnant shape to not let go of the baby"58 (USA) Stillbirth has an adverse eff ect on the wider family 3% "This sense of constrained grieving caused by social discomfort and taboo extended to husbands and grandparents, who were not expected to grieve the loss of a stillborn baby beyond feeling some transient disappointment or sadness for their wife or daughter"59 (USA) For some parents, quality of life might be aff ected in the long term 2% "Women with histories of fetal death seem to have poorer quality of life"12 (Brazil) Some couples experience competing emotional reactions to sexual relationships 1% "Women also wrote somewhat more often than men about increased activity in order to have another child, as well as sex being used for comfort, closeness, and tension reduction"45 (Norway) "Women more frequently reported disturbing images, thoughts and feelings that interfered with sex than did men"45 (Norway) Psychological eff ect of stillbirth on professionals Stillbirth has a powerful psychological eff ect 95% "I think it's possible to experience too much grief in this work"60 (Ireland) Emotional response or distancing 40% "It is a mixture of everything, anxiety, rage, oppression, impotence..."61 (Spain) Trauma 42% "...I had to cut off my emotions to just get through it"48 (USA) Guilt 35% "It shook me to my core"48 (USA) Anger 30% "...you've got anger, huge anger, especially where a mistake has been made or something has been missed"60 (Ireland) Fear 30% "It sort of haunted me for a couple of days...I had some issues falling asleep that night and getting the images out of my head"51 (Australia) Stress 30% ** Anxiety 25% ** Blame 20% ** Depression 20% ** Frustration 15% ** Sadness 15% ** Powerlessness 10% ** Challenge to faith 5% ** Humiliation 5% ** Stillbirth has a professional eff ect 65% "Is this the one that is going to blame you?"62 (USA) Eff ect of litigation 30% "If you...lose a mother or a baby, you will lose your license, your income, your work"63 (USA) Fear of disciplinary action 10% ** Fear of public censure 5% ** Exposure 5% ** Professionals need support 65% "I think what would be helpful...is having that debriefi ng time after it's over and not being directly assigned"64 (Canada) Education 30% "...they do not teach you the necessary strategies to provide support in these situations"61 (Spain) Peer support 5% "...we need to support each other and not tear each other down."47 (USA) (Table continues on next page) Series 610 www.thelancet.com Vol 387 February 6, 2016 Clinical Center Department of Bioethics, Fogarty International Center, National Institutes of Health, Bethesda, MD, USA (J Millum PhD); Department of Obstetrics and Gynaecology, University College Cork, Cork University Maternity Hospital, Cork, Ireland (D Nuzum BTh, K O'Donoghue PhD); National Perinatal Epidemiology Unit, Nuffield Department of Population Health, University of Oxford, Oxford, UK (M Redshaw PhD); Health Economics Unit, School of Health and Population Sciences, University of Birmingham, Birmingham, UK (Prof T Roberts PhD); Wellbeing Foundation Africa, Lagos, Nigeria (H E Toyin Saraki LLB); ReaCH group, University of Central Lancashire, Preston, UK (Prof S Downe PhD) Correspondence to: Dr Alexander E P Heazell, Maternal and Fetal Health Research Centre, St Mary's Hospital, Manchester M13 9WL, UK alexander.heazell@manchester. ac.uk See Online for appendix For the OneHealth Tool database see http://www.who. int/workforcealliance/ knowledge/toolkit/10/en For the International Labour Organization database see http://www.ilo.org/global/lang-en/index.htm diffi culties, and fi nancial debt were also reported. Some studies39,53,57 described a longlasting negative eff ect on quality of life. The consequences of a stillbirth were not exclusively negative. Some couples reported feeling closer.39 Parental pride was reported by some parents after contact with their baby.33,52,56 For some, deciding to see or hold their baby brought a sense of fi nality that contributed to the grieving process.70 Some parents engaged in therapeutic activities; seeking solitude, changing their uptake of religious practice, and changing their approach to sexual intercourse or engagement with health promoting activities, work, and social media (table).34,37,40,45,71 Some parents campaigned for, and contributed to, health service improvements to help other families. Many parents changed the way they accessed health-care services, especially in subsequent pregnancies when fathers became more involved.46,55 Eff ect of stillbirth on professionals All 20 studies (19 exclusively from HICs and one with respondents from HICs and LMICs) included in the systematic review of the eff ect on professionals undertaken for this paper (appendix pp 51–53) documented a substantial personal and professional burden for staff involved with caring for families during and after stillbirth. Four themes emerged from the data for staff : psychological eff ects, professional eff ects, need for support, and positive eff ects (table). The psychological eff ect was most frequently reported as somatic, including symptoms of trauma, diminished emotional availability, stress, and aff ective states such as guilt, anger, blame, anxiety, and sadness.59,72–74 The professional eff ect of stillbirth was characterised by fear of litigation and disciplinary action. In one study,75 data from LMICs suggested that professionals attending to a woman who has had a stillbirth could result in loss of livelihood and public humiliation. Most studies (n=13) emphasised the need for further education and professional support for staff , especially in terms of the psychosocial care and communication skills needed after a stillbirth. 