DOI: 10.1111/1471-0528.12656
Systematic review
www.bjog.org
Parents’ experiences and expectations of care in pregnancy after stillbirth or neonatal death: a metasynthesis TA Mills,a C Ricklesford,b A Cooke,a AEP Heazell,b,c M Whitworth,b,c T Lavendera a
School of Nursing, Midwifery and Social Work, The University of Manchester, b Central Manchester University Hospitals NHS Trust, Maternal and Fetal Health Research Group, Institute of Human Development, The University of Manchester, Manchester Academic Health Sciences Centre, Manchester, UK Correspondence: TA Mills, School of Nursing, Midwifery and Social Work, The University of Manchester, Room 4.334 Jean McFarlane Building, Oxford Road, Manchester, M13 9PL, UK. Email [email protected] c
Accepted 6 December 2013. Published Online 4 March 2014.
Background Pregnancy after perinatal death is characterised by
elevated stress and anxiety, increasing the risk of adverse short-term and long-term outcomes. Objectives This metasynthesis aimed to improve understanding of
parents’ experiences of maternity care in pregnancy after stillbirth or neonatal death. Search strategy Six electronic databases were searched using
predefined search terms. Selection criteria English language studies using qualitative
methods to explore the experiences of parents in pregnancy after perinatal loss, were included subject to quality appraisal framework. Data collection and analysis Searches were initiated in December 2011 and finalised in March 2013. Studies were synthesised using an interpretive approach derived from meta-ethnography.
Main results Fourteen studies were included in the synthesis, graded A (no or few flaws, high trustworthiness; n = 5), B (some flaws, unlikely to affect trustworthiness; n = 5) and C (some flaws, possible impact on trustworthiness; n = 4). Three main themes were identified; co-existence of emotions, helpful and unhelpful coping activities and seeking reasssurance through interactions. Conclusion Parents’ experiences of pregnancy are profoundly
altered by previous perinatal death; conflicted emotions, extreme anxiety, isolation and a lack of trust in a good outcome are commonly reported. Emotional and psychological support improves parents’ experiences of subsequent pregnancy, but the absence of an evidence base may limit consistent delivery of optimal care within current services. Keywords Metasynthesis, neonatal death, perinatal loss, stillbirth, subsequent pregnancy.
Please cite this paper as: Mills TA, Ricklesford C, Cooke A, Heazell AEP, Whitworth M, Lavender T. Parents’ experiences and expectations of care in pregnancy after stillbirth or neonatal death: a metasynthesis. BJOG 2014;121:943–950.
Introduction In the UK, around 1 in 200 pregnancies ends in stillbirth.1 Perinatal loss is recognised as precipitating prolonged grief, comparable with any child death.2 However, the majority of women embark on another pregnancy, 50% within a year of the loss.3 Pregnancy after stillbirth or neonatal death is associated with increased maternal anxiety and emotional vulnerability,4 particularly when the inter-pregnancy interval is short.3 This is a concern because elevated maternal stress increases the risk of adverse pregnancy outcomes, notably preterm birth and low birthweight.5 In addition, a recent UK longitudinal study reported that negative psychological impacts of perinatal loss persisted far beyond the next pregnancy and despite the birth of a
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healthy child.6 This might explain the association between perinatal loss, disrupted maternal attachment and parenting difficulties;7 which increase the risk of long-term psychological and social morbidity. Recent evidence suggests that these effects are not confined solely to mothers and that fathers also suffer emotional distress during subsequent pregnancies.8 Due to the increased risks of recurrence,9,10 the Royal College of Obstetricians and Gynaecologists (RCOG)11 recommends obstetric-led care and birth in a specialist maternity unit for the majority of women pregnant after a stillbirth. Guidance acknowledges the potential for psychological distress but provides little practical advice on how professionals should support parents.11 In the absence of a defined pathway, provision is likely to vary according
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to the interest and expertise of individual clinicians. Whether current services meet parents’ psychological and emotional needs in subsequent pregnancies is unclear; exploring their experiences and expectations could provide insight. Therefore, we conducted a metasynthesis of the qualitative research surrounding care during pregnancies after perinatal loss. Metasynthesis is gaining popularity as a method to advance understanding of a phenomenon through critical reinterpretation of isolated qualitative studies, across a body of literature.12 This study aimed to improve understanding of parents’ experiences of maternity care during pregnancy after perinatal loss and the implications for developing services to meet their needs.
Methods Search strategy A systematic search strategy was developed based on a preliminary scoping search of studies surrounding parents’ experiences of pregnancy after perinatal loss, published in English using recognised qualitative methods. Search terms were formulated based on an interpretation of the population/problem of interest, intervention and context (or PICO) framework.13 Initial scoping identified a lack of UK research; so the search was not restricted by country of origin. No limit of start date was imposed, as no key time-point was relevant. Searches of the databases Cumulative Index to Nursing and Allied Health (CINAHL) plus, Medline, EMBASE, PsycINFO, British Nursing Index and ProQuest were initiated in December 2011 and repeated in March 2013. The search strategy in CINAHL is illustrated in Figure 1. As retrieval of qualitative research using databases alone is limited by issues with indexing,14 papers meeting the inclusion criteria were also citation searched.
S1
TX wom* OR “health professional*” OR midwi* OR obstetric* OR “skilled birth attend*” OR “birth attend*” OR famil* OR parent* OR mother* OR father* OR sibling* OR brother* OR sister*
S2
TX (stillb* OR still b* OR “neonat* death” OR “perinatal loss*” OR “pregnan* loss*” OR loss* OR “perinatal death”) AND TX (“next pregnan*” OR “following pregnan*” OR “subsequent pregnan*”)
S3
Quality appraisal As the quality of included papers critically influences the credibility of a metasynthesis, prior quality assessment is crucial.15,16 An appraisal tool devised by Walsh and Downe16 incorporating a checklist (see Table S1) and grading system as described by Downe et al.17 (Table 1) was used. This tool focused on assessment of integrity, transparency and transferability to evaluate the rigour of studies. Two authors independently ranked papers with a quality grade between ‘A’ and ‘D’; papers graded ‘D’ were automatically excluded, whereas those graded ‘A’ or ‘B’ were included. Papers graded ‘C’ were discussed between the wider research team to reach consensus regarding inclusion or exclusion.