11 studies suggested that peer support was valuable, even though this guidance was usually informal. However, an absence of structured institutional and peer support was stressed. Seven studies showed the risk of vicarious traumatic stress, and depressive and psychological symptoms such as guilt, self-blame, self-doubt, and grief. Importantly, those health workers who felt that they had received adequate training in stillbirth care were less likely to report guilt and fear of litigation. In six studies,76–81 staff also reported feeling some positive gains, such as a sense of honour or privilege at being able to support parents experiencing the death of their baby. Some staff cited personal growth and the development of a special bond with parents and staff . In four studies,76–78,82 staff reported more confi dence and comfort, with fewer negative eff ects, when they had more direct clinical experience with stillbirth. These fi ndings suggest that, although mothers, partners, and their families endure most of the eff ects of stillbirth, the event also has a substantial eff ect on healthcare providers. The negative eff ects could be addressed by education, training, and provision of formal and informal support during and after stillbirth, and encouragement of positive experiences of caring for parents after stillbirth. Interventions to maximise wellbeing for bereaved parents and families What works? 43 studies provided evidence on what works to reduce the negative eff ects of stillbirths (appendix pp 56–67). The search strategies for two systematic reviews83,84 of randomised controlled trials that were relevant to our review (one on social support and the other on autopsy) were unable to identify any studies that met their inclusion criteria. No other randomised controlled trials were identifi ed in the 43 studies. No intervention studies were identifi ed for Africa, Asia, or the Middle East. Of the 16 studies that directly assessed interventions, ten included mothers only, one had fathers only, one had parents and care providers, and the remaining four studies included parents or the wider family, or both. Eff ective interventions (in HIC settings) included: families seeing and holding the baby, social support and Frequency eff ect size* Example quotes (country) (Continued from previous page) Institutional support 5% ** Stillbirth can have a positive eff ect 30% "I think having that experience, I've grown as a person."64 (Canada) Benefi t of experience 20% "I feel like I make a diff erence, and if I can ease their pain I am happy"46 (USA) Sense of honour 10% ** Privilege 5% ** Special bond with parents 5% ** Making a diff erence 5% ** Frequency eff ect sizes and representative quotes are shown for each theme. *Frequency eff ect size is the proportion of included reports containing a theme. Table: Thematic sentences derived from meta-synthesis of studies assessing psychological eff ect of stillbirth on parents and on health-care professionals. Series www.thelancet.com Vol 387 February 6, 2016 611 support groups, families making and sharing memories, autopsy, psychological interventions, and interventions with various components.85–90 Professional support to enable parents to share their experiences with others, and social support from family and local social networks were both associated with lower rates of depression and better mental health than those without this support.87 A specifi c psychological intervention91 in Brazil was associated with a range of positive eff ects, fi nding that inclusion of family members in the intervention reinforced network support. A US study87 reported that support groups were associated with signifi cant improvement in scores on the revised Impact of Events Scale. Programmes with many components generally increased parents' satisfaction, with those more satisfi ed reporting less grief.87–89,92 Where measured longitudinally, this eff ect was maintained for up to 2 years. Finnish fathers receiving an intervention with various components reported stronger personal growth and less blame and anger than Finnish fathers who did not receive the intervention.92 The key fi ndings of all included studies (qualitative and quantitative) were mapped to Sarafi no's taxonomy of social support. This system comprises fi ve support elements: tangible, emotional, esteem, informational, and network and belonging (appendix pp 56–67).93 All eff ective interventions, and all qualitative studies of interventions with positive participant responses, included emotional support. Nine studies87,88,90,92,94–98 included informational support and ten addressed tangible support. Usually, this tangible support was help from staff to see and hold the baby after birth (14 studies, including HICs and LMICs). Two studies85,90 included esteem support, such as helping parents to reclaim a lost sense of motherhood or fatherhood. Eight studies85,88–92,94,99 were associated with networking and belonging. Positive staff attitude was universally appreciated. Data pertaining to specifi c groups of people were reported only from HICs. This fi nding showed that fathers, siblings, and female partners need to be acknowledged and included in interventions, to mitigate their experiences of the negative eff ects of stillbirth. Interventions for siblings need to be tailored for their age and maturity. The need for esteem support for family members was particularly apparent, including recognition of continuing status as father or co-mother, sister or brother, and grandparent, even after the death of the baby that created these social roles. Variation in access to what works by cultural context Access to support groups or services is not equitable. In the three surveys14,30 (panel 2) on parents that were analysed in this paper, 54–93% of parents in HICs were given information about support groups or services compared with 12% of parents in MICs. Information about grief and psychological symptoms (16% in MICs vs 52% in HICs) was given less frequently in MICs than for physical symptoms (28% vs 47%), but this was not the case in HICs. The perceived eff ectiveness of support groups varied, but 77% of respondents to the ISA survey who used a group reported benefi t. Lower amounts of support available for parents in MICs might account for a greater proportion of parents rating their follow-up care as poor compared with HICs (60% vs 38%; appendix p 69). In the systematic review of what works for mitigating the negative consequences of stillbirths, eight of ten studies in LMICs included only women. The only positive factors reported by respondents from Malawi were basic physical care and brief information giving from nurses, which were seen as surprising but welcome occurrences.100 Studies in Tanzania,64 Ethiopia,51 and India,68 suggested that having a stillborn baby can lead to maternal abuse, social abandonment, and divorce. Despite feelings of grief and loss, mourning in these countries was actively discouraged and suppressed, and interventions such as families seeing and holding the baby and taking mementoes, were not culturally acceptable. This situation was echoed in care providers' responses to the ISA survey (LMIC n=117, HIC n=2020), which reported that parents in LMICs were less likely than those in HICs to be off ered contact with their baby (35% in LMICs vs 94% in HICs), the opportunity to see and hold their baby (42% in LMICs vs 95% HICs), make memories (35% in LMICs vs 87% in HICs), and name their baby (39% in LMICs vs 83% in HICs) after a stillbirth. The main support mechanisms reported in the included LMIC studies were family and local religious communities, rather than health-care professionals and wider society as noted in HICs. In these contexts, interventions designed to improve emotional and informational support might depend on enhancement of community esteem for those who have had a stillbirth, especially through key religious groups. Networking and belonging support interventions could be primary mechanisms for improving women's wellbeing after a stillbirth in LMICs. Summary of what works On the basis of these data, the key element of what works to reduce the eff ects of stillbirth on bereaved parents and families can be summarised as seeing through the eyes of those aff ected. This includes staff who understand what diff erent parents and families need and when they need it; communities that acknowledge grief and loss and do not stigmatise those who have had stillbirths; employers who provide eff ective leave arrangements; and governments that provide tangible support, such as funeral costs, and paid leave from work commitments. The consequences of stillbirth Stillbirth is associated with substantial direct, indirect, psychological, and social costs to women, and to their families, society, and government (fi gure). These Series 612 www.thelancet.com Vol 387 February 6, 2016 include: medical care and investigations at the time of stillbirth and in subsequent pregnancies; funeral costs; grief and negative psychological eff ects; reduced social functioning; family and relationship disruption and breakdown; and negative eff ects on employment. The eff ect of stillbirth is enduring, and can persist for years. Similar issues, particularly direct health care and funeral costs101 and the lasting eff ects on family function have been described for maternal death.61,62,84,102,103 In addition to families, the eff ects on staff and subsequent implications for staff wellbeing and future service quality and delivery must be considered. Depending on the setting, costs might be met by the government, insurance companies, or individuals and their families. Before this Series paper, these various costs of stillbirth have not been considered together. We argue that this situation has led to an underestimation of the economic, social, emotional, and psychological burden of stillbirth. The worldwide eff ect of stillbirth: how to address research gaps Our systematic approach has shown large gaps in available data with respect to costs and interventions that might reduce the burden of stillbirth by preventing these events or their negative consequences. Few studies established the direct costs of stillbirth in the perinatal period or subsequent pregnancies; all studies were from HICs. Studies that reported on the psychological and social costs of stillbirth or practices that might reduce the subsequent negative eff ects are concentrated in HICs (n=177), which have a low-burden of stillbirth, with little or no data available from high-burden LMICs (n=26; appendix p 68). As most components of eff ective care were identifi ed from studies in HICs, the data obtained are similar to a review restricted to only HICs.60 Although some themes are consistent between HICs and LMICs, other factors, such as stigma and social isolation, seem to be particularly relevant in LMICs.47,51,53,63,104,105 Therefore, to appreciate the full cost of stillbirth, tailored research is urgently needed to establish direct, psychological, and social costs of stillbirth, particularly in LMICs and in marginalised women and their families. In all settings, very little information is available about what works for fathers or partners and other family members. Substantial comparative research on eff ective interventions to mitigate the eff ects of stillbirth is missing in all contexts. Where evidence does exist, eff ective care seems to include emotional, informational, and to an extent, tangible support, in terms of practical or fi nancial help, at and around the time of diagnosis and birth. On the basis of questionnaire data, parents greatly valued support to help with direct fi nancial costs (such as funeral arrangements) when it was provided by governments or insurance schemes. Little emphasis is given in intervention studies to networking and belonging support, and almost none to esteem support. In all settings, but particularly in LMICs, these components can form a basis to address stigma, taboos, and social rejection for bereaved mothers. Fear of loss of esteem and of exclusion