Synthesis This metasynthesis was based on an approach derived from meta-ethnography, developed by Noblit and Hare.15 Analysis began with one paper being read and re-read by two authors, independently, to identify and understand the key concepts and themes in the findings. These were then compared with analysis of findings from another paper and the process was repeated across all 14 studies. Synthesis was conducted in two phases, first by seeking similarities in themes and concepts across the studies (reciprocal findings) and then identifying any conflict in findings with the emerging theory (refutational analysis). In the final phase the similarities and differences in the findings were drawn together to develop a line of argument.15
Results Literature search Of the 991 papers identified by initial search, 817 were excluded by irrelevant title. After detailed review of the remaining abstracts, a further 128 papers were excluded (Figure 2), including eight duplicates. Full text review of 46 papers resulted in exclusion of a further 23, which failed to meet the inclusion criteria specified in the search strategy (see Figure 2 for specific reasons). The remaining 23 papers were quality assessed by two authors, those graded ‘C’ were
TX (questionnaire* OR survey* OR interview* OR “focus group*” OR “case stud*” OR observ*) OR TX (view* OR experience* OR opinion* OR attitude* OR perce* OR belie* OR feel* OR know* OR understand*)
Table 1. Quality grading system16,17 A
S4
TX qualitative OR “mixed method*”
S5
S1 AND S2
S6
S3 AND S4
S7
S5 AND S6
C
Key: S = search
D
TX = all text
B
No, or few flaws. The study credibility, transferability, dependability and confirmability are high Some flaws, unlikely to affect the credibility, transferability, dependability and/or confirmability of the study Some flaws that may affect the credibility, transferability, dependability and/or confirmability of the study Significant flaws that are very likely to affect the credibility, transferability, dependability and/or confirmability of the study
Figure 1. Search strategy in CINAHL.
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991 papers
After removal by title (n = 817)
174 papers (abstracts)
After removal of: Duplicates (n = 8), Quantitative/epidemiological papers (n = 24), Review/opinion-based papers (n = 13), Irrelevant (n = 84)
45 papers (full text)
After removal of: Studies related specifically to extended family grandparents, siblings (n = 3), studies related to bereavement/grief only (n = 6), studies related to parenting/‘replacement child’ (n = 5), review/quantitative papers not obvious through abstract (n = 8)
23 papers for quality assessment
After application of quality criteria
14 papers Figure 2. Flow chart of search strategy and outcomes.
assessed by a third author and consensus was reached. Following this process, a further nine papers were excluded because of limited relevance, limited qualitative data or analysis. A summary of the characteristics of the final 14 papers taken forward for synthesis is presented in Table S3.
Findings Papers were published over a 15-year period, from 199618 to 2011,19 reporting studies conducted in four countries: USA,18,20–28 Canada,29–31 Australia29 and Taiwan.19 O’Leary25,26 and O’Leary and Thorwick,27 reported data from one study; as did Cote-Arsenault and co-workers20,21 so 12 studies were represented. Five papers were unpublished dissertations.18,25,28,30,31 The majority (n = 10) focused solely on womens’ experiences;18–24,28,30,31 one study considered fathers alone,27 three included both parents.25,26,29 Two studies examined the impact of specialist support programmes for parents in pregnancy after perinatal loss.22,29 Samples ranged from 4 to 82 participants, consistent with the qualitative approaches used. Losses occurred across a range of gestations from early miscarriage20 to an infant death at 8 weeks old,25,26 and numbered between one and seven per woman. Where ethnicity was reported, participants were
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predominantly white,18,20–24,28,30,31 educational attainment ranged from high school to Masters degree. Data collection methods included interviews,18,19,22–31 focus groups,23,24 observations,22 questionnaires22 and examination of support group ‘artefacts’ (including newsletters, videotapes and professional information).22 Two studies used pregnancy calendars, which included blank spaces for participants to record thoughts and feelings throughout gestation.20,21 Nine subthemes were identified and summarised by three main themes: co-existence of emotions, helpful and unhelpful coping activities and seeking reasssurance through interactions and core concepts. These are discussed with examples of supporting qualitative data presented in Table 2 (further suppporting data is included in Table S2).
Co-existence of emotions Profound ongoing grief and anxiety. The death of their previous baby significantly affected parents’ experiences. Many felt robbed of the ‘normal’ postive feelings they expected18,20–25,28–30 and described how their subsequent pregnancies were characterised by heightened anxiety and fear. As the risk of recurrence is increased after previous perinatal death, it is not surprising that parents were extremely fearful of another loss. However, anxiety and stress were intensified by specific experiences and at particular time-points during subsequent pregnancies.18–31 Women frequently described escalating agitiation and worry as they approached the gestation at which their previous baby had died.19,20,23,28,29 The lead up to routine appointments and ultrasound scans was also associated with increased stress, related to potential for discovery of a problem.19,23,25,26,28 Intense emotional reactions were precipitated by suspicion or confirmation of a complication as the threat to the pregnancy increased.18–20,23 Women repeatedly expressed doubts regarding their physical capacity to maintain a sucessful pregnancy. In a few cases this ‘mistrust’ prompted requests for intervention and even significantly premature delivery. These women sincerely believed that their babies would be safer ‘out than in’.18–20,24,25,28,30 Isolation from family, friends and others. The emotional flux of subsequent pregnancies often led to tensions in personal relationships.18,22,23,29 Within couples, conflict frequently stemmed from divergence in communication of emotions; women tended to be more open about feelings, whereas men often conformed to social expectations and suppressed expression.18,22–25,27,29 Family and friends often assumed that the new pregnancy would allow bereaved parents to ‘get over’ the death of the previous baby, ‘move on’ and be happy and excited about the future.20,22,28,29,31 These unrealistic expectations left parents feeling isolated from their normal support networks.20,22,24,25,28–31
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Table 2. Themes, subthemes and core concept Subthemes
Relevant studies
Profound ongoing grief and anxiety Isolation from family, friends and others Maintaining hope Delaying emotional involvement Seeking control
18–31
Interactions with care providers Interactions with the baby Interactions with technology Specialist support
18–31
18–20, 22, 24,25, 27–29,31 18, 20–25 18–25, 28, 30, 31 18–25, 28, 30, 31
18–22, 24, 25, 28–31 18–28, 30, 31 18, 21, 22, 28–30
Main themes
Example quotation
Co-existence of emotions
I think any death or tragic circumstance affects people strongly. . .but it’s a terrible, terrible thing to lose a baby. It took all the joy out of having a baby. All I did was worry and fear. . .29
Helpful and unhelpful coping activities