from social networks has the potential to stifl e attempts to allow women to express and to deal with their grief, potentially leading to longterm costs. By contrast, where local family and social (notably religious) networks were supportive, mothers, in particular, reported positive benefi ts. Likewise, some parents and staff (in both LMICs and HICs) believed that they had grown spiritually, and had gained substantial coping skills as a result of their experience. Figure: The eff ect of stillbirth originating with the death of the baby, aff ecting mother, family, health services, society, and government Widespread themes of direct, indirect, and intangible costs are shown. Government Effect of stillbirth Reduced earnings from employment, maternity and paternity leave, and health-care expenses Clinic Increased health-care costs Negative effect on staff Psychological and emotional distress, and isolation Increased risk of family breakdown Stigma, abandonment, and abuse 2*6 million stillbirths Series www.thelancet.com Vol 387 February 6, 2016 613 Acknowledgment of the personal and professional cost of stillbirth on staff is essential, for their personal wellbeing and to enable health workers to deliver eff ective care to bereaved parents. In LMICs, an intervention that addresses stillbirth at a health-care, societal, and community level could make two major gains. The fi rst could be the adoption of preventive measures, including improved com munication of health messages, monitoring, support and care for women prepregnancy, antenatally, and during delivery, and improving the health of the mother and her baby. The second could be destigmatisation of stillbirth, thereby reducing the negative consequences, especially for women. Interpretation of the cost of stillbirth In view of the research gaps identifi ed, comprehensive estimates of the costs of stillbirth cannot be derived at present to inform cost-eff ectiveness analyses. Data for the fi nancial costs of the sequelae of stillbirth are not routinely collected in any country. Wide variation in monetary and opportunity costs between diff erent countries, such as those relevant to health-care provision or lost labour productivity, mean that such data must be local to be meaningful. Data for the psychological and social costs are also scarce, particularly with regard to LMIC settings, fathers, the wider family, and health-care providers. Finally, any cost-eff ectiveness analysis must include a decision on how the loss of life to the baby is to be measured. Consequently, any attempt to assign a worldwide cost to stillbirth-in monetary terms or with summary measures of health such as QALYs and DALYs-would be misleading at present. Despite the substantial costs of stillbirth set out in this Series paper, the extent of the total loss associated with stillbirth is substantially aff ected by whether the stillbirth is also counted as a loss in its own right (ie, as a loss to the baby). Economic evaluations of interventions to prevent stillbirths have to make the critical decision of whether and how to count this loss. Women's rights and values must be respected, including access to safe termination of pregnancy; however, recognition must also be given to the fact that most women who have had stillbirths had wanted pregnancies. Similar evaluations of interventions to reduce neonatal mortality typically show results based on the time-discounted life expectancy of surviving infants.89 To avoid undervaluation of interventions that106 prevent stillbirth, these controls should be assessed in this same way.107,108 The use of QALYs in guidance by National Institute for Health and Care Excellence and early iterations of DALYs apply discounting techniques to accommodate time-discounting of future benefi ts, such as a longer life, giving 25 QALYs lost or 32 DALYs associated with stillbirth.109,110 The appropriateness of time-discounting of health benefi ts is the subject of debate. Without discounting, stillbirth would be associated with 86 DALYs on account of the loss to the baby. Alternatively, Jamison and colleagues111 suggest that deaths before age 2 years should be adjusted according to extent of cognitive development or so-called acquired life potential. With time-discounting, this adjustment gives stillbirth DALY values of between 5 and 9 years; without time-discounting this fi gure would give DALY values of between 14 years and 26 years. Thus, proposals for how to value the life of a stillborn baby vary greatly. How these babies are valued can make a diff erence of orders of magnitude to the overall loss attributable to stillbirth (appendix p 26). For example, a study110 of the cost-eff ectiveness of a syphilis screening programme for pregnant women in Mwanza City, Tanzania, estimated a cost of $92*56 per DALY averted without including stillbirths and $8*88 per DALY averted if stillbirths were included as a loss to the deceased. Conclusion Despite the gaps in the evidence, the fi ndings in this Series paper suggest that the burden of stillbirths is substantial yet greatly underappreciated. This undervaluation might contribute to the slow pace of change to address stillbirths on national and international platforms, as identifi ed by Frøen and colleagues.112 Crucially, although the costs of stillbirth prevention might seem substantial in LMICs and HICs, the combined direct, indirect, and intangible costs of stillbirth are almost certainly greater still. We call on the global community to recognise the enduring eff ect of stillbirth on parents, families, staff , societies, and health and social care systems, to develop strategies to collect data for the cost of stillbirths and to use that information to invest in strategies, local services, and practices to prevent stillbirth and to invest in interventions to reduce the negative eff ects of stillbirth. Contributors AEPH was responsible for overall coordination and oversight of the Series paper and the writing process. JM and TR modelled values assigned to stillbirth. AEPH and TR were responsible for the systematic review of economic studies, direct costs, and costs in subsequent pregnancies. VF and AMW were responsible for the design and analysis of the international questionnaire. AEPH and MR analysed published questionnaire data. HB searched the International Labour Organization databases. DS and CS were responsible for the systematic review of psychological eff ects on parents. JC, KJG, DN, and KO'D were responsible for the systematic review of psychological eff ects on professionals. SD and OKM were responsible for systematic review of interventions to ameliorate eff ects on parents. ND and HETS helped put the paper into international context. JD, AR, and ZAB were responsible for using the Lives Saved Tool analysis. All named authors contributed to the conceptualisation, development, writing, and fi nalisation of the paper. AEPH is the overall guarantor. The Lancet Ending Preventable Stillbirths Series study group Australia Vicki Flenady (Mater Research Institute, University of Queensland, QLD, Brisbane), Norway J Frederik Frøen (Norwegian Institute of Public Health, Oslo); South Africa Mary V Kinney (Save the Children, Edgemead); Switzerland Luc de Bernis (UN Population Fund, Geneva); UK Joy E Lawn, Hannah Blencowe (London School of Hygiene & Tropical Medicine, London), Alexander E P Heazell (University of Manchester, Manchester); USA Susannah Hopkins Leisher (International Stillbirth Alliance, NJ). Series 614 www.thelancet.com Vol 387 February 6, 2016 The Lancet Ending Preventable Stillbirths investigator group Sweden Ingela Radestad (Sophiahemmet Hogskola, Stockholm); UK Louise Jackson, Chidubem Ogwulu (University of Birmingham, Birmingham), Alison Hills (University of Bristol, Bristol), Stephanie Bradley (North Bristol Primary Care Trust, Bristol), Wendy Taylor, Jayne Budd (University of Manchester, Manchester). Declaration of interests DS received grants from Stillbirth and Neonatal Death charity (Sands), is a member of International Stillbirth Alliance, and on the executive committee of the Stillbirth Clinical Study Group, Department of Health Stillbirth task-and-fi nish groups, and PRactical Obstetric Multi-Professional Training maternity foundation. AEPH is chair of the board International Stillbirth Alliance and on the executive committee of the Stillbirth Clinical Study Group, and Department of Health Stillbirth task-and-fi nish groups. CS received grants from Sands during the conduct of the study. All other authors have no competing interests. Acknowledgments HB received grants from Save the Children/Saving Newborn Lives. DS is a member of Department of Health Stillbirth task-and-fi nish groups, and the PRactical Obstetric Multi-Professional Training maternity foundation. AEPH received grants from Tommy's during the conduct of the paper and was funded by the Holly Martin Stillbirth Research Fund to do qualitative analysis. JM was also supported, in part, by intramural funds from the National Institutes of Health Clinical Center. The views expressed are the author's own and do not represent the position or policy of the US National Institutes of Health, Public Health Service, or the Department of Health and Human Services. This Series paper also reports on independent studies which are part funded by the UK Policy Research Programme in the Department of Health. AEPH is supported by Tommy's and by a Clinician Scientist Fellowship from the UK National Institute of Health Research. This Series paper provides independent research funded by the National Institute for Health Research (NIHR). The views expressed are those of the authors and not necessarily those of the National Health Service, the NIHR, or the Department of Health. KJG receives salary support through a K23 training grant from the National Institutes of Health. None of the funding bodies had any infl uence on the content and scope of the paper. We thank Mater Research Institute, University of Queensland, Australia, for funding the International Stillbirth Alliance questionnaire. We thank Bishal Mohindru for his assistance in the costs analysis for care in subsequent pregnancies. References 1 Lawn JE, Blencowe H, Waiswa P, et al, for The Lancet Ending Preventable Stillbirths Series study group with The Lancet Stillbirth Epidemiology investigator group. Stillbirths: rates, risk factors, and acceleration towards 2030. Lancet 2016; published online Jan 18. http://dx.doi.org/10.1016/S0140-6736(15)00837-5. 2 Sutherland T, Meyer C, Bishai DM, Geller S, Miller S. Community-based distribution of misoprostol for treatment or prevention of postpartum hemorrhage: cost-eff ectiveness, mortality, and morbidity reduction analysis. Int J Gynaecol Obstet 2010; 108: 289–94. 3 Petrou S, Khan K. Economic costs associated with moderate and late preterm birth: primary and secondary evidence. Semin Fetal Neonatal Med 2012; 17: 170–78. 4 Bhutta ZA, Das JK, Bahl R, et al, for The Lancet Newborn Interventions Review Group, The Lancet Every Newborn Study Group. Can available interventions end preventable deaths in mothers, newborn babies, and stillbirths, and at what cost? Lancet 2014; 384: 347–70. 5 Honest H, Forbes CA, Durée KH, et al. Screening to prevent spontaneous preterm birth: systematic reviews of accuracy and eff ectiveness literature with economic modelling. Health Technol Assess 2009; 13: 1–627. 6 Pattinson R, Kerber K, Buchmann E, et al, for the Lancet's Stillbirths Series steering committee. Stillbirths: how can health systems deliver for mothers and babies? Lancet 2011; 377: 1610–23. 7 Bhutta ZA, Yakoob MY, Lawn JE, et al, for the Lancet's Stillbirths Series steering committee. Stillbirths: what diff erence can we make and at what cost? Lancet 2011; 377: 1523–38. 8 Marks JS, Koplan JP, Hogue CJ, Dalmat ME. A cost-benefi t/ cost-eff ectiveness analysis of smoking cessation for pregnant women. Am J Prev Med 1990; 6: 282–89. 