I was in denial to others, total denial. No one knew but me, my husband, mother and mother-in-law. . .a friend called who I hadn’t spoken to in a while and said ‘what’s new?’. . .There was no way in hell I would tell her I was pregnant. Finally at 26 weeks I started to tell people.18 My doctor didn’t show up right away, so the resident came in and he asked how many pregnancies I had and I practically blew up in his face READ THE CHART! I said, ‘Look, I’m worried’ and he said ‘What do you have to be worried about?’ and I thought ‘are you from Mars?’ I was furious. . .18 I suppose the best feeling is when I leave the doctor’s office and he’s just checked everything and he says ‘you’re okay’ and then I’m fine. Two days later I’m back again, freaking out! ‘Are you going to check this? . . .you didn’t check this!’23 You can’t be like other people anymore, so that support group helps you with that. . .to feel like that is okay and common, and doesn’t mean that you’re not gonna love your baby when the baby is born, but it’s okay that the pregnancy is not like any pregnancy before.22
Seeking reassurance through interactions
Maintaining hope. Despite fears for the outcome, fostering and maintaining hope was recognised as essential to allow active preparations for the new baby.20–23,25 Parents described how positive feelings accumulated incrementally as specific milestones were successfully negotiated, including fetal viability,20,24 receiving reassurance from antenatal screening20 and progression beyond the gestation of the previous loss.29,30 Despite their worries, a few women expressed determination to enjoy their pregnancies.18,28
Helpful and unhelpful coping activities Delaying emotional involvement. Parents employed various strategies to counter the emotional turmoil that they experienced from the early weeks of pregnancy. There was often a reluctance to share the news; some delayed telling even family or close friends until the third trimester.18,19,21– 23,25,28,30,31 Several studies reported that parents also tried to consciously ‘hold back’ from forming an attachment to
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Core concept The impacts of stillbirth or neonatal death on parents’ experiences of subsequent pregnancies are not fully recognised. Maternity care focuses on increased surveillance to minimise the risk of recurrence, but emotional and psychological support may be lacking. Additional targeted support may improve parents’ experiences and pregnancy outcomes.
their baby, in an attempt at self-protection in case of another loss.22–25,30,31 The impetus for these behaviours is understandable; however, there were also potential negative effects. Parents’ accounts demonstrated that secrecy deprived them of social support in the early weeks of pregnancy18,22 and attempts at emotional disengagement were counterproductive.18,25 Seeking control. Parents reported that they were more assertive with health professionals19,21–23,25,28,30,31 in seeking a more active role in the direction of their care than in previous pregnancies.18–22,24,25,28 Several women changed their obstetrician to access particular expertise in high-risk pregnancy.19,23,25,28–30 Seeing a new doctor, in a different hospital, was also motivated by a desire to avoid reawakening of traumatic memories and to clearly differentiate the current pregnancy from their previous experience.19,21,22,24–26,28
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A number of women felt that they had missed warning signs related to the previous baby’s death and were more vigilant for development of any complication in this pregnancy.19–21,25,28,30 Although women derived some comfort from self-monitoring, these activities also had psychological drawbacks. Analysing the significance of every minor change in pregnancy signs or symptoms actively generated anxiety19,21,25,28,30 and frequently led to additional contacts with care providers to seek reassurance.19,21,25
Seeking reassurance through interactions Interactions with care providers. Although maternity care professionals were perceived as a crucial source of support by parents, the included studies consistently demonstrated that actual contacts often failed to meet expectations.18–31 Parents frequently encountered professionals who were unaware of their history, through lack of access to/or reading of notes before a consultation.18,20,23,26,30 Dismissive attitudes to fears and concerns18–20,23,24,26–31 and insensitive and inappropriate comments sometimes resulted.18–20,23,24,26–31 These often remained with parents long after the event. In contrast, emotional wellbeing was enhanced when care providers demonstrated empathy, listened to concerns and committed to a collaborative and supportive relationship.18,20,22–25,27–29,31 Parents valued direct acknowledgment of the baby who had died,22–24,28,29 including using his or her name.23,24,28–30 Flexible antenatal care including extra appointments, routinely or on request, was also welcomed. The lack of appropriate antenatal education and preparation for parenting were raised in several studies. Standard antenatal classes were often perceived as problematic; bereaved parents particularly expressed discomfort at the prospect of disclosing previous experiences and discussing their baby’s death. Concerns related not only to personal impact of reliving severe trauma, but also to potential effects on other parents-to-be, who were unlikely to have been exposed to poor outcomes.28–30 As a result, some women admitted they concealed the number of children they had in group discussions and several stopped attending sessions altogether.28,30 Reflecting the desire for a greater control over the direction of care, women often expressed a desire to plan the birth at an early stage of pregnancy. This occasionally led to conflict with professionals who preferred to delay making concrete arrangements until nearer term.22,29–31 Most women were offered and accepted induction of labour or elective caesarean delivery before term,18,19,30 a few decided to await spontaneous labour.29,30 Two women planned homebirths,28,30 one to avoid attending the same hospital where the stillbirth had occurred.30
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Interactions with the baby. Women reported being more aware of fetal behaviour during subsequent pregnancies25,27,28,30 and particularly vigilant in monitoring fetal movements, as tangible evidence of wellbeing.18–31 They also found it increasingly difficult to distance themselves from the pregnancy after the onset of fetal activity. Women experienced cycles of fear and relief, related to changes in the frequency of movements.18,19,21,25,28–30 Insomnia and disturbed sleep, often as a result of waking purposely to ensure movements were still present, were also common.18,25,30 Fathers’ accounts demonstrated that they also had a more watchful attitude compared with previous pregnancies, and sought regular reassurance of fetal wellbeing.25,27 Interactions with technology. Obstetric technologies were widely used to provide objective evidence of fetal wellbeing. Due to their high-risk status, women generally had more frequent ultrasound scans compared with their previous pregnancy.18–20,22,25,26,30,31 Despite increased anxiety before scans, normal findings were reassuring. Where the previous baby’s death was related to abnormalities or other features that could be visualised, normal ultrasound appearances increased optimism for a positive outcome.22 However, for many women reassurance derived from ultrasound was surprisingly short-lived.19,23,25–28 Several studies identified ultrasound examinations as a source of extreme anxiety for some women, because of the direct association with the confirmation of the death of their previous baby. The darkened environment of the room and the wait for information was central to their experience, and every subsequent scan reawakened traumatic memories.18,22,25,26,30 The experience of ultrasound was improved when scans were performed by a known professional, and by simple measures such as allowing parents to hear the fetal heartbeat at the start.22 Despite the reservations of professionals regarding efficacy and safety, several parents acquired hand-held Doppler probes enabling fetal heart auscultation at home.21,24,25,30 Many women also had regular cardiotocographic assessment in late gestation; extra monitoring was generally viewed positively.20,30 Specialist support. Nurse/Social Worker and Midwife-led specialist support programmes in the USA,22 Canada and Australia29 were evaluated in two studies. These provided additional support including telephone access to the co-ordinators, regular support group meetings, preparation for birth, parent education and postnatal support. The Special Delivery Service (SDS; Canada and Australia) included 24-hour telephone access to midwives, antenatal care and support during labour if required.29 Almost all participants in the US groups were women; men participated alongside
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their partners in the Canadian and Australian programmes. These groups were evaluated very positively, providing refuge to help parents ‘survive’ subsequent pregnancies. Peer support, empathy and the safe environment provided by groups were highly valued by parents. They were able to discuss experiences openly, referring to the baby who had died, which helped to work through ongoing grief. Group support was empowering and encouraged parents to be assertive with care providers to ensure their needs were met.22 Couples who attended together reported better understanding of how gender affected the expression and communication of emotions, and that this promoted dialogue enabling effective mutual support. The only reported disadvantage of participation in support groups was raised awareness of other complications associated with poor pregnancy outcomes, which was a potential source of anxiety for some.22
Line of argument synthesis In the final stage of analysis findings were combined to represent our interpretation, through a line of argument: Stillbirth or neonatal death profoundly alters the reality of subsequent pregnancies. To ‘survive’ the mire of anxiety and fears, parents frequently delay emotional engagement with their new pregnancy/baby. Misunderstanding of this self-protective behaviour and cultural misconceptions which underplay the lasting effects of perinatal loss deprive parents of support via traditional social networks. Well-intentioned health professionals attempt to meet needs by providing additional antenatal appointments and technological surveillance. However, normal antenatal findings provide only limited reassurance. Lack of understanding of the impact of perinatal death reduces the professional’s capacity to provide adequate emotional and psychological support during subsequent pregnancies. Targeted additional support was rated highly in delivering sensitive care.
programmes was perceived to have significant benefits. Our interpretation is outlined in the novel line of argument which emphasises the importance of access to high-quality psychosocial support to previously bereaved parents within maternity services.
Strengths and weaknesses The focus on English-language studies meant that relevant papers published in other languages might have been missed. However, only one non-English-language abstract was identified and following translation it was apparent that the study did not meet the inclusion criteria. None of the included studies was conducted in the UK. Ten of the fourteen were based in the USA, where maternity care is more highly medicalised, even in comparison with other high-resource settings.33 Obstetric dominance, the lesser role of midwives as primary caregivers and a greater reliance on technological interventions may have influenced the findings of this synthesis. Nevertheless, there was considerable resonance in issues raised across studies; Canada and Australia have publicly-funded care with similarities to Europe. Generalisabilty may also be restricted by the lack of low-resource settings and black and minority ethnic participants. Cultural norms and practices significantly impact on bereavement experiences,34 but none of the studies directly addressed ethnic diversity. A further limitation could be the range of losses included; some parents had early miscarriages rather than stillbirths or neonatal death. It seems plausible that grief reactions intensify with advancing gestation, reflecting increased attachment to the fetus.35 However, evidence for a direct correlation between gestation and the level of maternal psychological distress is equivocal.35,36 Reactions to bereavement are acknowledged to be highly personal, therefore experiences of early loss may precipitate similar responses to late losses for some individuals.37
Interpretation
Discussion Main findings This metasynthesis was conducted to enhance understanding of parents’ experiences of pregnancy after stillbirth or neonatal death through exploration of the qualitative literature. As life events, pregnancy and childbearing are generally associated with positive and affirmative emotions.32 In contrast, pregnancy after perinatal death was perceived by parents as ‘a trial to be endured’.30 Emotional stress was exacerbated by a lack of support through informal networks of family and friends or formal structures. Parents regarded contacts with health professionals as their central source of reassurance; but experiences often fell short of expectations. Participation in tailored support
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This metasynthesis highlights the significant impact of previous perinatal loss on parents during subsequent pregnancies. The concept of ‘perinatal bereavement’, encapsulating the unique experiences of parents immediately following pregnancy loss or the death of an infant, was only recognised relatively recently by professionals.38 This phenomenon describes a complex emotional response, most commonly expressed through grief, with no prescribed timeframe.37 Individual experiences are shaped by a multiplicity of internal and external factors and emotions are often intensified or reawakened in subsequent pregnancies.39 Although parents suffer similar anxieties and fears, fathers tend to suppress outward expression, as social norms dictate that their primary role should be to support the mother.40 Misinterpretation of paternal behaviour as
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Parents’ experiences of pregnancy after perinatal death
indifference is a recognised source of discord between bereaved couples. The synthesis also emphasised how common misconceptions, such as the belief that a new pregnancy helps to ‘get over’ grief for a lost child, effectively isolated parents from social support networks, increasing reliance on external and professional intervention. Antenatal appointments and assessments of fetal wellbeing were eagerly anticipated; however, discontent was expressed with aspects of care received. Parents felt ill-prepared for the anxiety generated in advance of appointments, the short duration of reassurance gained from tests and the lack of empathy demonstrated by some professionals. These findings may relate predominantly to US maternity care, the particular framing of birth as potentially pathological and emphasis on technological management.33 However, UK women pregnant after preterm birth, many of whom had experienced perinatal loss, also expressed significant anxiety surrounding investigations.41 Taken together, these findings caution against an over-reliance on technology to provide reassurance in pregnancy after perinatal death and emphasise the need for regular contact and sustained psychological support. Tailored support programmes appeared to improve experiences of subsequent pregnancy for participants. These included several components and the contribution of individual parts to overall success of the intervention was unclear. Continuous access to experienced professional facilitators, who provided multidimensional support through individual and group contacts, was considered important. In addition, benefits were derived from building relationships with other bereaved parents. Peer support in these groups appeared to foster many of the therapeutic factors described by Yalom and Leszcz42 as underlying the success of group approaches. Despite these obvious benefits, a ‘one size fits all’ approach is unlikely to meet the needs of all parents; support groups are most frequently used by affluent and better-educated individuals43 who were over-represented in the included studies. Rajan and Oakley44 identified improved postnatal emotional wellbeing in UK women who received additional one-to-one midwife contact in pregnancy after perinatal loss.