9 UNICEF and World Health Organization, 2015. A decade of tracking progress for maternal, newborn and child survival: the 2015 report. http://www.countdown2015mnch.org/ documents/2015Report/Countdown_to_2015-A_Decade_of_ Tracking_Progress_for_Maternal_Newborn_and_Child_SurvivalThe2015Report-Conference_Draft.pdf (accessed on Nov 13, 2015). 10 Christensen H, Trotter CL, Hickman M, Edmunds WJ. Re-evaluating cost eff ectiveness of universal meningitis vaccination (Bexsero) in England: modelling study. BMJ 2014; 349: g5725. 11 Mistry H, Heazell AE, Vincent O, Roberts T. A structured review and exploration of the healthcare costs associated with stillbirth and a subsequent pregnancy in England and Wales. BMC Pregnancy Childbirth 2013; 13: 236. 12 Cooke A, Smith D, Booth A. Beyond PICO: the SPIDER tool for qualitative evidence synthesis. Qual Health Res 2012; 22: 1435–43. 13 Moher D, Liberati A, Tetzlaff J, Altman DG, and the PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. BMJ 2009; 339: b2535. 14 Redshaw M, Rowe R, Henderson J. Listening to parents: after stillbirth or the death of their baby after birth. Oxford: Policy Research Unit in Maternal Health and Care, National Perinatal Epidemiology Unit, 2014. 15 Lacasse JR, Cacciatore J. Prescribing of psychiatric medication to bereaved parents following perinatal/neonatal death: an observational study. Death Stud 2014; 38: 589–96. 16 Ogwulu CB, Jackson LJ, Heazell AE, Roberts TE. Exploring the intangible economic costs of stillbirth. BMC Pregnancy Childbirth 2015; 15: 188. 17 Curtis L. Unit costs of health and social care. 2014. http://www. pssru.ac.uk/project-pages/unit-costs/2014 (accessed May 1, 2015). 18 US Bureau of Labor Statistics. Consumer price index infl ation calculator. 2015. http://www.bls.gov/data/infl ation_calculator.htm (accessed May 2, 2015). 19 Michalski ST, Porter J, Pauli RM. Costs and consequences of comprehensive stillbirth assessment. Am J Obstet Gynecol 2002; 186: 1027–34. 20 Gold KJ, Sen A, Xu X. Hospital costs associated with stillbirth delivery. Matern Child Health J 2013; 17: 1835–41. 21 Reddy UM. Management of pregnancy after stillbirth. Clin Obstet Gynecol 2010; 53: 700–09. 22 Robson S, Thompson J, Ellwood D. Obstetric management of the next pregnancy after an unexplained stillbirth: an anonymous postal survey of Australian obstetricians. Aust N Z J Obstet Gynaecol 2006; 46: 278–81. 23 Weeks JW, Asrat T, Morgan MA, Nageotte M, Thomas SJ, Freeman RK. Antepartum surveillance for a history of stillbirth: when to begin? Am J Obstet Gynecol 1995; 172: 486–92. 24 Heintz E, Brodtkorb TH, Nelson N, Levin LA. The long-term cost-eff ectiveness of fetal monitoring during labour: a comparison of cardiotocography complemented with ST analysis versus cardiotocography alone. BJOG 2008; 115: 1676–87. 25 Vijgen SM, Westerhuis ME, Opmeer BC, et al. Cost-eff ectiveness of cardiotocography plus ST analysis of the fetal electrocardiogram compared with cardiotocography only. Acta Obstet Gynecol Scand 2011; 90: 772–78. 26 Tarricone R. Cost-of-illness analysis. What room in health economics? Health Policy 2006; 77: 51–63. 27 Evans J, Melotti R, Heron J, et al. The timing of maternal depressive symptoms and child cognitive development: a longitudinal study. J Child Psychol Psychiatry 2012; 53: 632–40. 28 Redshaw M, Henderson J. Safely delivered: a national survey of women's experience of maternity care. Oxford: National Perinatal Epidemiology Unit, 2015. 29 Ramchandani PG, Stein A, O'Connor TG, Heron J, Murray L, Evans J. Depression in men in the postnatal period and later child psychopathology: a population cohort study. J Am Acad Child Adolesc Psychiatry 2008; 47: 390–98. 30 Cacciatore J, Lacasse JR, Lietz CA, McPherson J. A parent's tears: primary results from the traumatic experiences and resiliency study. Omega (Westport) 2013–2014; 68: 183–205. 31 Gold KJ, Sen A, Hayward RA. Marriage and cohabitation outcomes after pregnancy loss. Pediatrics 2010; 125: e1202–07. Series www.thelancet.com Vol 387 February 6, 2016 615 32 Rådestad I, Sjögren B, Nordin C, Steineck G. Stillbirth and maternal well-being. Acta Obstet Gynecol Scand 1997; 76: 849–55. 33 Lee C. "She was a person, she was here": The experience of late pregnancy loss in Australia. J Reprod Infant Psychol 2012; 30: 62–76. 34 Kavanaugh K, Hershberger P. Perinatal loss in low-income African American parents. J Obstet Gynecol Neonatal Nurs 2005; 34: 595–605. 35 Côté-Arsenault D, Freije MM. Support groups helping women through pregnancies after loss. West J Nurs Res 2004; 26: 650–70. 36 St John A, Cooke M, Goopy S. Shrouds of silence: three women's stories of prenatal loss. Aust J Adv Nurs 2006; 23: 8–12. 37 Gold KJ, Boggs ME, Mugisha E, Palladino CL. Internet message boards for pregnancy loss: who's on-line and why? Womens Health Issues 2012; 22: e67–72. 38 Tseng YF, Chen CH, Wang HH. Taiwanese women's process of recovery from stillbirth: a qualitative descriptive study. Res Nurs Health 2014; 37: 219–28. 39 Avelin P, Rådestad I, Säfl und K, Wredling R, Erlandsson K. Parental grief and relationships after the loss of a stillborn baby. Midwifery 2013; 29: 668–73. 40 O'Leary J, Thorwick C. Fathers' perspectives during pregnancy, postperinatal loss. J Obstet Gynecol Neonatal Nurs 2006; 35: 78–86. 41 Säfl und K, Sjögren B, Wredling R. The role of caregivers after a stillbirth: views and experiences of parents. Birth 2004; 31: 132–37. 42 McCreight BS. A grief ignored: narratives of pregnancy loss from a male perspective. Sociol Health Illn 2004; 26: 326–50. 43 Erlandsson K, Avelin P, Safl und K, Wredling R, Radestad I. Siblings' farewell to a stillborn sister or brother and parents' support to their older children: a questionnaire study from the parents' perspective. J Child Health Care 2010; 14: 151–60. 