Conclusion This metasynthesis has important implications for professionals providing maternity care to families who have experienced perinatal bereavement. Provision of sensitive and effective care in subsequent pregnancies is dependent on understanding and acknowledging the impact of previous loss. Ensuring adequate access to and opportunities for reading of case notes before consultations would alert professionals to parents’ history and promote appropriate communication. Continuity of care and access
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to known professionals between scheduled visits appears to be particularly important and should be facilitated wherever possible. Professionals should be mindful that women may delay accessing care in an attempt to self protect through emotional disengagement. Given the high levels of psychological distress reported in this group, provision of support in pregnancy after perinatal loss within UK maternity services requires urgent examination. Extending the evidence base could support the development of interventions to reduce psychological morbidity and other adverse outcomes.
Disclosure of interests The authors confirm that they have no interests to declare.
Contribution to authorship TAM and TL conceived the idea for the metasynthesis. CR and AC performed the searches, reviewed the papers, conducted the synthesis and contributed to drafting the article. TAM reviewed the papers, conducted the synthesis and drafted the final article. TL, AEH and MW conducted the synthesis and revised the article for important intellectual content.
Details of ethics approval This was a literature review; therefore no ethical approval was necessary.
Funding This study was funded by Tommy’s.
Acknowledgement The authors would like to thank Claire Storey for her comments and suggestions.
Supporting Information Additional Supporting Information may be found in the online version of this article: Table S1. Grading criteria. Table S2. Themes and subthemes. Table S3. Characteristics of studies included after quality review. &
References 1 Office for National Statistics. Births and Deaths in England and Wales, 2010. Newport, UK: ONS, 2011. 2 Barkway P. Perinatal Death: A Phenomenological Study of Bereaved Parents’ Experience. Adelaide, SA, Australia: Flinders University of South Australia, 1997. 3 Hughes PM, Turton P, Evans CD. Stillbirth as risk factor for depression and anxiety in the subsequent pregnancy: cohort study. BMJ 1999;318:1721–4.
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4 Armstrong D, Hutti M. Pregnancy after perinatal loss: the relationship between anxiety and prenatal attachment. J Obstet Gynecol Neonatal Nurs 1998;27:183–9. 5 Mulder EJ, Robles de Medina PG, Huizink AC, Van den Bergh BR, Buitelaar JK, Visser GH. Prenatal maternal stress: effects on pregnancy and the (unborn) child. Early Hum Dev 2002;70:3–14. 6 Blackmore ER, Cote-Arsenault D, Tang W, Glover V, Evans J, Golding J, et al. Previous prenatal loss as a predictor of perinatal depression and anxiety. Br J Psychiatry 2011;198:373–8. 7 Warland J, O’Leary J, McCutcheon H, Williamson V. Parenting paradox: parenting after infant loss. Midwifery 2011;27:e163–9. 8 Turton P, Badenhorst W, Hughes P, Ward J, Riches S, White S. Psychological impact of stillbirth on fathers in the subsequent pregnancy and puerperium. Br J Psychiatry 2006;188:165–72. 9 Bhattacharya S, Prescott GJ, Black M, Shetty A. Recurrence risk of stillbirth in a second pregnancy. BJOG 2010;117:1243–7. 10 Getahun D, Lawrence JM, Fassett MJ, Strickland D, Koebnick C, Chen W, et al. The association between stillbirth in the first pregnancy and subsequent adverse perinatal outcomes. Am J Obstet Gynecol 2009;201:e1–6. 11 Royal College of Obstetricians and Gynaecologists. Late Intrauterine Fetal Death and Stillbirth (Green-top 55), 1st edn. London: RCOG, 2010. 12 Jensen LA, Allen MN. Meta-synthesis of qualitative findings. Qual Health Res 1996;6:553–60. 13 Cooke A, Smith D, Booth A. Beyond PICO: the SPIDER tool for qualitative evidence synthesis. Qual Health Res 2012;22:1435–43. 14 Shaw RL, Booth A, Sutton AJ, Miller T, Smith JA, Young B, et al. Finding qualitative research: an evaluation of search strategies. BMC Med Res Methodol 2004;4:5. 15 Noblit GW, Hare RD. Meta-Ethnography: Synthesizing Qualitative Studies. Thousand Oaks, CA: Sage, 1988. 16 Walsh D, Downe S. Appraising the quality of qualitative research. Midwifery 2006;22:108–19. 17 Downe S, Finlayson K, Walsh D, Lavender T. ‘Weighing up and balancing out’: a meta-synthesis of barriers to antenatal care for marginalised women in high-income countries. BJOG 2009;116:518–29. 18 Morton PA. Perinatal Loss and the Replacement Child: The Emotional Limits of Reproductive Technology. New York, NY: City University of New York, 1996. 19 Sun HL, Sinclair M, Kernohan GW, Chang TH, Paterson H. Sailing against the tide: Taiwanese women’s journey from pregnancy loss to motherhood. MCN Am J Matern Child Nurs 2011;36:127–33. 20 Cote-Arsenault D, Donato KL. Restrained expectations in late pregnancy following loss. J Obstet Gynecol Neonatal Nurs 2007;36:550–7. 21 Cote-Arsenault D, Donato KL, Earl SS. Watching and worrying: early pregnancy after loss experiences. MCN Am J Matern Child Nurs 2006;31:356–63. 22 Cote-Arsenault D, Freije MM. Support groups helping women through pregnancies after loss. West J Nurs Res 2004;26:650–70. 23 Cote-Arsenault D, Marshall R. One foot in-one foot out: weathering the storm of pregnancy after perinatal loss. Res Nurs Health 2000;23:473–85.