44 Hughes P, Turton P, Hopper E, McGauley GA, Fonagy P. Disorganised attachment behaviour among infants born subsequent to stillbirth. J Child Psychol Psychiatry 2001; 42: 791–801. 45 Dyregrov A, Gjestad R. Sexuality following the loss of a child. Death Stud 2011; 35: 289–315. 46 Armstrong D. Exploring fathers' experiences of pregnancy after a prior perinatal loss. MCN Am J Matern Child Nurs 2001; 26: 147–53. 47 Sun HL, Sinclair M, Kernohan GW, Chang TH, Patterson H. Sailing against the tide: Taiwanese women's journey from pregnancy loss to motherhood. MCN Am J Matern Child Nurs 2011; 36: 127–33. 48 Cote-Arsenault D, Donato K. Emotional cushioning in pregnancy after perinatal loss. J Reprod Infant Psychol 2011; 29: 81–92. 49 Murphy S. Reclaiming a moral identity: stillbirth, stigma and 'moral mothers'. Midwifery 2012; 28: 476–80. 50 Hsu MT, Tseng YF, Banks JM, Kuo LL. Interpretations of stillbirth. J Adv Nurs 2004; 47: 408–16. 51 Sisay MM, Yirgu R, Gobezayehu AG, Sibley LM. A qualitative study of attitudes and values surrounding stillbirth and neonatal mortality among grandmothers, mothers, and unmarried girls in rural Amhara and Oromiya regions, Ethiopia: unheard souls in the backyard. J Midwifery Womens Health 2014; 59 (suppl 1): S110–17. 52 Akiko H. The existence of the deceased children in the grieving process of mothers who have experienced stillbirth. Japan Midwifery J 2009; 23: 59–71 (in Japanese). 53 Fottrell E, Kanhonou L, Goufodji S, et al. Risk of psychological distress following severe obstetric complications in Benin: the role of economics, physical health and spousal abuse. Br J Psychiatry 2010; 196: 18–25. 54 Warland J, O'Leary J, McCutcheon H, Williamson V. Parenting paradox: parenting after infant loss. Midwifery 2011; 27: e163–69. 55 Hutti MH, Armstrong DS, Myers J. Healthcare utilization in the pregnancy following a perinatal loss. MCN Am J Matern Child Nurs 2011; 36: 104–11. 56 Radestad I, Safl und K, Wredling R, Onelov E, Steineck G. Holding a stillborn baby: mothers' feelings of tenderness and grief. Br J Midwifery 2009; 17: 178–80. 57 O'Leary J, Warland J. Untold stories of infant loss: the importance of contact with the baby for bereaved parents. J Fam Nurs 2013; 19: 324–47. 58 Huberty JL, Coleman J, Rolfsmeyer K, Wu S. A qualitative study exploring women's beliefs about physical activity after stillbirth. BMC Pregnancy Childbirth 2014; 14: 26. 59 Kelley MC, Trinidad SB. Silent loss and the clinical encounter: Parents' and physicians' experiences of stillbirth-a qualitative analysis. BMC Pregnancy Childbirth 2012; 12: 137. 60 Peters M, Riitano D, Lisy K, Jordan Z, Aromataris E. Providing care for families who have experienced stillbirth: a comprehensive systematic review. Adelaide: Joanna Briggs Institute, 2014. 61 Molla M, Mitiku I, Worku A, Yamin A. Impacts of maternal mortality on living children and families: a qualitative study from Butajira, Ethiopia. Reprod Health 2015; 12 (suppl 1): S6. 62 Pande R, Ogwang S, Karuga R, et al. Continuing with "...a heavy heart"-consequences of maternal death in rural Kenya. Reprod Health 2015; 12 (suppl 1): S2. 63 Gausia K, Moran AC, Ali M, Ryder D, Fisher C, Koblinsky M. Psychological and social consequences among mothers suff ering from perinatal loss: perspective from a low income country. BMC Public Health 2011; 11: 451. 64 Haws RA, Mashasi I, Mrisho M, Schellenberg JA, Darmstadt GL, Winch PJ. "These are not good things for other people to know": how rural Tanzanian women's experiences of pregnancy loss and early neonatal death may impact survey data quality. Soc Sci Med 2010; 71: 1764–72. 65 Hogue CJ, Parker CB, Willinger M, et al, for the Eunice Kennedy Shriver National Institute of Child Health and Human Development Stillbirth Collaborative Research Network Writing Group. The association of stillbirth with depressive symptoms 6–36 months post-delivery. Paediatr Perinat Epidemiol 2015; 29: 131–43. 66 Mills TA, Ricklesford C, Cooke A, Heazell AE, Whitworth M, Lavender T. Parents' experiences and expectations of care in pregnancy after stillbirth or neonatal death: a metasynthesis. BJOG 2014; 121: 943–50. 67 Forhan M. Doing, being, and becoming: a family's journey through perinatal loss. Am J Occup Ther 2010; 64: 142–51. 68 Roberts LR, Montgomery S, Lee JW, Anderson BA. Social and cultural factors associated with perinatal grief in Chhattisgarh, India. J Community Health 2012; 37: 572–82. 69 Sun JC, Rei W, Sheu SJ. Seeing or not seeing: Taiwan's parents' experiences during stillbirth. Int J Nurs Stud 2014; 51: 1153–59. 70 Kingdon C, Givens JL, O'Donnell E, Turner M. Seeing and holding baby: systematic review of clinical management and parental outcomes after stillbirth. Birth 2015; 42: 206–18. 71 Murphy SL. Finding the positive in loss: stillbirth and its potential for parental empowerment. Bereave Care 2012; 31: 98–103. 72 Farrow VA, Goldenberg RL, Fretts R, Schulkin J. Psychological impact of stillbirths on obstetricians. J Matern Fetal Neonatal Med 2013; 26: 748–52. 73 Gold KJ, Kuznia AL, Hayward RA. How physicians cope with stillbirth or neonatal death: a national survey of obstetricians. Obstet Gynecol 2008; 112: 29–34. 74 Nuzum D, Meaney S, O'Donoghue K. The impact of stillbirth on consultant obstetrician gynaecologists: a qualitative study. BJOG 2014; 121: 1020–28. 75 McCool W, Guidera M, Stenson M, Dauphinee L. The pain that binds us: midwives' experiences of loss and adverse outcomes around the world. Health Care Women Int 2009; 30: 1003–13 76 Liisa AA, Marja-Terttu T, Paivi AK, Marja K. Health care personnel's experiences of a bereavement follow-up intervention for grieving parents. Scand J Caring Sci 2011; 25: 373–82. 77 McKenna L, Rolls C. Undergraduate midwifery students' fi rst experiences with stillbirth and neonatal death. Contemp Nurse 2011; 38: 76–83. 78 Steen SE. Perinatal death: bereavement interventions used by US and Spanish nurses and midwives. Int J Palliat Nurs 2015; 21: 79–86. 