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24 Cote-Arsenault D, Morrison-Beedy D. Women’s voices reflecting changed expectations for pregnancy after perinatal loss. J Nurs Scholarsh 2001;33:239–44. 25 O’Leary J. The Meaning of Parenting During Pregnancy: A Descriptive Phenomenological Study of Parenting a Subsequent Baby Following a Perinatal Loss. Minneapolis, MN: University of Minnesota, 2002. 26 O’Leary J. The trauma of ultrasound during a pregnancy following perinatal loss. J Loss Trauma 2005;10:183–204. 27 O’Leary J, Thorwick C. Fathers’ perspectives during pregnancy, postperinatal loss. J Obstet Gynecol Neonatal Nurs 2006;35:78–86. 28 Thebert-Wright C. Women’s Experiences with Coping After Late Pregnancy Loss: A Qualitative Study. Kalamazoo, MI: Western Michigan University, 2002. 29 Caelli K, Downie J, Letendre A. Parents’ experiences of midwife-managed care following the loss of a baby in a previous pregnancy. J Adv Nurs 2002;39:127–36. 30 Keith PM. Womens’ Experiences of Livebirth After Stillbirth. Vancouver, BC, Canada: University of British Columbia, 2005. 31 Simmons H. The Experience of ‘High-Risk’ Pregnancy Following Perinatal Loss. Halifax, NS, Canada: Dalhousie University, 2007. 32 Geller PA. Pregnancy as a stressful life event. CNS Spectr 2004;9:188–97. 33 van Teijlingen E, Wrede S, Benoit C, Sandall J, DeVries R. Born in the USA: exceptionalism in maternity care organisation among high income countries. Sociol Res Online 2009;14:1–19. 34 Hebert MP. Perinatal bereavement in its cultural context. Death Stud 1998;22:61–78. 35 Klier CM, Geller PA, Ritsher JB. Affective disorders in the aftermath of miscarriage: a comprehensive review. Arch Womens Ment Health 2002;5:129–49. 36 Brier N. Grief following miscarriage: a comprehensive review of the literature. J Womens Health (Larchmt) 2008;17:451–64. 37 Fenstermacher K, Hupcey JE. Perinatal bereavement: a principle-based concept analysis. J Adv Nurs 2013;69:2389–400. 38 Kennell JH, Slyter H, Klaus MH. The mourning response of parents to the death of a newborn infant. N Engl J Med 1970;283:344–9. 39 Cote-Arsenault D. Pregnancy after loss. J Obstet Gynecol Neonatal Nurs 1999;28:13–14. 40 de Montigny F, Beaudet L, Dumas L. A baby has died: the impact of perinatal loss on family social networks. J Obstet Gynecol Neonatal Nurs 1999;28:151–6. 41 O’Brien ET, Quenby S, Lavender T. Women’s views of high risk pregnancy under threat of preterm birth. Sex Reprod Healthc 2010;1:79–84. 42 Yalom I, Leszcz M. The Theory and Practice of Group Psychotherapy, 5th edn. New York, NY: Basic Books, 2005. 43 De Frain J. Learning about grief from normal families: SIDS, stillbirth, and miscarriage. J Marital Fam Ther 1991;17:215–32. 44 Rajan L, Oakley A. No pills for the heartache. The importance of social support for women who suffer pregnancy loss. J Reprod Infant Psychol 1993;11:75–87.
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Systematic review
www.bjog.org
Parents’ experiences and expectations of care in pregnancy after stillbirth or neonatal death: a metasynthesis TA Mills,a C Ricklesford,b A Cooke,a AEP Heazell,b,c M Whitworth,b,c T Lavendera a
School of Nursing, Midwifery and Social Work, The University of Manchester, b Central Manchester University Hospitals NHS Trust, Maternal and Fetal Health Research Group, Institute of Human Development, The University of Manchester, Manchester Academic Health Sciences Centre, Manchester, UK Correspondence: TA Mills, School of Nursing, Midwifery and Social Work, The University of Manchester, Room 4.334 Jean McFarlane Building, Oxford Road, Manchester, M13 9PL, UK. Email [email protected] c
Accepted 6 December 2013. Published Online 4 March 2014.
Background Pregnancy after perinatal death is characterised by
elevated stress and anxiety, increasing the risk of adverse short-term and long-term outcomes. Objectives This metasynthesis aimed to improve understanding of
parents’ experiences of maternity care in pregnancy after stillbirth or neonatal death. Search strategy Six electronic databases were searched using
predefined search terms. Selection criteria English language studies using qualitative
methods to explore the experiences of parents in pregnancy after perinatal loss, were included subject to quality appraisal framework. Data collection and analysis Searches were initiated in December 2011 and finalised in March 2013. Studies were synthesised using an interpretive approach derived from meta-ethnography.
Main results Fourteen studies were included in the synthesis, graded A (no or few flaws, high trustworthiness; n = 5), B (some flaws, unlikely to affect trustworthiness; n = 5) and C (some flaws, possible impact on trustworthiness; n = 4). Three main themes were identified; co-existence of emotions, helpful and unhelpful coping activities and seeking reasssurance through interactions. Conclusion Parents’ experiences of pregnancy are profoundly
altered by previous perinatal death; conflicted emotions, extreme anxiety, isolation and a lack of trust in a good outcome are commonly reported. Emotional and psychological support improves parents’ experiences of subsequent pregnancy, but the absence of an evidence base may limit consistent delivery of optimal care within current services. Keywords Metasynthesis, neonatal death, perinatal loss, stillbirth, subsequent pregnancy.