79 Jonas-Simpson C, McMahon E, Watson J, Andrews L. Nurses' experiences of caring for families whose babies were born still or died shortly after birth. Int J Hum Caring 2010; 14: 14–21. 80 Puia DM, Lewis L, Beck CT. Experiences of obstetric nurses who are present for a perinatal loss. J Obstet Gynecol Neonatal Nurs 2013; 42: 321–31. 81 Roehrs C, Masterson A, Alles R, Witt C, Rutt P. Caring for families coping with perinatal loss. J Obstet Gynecol Neonatal Nurs 2008; 37: 631–39. 82 Roehrs C, Masterson A, Alles R, Witt C, Rutt P. Caring for families coping with perinatal loss. J Obstet Gynecol Neonatal Nurs 2008; 37: 631–39. Series 616 www.thelancet.com Vol 387 February 6, 2016 83 Horey D, Flenady V, Heazell AE, Khong TY. Interventions for supporting parents' decisions about autopsy after stillbirth. Cochrane Database Syst Rev 2013; 2: CD009932. 84 Koopmans L, Wilson T, Cacciatore J, Flenady V. Support for mothers, fathers and families after perinatal death. Cochrane Database Syst Rev 2013; 6: CD000452. 85 Cacciatore J, Schnebly S, Frøen JF. The eff ects of social support on maternal anxiety and depression after stillbirth. Health Soc Care Community 2009; 17: 167–76. 86 Cacciatore J, Rådestad I, Frøen JF. Eff ects of contact with stillborn babies on maternal anxiety and depression. Birth 2008; 35: 313–20. 87 Cacciatore J. Eff ects of support groups on post traumatic stress responses in women experiencing stillbirth. Omega (Westport) 2007; 55: 71–90. 88 Nikkola I, Kaunonen M, Aho AL. Mother's experience of the support from a bereavement follow-up intervention after the death of a child. J Clin Nurs 2013; 22: 1151–62. 89 Bennett SM, Ehrenreich-May J, Litz BT, Boisseau L, Barlow DH. Development and preliminary evaluation of a cognitive-behavioral intervention for perinatal grief. Cogn Behav Pract 2012; 19: 161–73. 90 Crawley R, Lomax S, Ayers S. Recovering from stillbirth: The eff ects of making and sharing memories on maternal mental health. J Reprod Infant Psychol 2013; 31: 195–207. 91 Costa Muza J, Nascimento de Sousa EN, da Rocha Arrais A, Iaconelli V. Quando a morte visita a maternidade: atenção psicológica durante a perda perinatal. Psicol Teor Prat 2013; 15: 34–48. 92 Aho AL, Tarkka MT, Astedt-Kurki P, Sorvari L, Kaunonen M. Evaluating a bereavement follow-up intervention for grieving fathers and their experiences of support after the death of a child-a pilot study. Death Stud 2011; 35: 879–904. 93 Sarafi no EP. Stress, biopsychosocial factors, and illness. Health psychology: biopsychosocial interactions. New York: John Wiley & Sons, 1994. 94 Avelin P, Erlandsson K, Hildingsson I, Radestad I. Swedish parents' experiences of parenthood and the need for support to siblings when a baby is stillborn. Birth 2011; 38: 150–58. 95 Ryninks K, Roberts-Collins C, McKenzie-McHarg K, Horsch A. Mothers' experience of their contact with their stillborn infant: an interpretative phenomenological analysis. BMC Pregnancy Childbirth 2014; 14: 203. 96 Downe S, Schmidt E, Kingdon C, Heazell AE. Bereaved parents' experience of stillbirth in UK hospitals: a qualitative interview study. BMJ Open 2013; 3: e002237. 97 Montesinos A, Roman A, Munoz M, Elias L. Asistencia al duelo neonatal: diez años de experiencia en una Unidad de Neonatología. Rev Chil Pediatr 2013; 84: 650–58. 98 Blood C, Cacciatore J. Parental grief and memento mori photography: narrative, meaning, culture, and context. Death Stud 2014; 38: 224–33. 99 Cacciatore J, Bushfi eld S. Stillbirth: the mother's experience and implications for improving care. J Soc Work End Life Palliat Care 2007; 3: 59–79. 100 Simwaka AN, de Kok B, Chilemba W. Women's perceptions of Nurse-Midwives' caring behaviours during perinatal loss in Lilongwe, Malawi: an exploratory study. Malawi Med J 2014; 26: 8–11. 101 Kes A, Ogwang S, Pande R, et al. The economic burden of maternal mortality on households: evidence from three sub-counties in rural western Kenya. Reprod Health 2015; 12 (suppl 1): S3. 102 Bazile J, Rigodon J, Berman L, et al. Intergenerational impacts of maternal mortality: Qualitative fi ndings from rural Malawi. Reprod Health 2015; 12 (suppl 1): S1. 103 Knight L, Yamin A. "Without a mother": caregivers and community members' views about the impacts of maternal mortality on families in KwaZulu-Natal, South Africa. Reprod Health 2015; 12 (suppl 1): S5. 104 Frøen JF, Cacciatore J, McClure EM, et al, for The Lancet's Stillbirths Series steering committee. Stillbirths: why they matter. Lancet 2011; 377: 1353–66. 105 van der Sijpt E. The unfortunate suff erer: discursive dynamics around pregnancy loss in Cameroon. Med Anthropol 2014; 33: 395–410. 106 LeFevre AE, Shillcutt SD, Waters HR, et al, for the Projahnmo Study Group. Economic evaluation of neonatal care packages in a cluster-randomized controlled trial in Sylhet, Bangladesh. Bull World Health Organ 2013; 91: 736–45. 107 Phillips J, Millum J. Valuing stillbirths. Bioethics 2015; 29: 413–23. 108 Kelley M. Counting stillbirths: women's health and reproductive rights. Lancet 2011; 377: 1636–37. 109 National Collaborating Centre for Women's and Children's Health. Diabetes in pregnancy-Management of diabetes and its complications from preconception to the postnatal period. London: National Institute for Health and Care Excellence, 2015: 579. 110 Terris-Prestholt F, Watson-Jones D, Mugeye K, et al. Is antenatal syphilis screening still cost eff ective in sub-Saharan Africa. Sex Transm Infect 2003; 79: 375–81. 111 Jamison DT, Shahid-Salles SA, Jamison J, Lawn JE, Zupan J. Incorporating deaths near the time of birth into estimates of the global burden of disease. In: Lopez AD, Mathers CD, Ezzati M, et al, eds. Global burden of disease and risk factors. Washington, DC: World Bank, 2006: 427–63. 112 Frøen JF, Friberg IK, Lawn JE, et al, for The Lancet Ending Preventable Stillbirths Series study group. Stillbirths: progress and unfi nished business. Lancet 2016; published online Jan 18. http://dx.doi.org/10.1016/S0140-6736(15)00818-1.