Please cite this paper as: Mills TA, Ricklesford C, Cooke A, Heazell AEP, Whitworth M, Lavender T. Parents’ experiences and expectations of care in pregnancy after stillbirth or neonatal death: a metasynthesis. BJOG 2014;121:943–950.
Introduction In the UK, around 1 in 200 pregnancies ends in stillbirth.1 Perinatal loss is recognised as precipitating prolonged grief, comparable with any child death.2 However, the majority of women embark on another pregnancy, 50% within a year of the loss.3 Pregnancy after stillbirth or neonatal death is associated with increased maternal anxiety and emotional vulnerability,4 particularly when the inter-pregnancy interval is short.3 This is a concern because elevated maternal stress increases the risk of adverse pregnancy outcomes, notably preterm birth and low birthweight.5 In addition, a recent UK longitudinal study reported that negative psychological impacts of perinatal loss persisted far beyond the next pregnancy and despite the birth of a
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healthy child.6 This might explain the association between perinatal loss, disrupted maternal attachment and parenting difficulties;7 which increase the risk of long-term psychological and social morbidity. Recent evidence suggests that these effects are not confined solely to mothers and that fathers also suffer emotional distress during subsequent pregnancies.8 Due to the increased risks of recurrence,9,10 the Royal College of Obstetricians and Gynaecologists (RCOG)11 recommends obstetric-led care and birth in a specialist maternity unit for the majority of women pregnant after a stillbirth. Guidance acknowledges the potential for psychological distress but provides little practical advice on how professionals should support parents.11 In the absence of a defined pathway, provision is likely to vary according
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to the interest and expertise of individual clinicians. Whether current services meet parents’ psychological and emotional needs in subsequent pregnancies is unclear; exploring their experiences and expectations could provide insight. Therefore, we conducted a metasynthesis of the qualitative research surrounding care during pregnancies after perinatal loss. Metasynthesis is gaining popularity as a method to advance understanding of a phenomenon through critical reinterpretation of isolated qualitative studies, across a body of literature.12 This study aimed to improve understanding of parents’ experiences of maternity care during pregnancy after perinatal loss and the implications for developing services to meet their needs.
Methods Search strategy A systematic search strategy was developed based on a preliminary scoping search of studies surrounding parents’ experiences of pregnancy after perinatal loss, published in English using recognised qualitative methods. Search terms were formulated based on an interpretation of the population/problem of interest, intervention and context (or PICO) framework.13 Initial scoping identified a lack of UK research; so the search was not restricted by country of origin. No limit of start date was imposed, as no key time-point was relevant. Searches of the databases Cumulative Index to Nursing and Allied Health (CINAHL) plus, Medline, EMBASE, PsycINFO, British Nursing Index and ProQuest were initiated in December 2011 and repeated in March 2013. The search strategy in CINAHL is illustrated in Figure 1. As retrieval of qualitative research using databases alone is limited by issues with indexing,14 papers meeting the inclusion criteria were also citation searched.
S1
TX wom* OR “health professional*” OR midwi* OR obstetric* OR “skilled birth attend*” OR “birth attend*” OR famil* OR parent* OR mother* OR father* OR sibling* OR brother* OR sister*
S2
TX (stillb* OR still b* OR “neonat* death” OR “perinatal loss*” OR “pregnan* loss*” OR loss* OR “perinatal death”) AND TX (“next pregnan*” OR “following pregnan*” OR “subsequent pregnan*”)
S3
Quality appraisal As the quality of included papers critically influences the credibility of a metasynthesis, prior quality assessment is crucial.15,16 An appraisal tool devised by Walsh and Downe16 incorporating a checklist (see Table S1) and grading system as described by Downe et al.17 (Table 1) was used. This tool focused on assessment of integrity, transparency and transferability to evaluate the rigour of studies. Two authors independently ranked papers with a quality grade between ‘A’ and ‘D’; papers graded ‘D’ were automatically excluded, whereas those graded ‘A’ or ‘B’ were included. Papers graded ‘C’ were discussed between the wider research team to reach consensus regarding inclusion or exclusion.
Synthesis This metasynthesis was based on an approach derived from meta-ethnography, developed by Noblit and Hare.15 Analysis began with one paper being read and re-read by two authors, independently, to identify and understand the key concepts and themes in the findings. These were then compared with analysis of findings from another paper and the process was repeated across all 14 studies. Synthesis was conducted in two phases, first by seeking similarities in themes and concepts across the studies (reciprocal findings) and then identifying any conflict in findings with the emerging theory (refutational analysis). In the final phase the similarities and differences in the findings were drawn together to develop a line of argument.15
Results Literature search Of the 991 papers identified by initial search, 817 were excluded by irrelevant title. After detailed review of the remaining abstracts, a further 128 papers were excluded (Figure 2), including eight duplicates. Full text review of 46 papers resulted in exclusion of a further 23, which failed to meet the inclusion criteria specified in the search strategy (see Figure 2 for specific reasons). The remaining 23 papers were quality assessed by two authors, those graded ‘C’ were
TX (questionnaire* OR survey* OR interview* OR “focus group*” OR “case stud*” OR observ*) OR TX (view* OR experience* OR opinion* OR attitude* OR perce* OR belie* OR feel* OR know* OR understand*)
Table 1. Quality grading system16,17 A
S4
TX qualitative OR “mixed method*”
S5
S1 AND S2
S6
S3 AND S4
S7
S5 AND S6
C
Key: S = search
D
TX = all text
B
No, or few flaws. The study credibility, transferability, dependability and confirmability are high Some flaws, unlikely to affect the credibility, transferability, dependability and/or confirmability of the study Some flaws that may affect the credibility, transferability, dependability and/or confirmability of the study Significant flaws that are very likely to affect the credibility, transferability, dependability and/or confirmability of the study
Figure 1. Search strategy in CINAHL.
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Parents’ experiences of pregnancy after perinatal death
991 papers
After removal by title (n = 817)
174 papers (abstracts)
After removal of: Duplicates (n = 8), Quantitative/epidemiological papers (n = 24), Review/opinion-based papers (n = 13), Irrelevant (n = 84)
45 papers (full text)
After removal of: Studies related specifically to extended family grandparents, siblings (n = 3), studies related to bereavement/grief only (n = 6), studies related to parenting/‘replacement child’ (n = 5), review/quantitative papers not obvious through abstract (n = 8)
23 papers for quality assessment
After application of quality criteria
14 papers Figure 2. Flow chart of search strategy and outcomes.
assessed by a third author and consensus was reached. Following this process, a further nine papers were excluded because of limited relevance, limited qualitative data or analysis. A summary of the characteristics of the final 14 papers taken forward for synthesis is presented in Table S3.
Findings Papers were published over a 15-year period, from 199618 to 2011,19 reporting studies conducted in four countries: USA,18,20–28 Canada,29–31 Australia29 and Taiwan.19 O’Leary25,26 and O’Leary and Thorwick,27 reported data from one study; as did Cote-Arsenault and co-workers20,21 so 12 studies were represented. Five papers were unpublished dissertations.18,25,28,30,31 The majority (n = 10) focused solely on womens’ experiences;18–24,28,30,31 one study considered fathers alone,27 three included both parents.25,26,29 Two studies examined the impact of specialist support programmes for parents in pregnancy after perinatal loss.22,29 Samples ranged from 4 to 82 participants, consistent with the qualitative approaches used. Losses occurred across a range of gestations from early miscarriage20 to an infant death at 8 weeks old,25,26 and numbered between one and seven per woman. Where ethnicity was reported, participants were
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predominantly white,18,20–24,28,30,31 educational attainment ranged from high school to Masters degree. Data collection methods included interviews,18,19,22–31 focus groups,23,24 observations,22 questionnaires22 and examination of support group ‘artefacts’ (including newsletters, videotapes and professional information).22 Two studies used pregnancy calendars, which included blank spaces for participants to record thoughts and feelings throughout gestation.20,21 Nine subthemes were identified and summarised by three main themes: co-existence of emotions, helpful and unhelpful coping activities and seeking reasssurance through interactions and core concepts. These are discussed with examples of supporting qualitative data presented in Table 2 (further suppporting data is included in Table S2).
Co-existence of emotions Profound ongoing grief and anxiety. The death of their previous baby significantly affected parents’ experiences. Many felt robbed of the ‘normal’ postive feelings they expected18,20–25,28–30 and described how their subsequent pregnancies were characterised by heightened anxiety and fear. As the risk of recurrence is increased after previous perinatal death, it is not surprising that parents were extremely fearful of another loss. However, anxiety and stress were intensified by specific experiences and at particular time-points during subsequent pregnancies.18–31 Women frequently described escalating agitiation and worry as they approached the gestation at which their previous baby had died.19,20,23,28,29 The lead up to routine appointments and ultrasound scans was also associated with increased stress, related to potential for discovery of a problem.19,23,25,26,28 Intense emotional reactions were precipitated by suspicion or confirmation of a complication as the threat to the pregnancy increased.18–20,23 Women repeatedly expressed doubts regarding their physical capacity to maintain a sucessful pregnancy. In a few cases this ‘mistrust’ prompted requests for intervention and even significantly premature delivery. These women sincerely believed that their babies would be safer ‘out than in’.18–20,24,25,28,30 Isolation from family, friends and others. The emotional flux of subsequent pregnancies often led to tensions in personal relationships.18,22,23,29 Within couples, conflict frequently stemmed from divergence in communication of emotions; women tended to be more open about feelings, whereas men often conformed to social expectations and suppressed expression.18,22–25,27,29 Family and friends often assumed that the new pregnancy would allow bereaved parents to ‘get over’ the death of the previous baby, ‘move on’ and be happy and excited about the future.20,22,28,29,31 These unrealistic expectations left parents feeling isolated from their normal support networks.20,22,24,25,28–31
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Table 2. Themes, subthemes and core concept Subthemes
Relevant studies
Profound ongoing grief and anxiety Isolation from family, friends and others Maintaining hope Delaying emotional involvement Seeking control
18–31
Interactions with care providers Interactions with the baby Interactions with technology Specialist support
18–31
18–20, 22, 24,25, 27–29,31 18, 20–25 18–25, 28, 30, 31 18–25, 28, 30, 31
18–22, 24, 25, 28–31 18–28, 30, 31 18, 21, 22, 28–30
Main themes
Example quotation
Co-existence of emotions
I think any death or tragic circumstance affects people strongly. . .but it’s a terrible, terrible thing to lose a baby. It took all the joy out of having a baby. All I did was worry and fear. . .29
Helpful and unhelpful coping activities
I was in denial to others, total denial. No one knew but me, my husband, mother and mother-in-law. . .a friend called who I hadn’t spoken to in a while and said ‘what’s new?’. . .There was no way in hell I would tell her I was pregnant. Finally at 26 weeks I started to tell people.18 My doctor didn’t show up right away, so the resident came in and he asked how many pregnancies I had and I practically blew up in his face READ THE CHART! I said, ‘Look, I’m worried’ and he said ‘What do you have to be worried about?’ and I thought ‘are you from Mars?’ I was furious. . .18 I suppose the best feeling is when I leave the doctor’s office and he’s just checked everything and he says ‘you’re okay’ and then I’m fine. Two days later I’m back again, freaking out! ‘Are you going to check this? . . .you didn’t check this!’23 You can’t be like other people anymore, so that support group helps you with that. . .to feel like that is okay and common, and doesn’t mean that you’re not gonna love your baby when the baby is born, but it’s okay that the pregnancy is not like any pregnancy before.22
Seeking reassurance through interactions
Maintaining hope. Despite fears for the outcome, fostering and maintaining hope was recognised as essential to allow active preparations for the new baby.20–23,25 Parents described how positive feelings accumulated incrementally as specific milestones were successfully negotiated, including fetal viability,20,24 receiving reassurance from antenatal screening20 and progression beyond the gestation of the previous loss.29,30 Despite their worries, a few women expressed determination to enjoy their pregnancies.18,28
Helpful and unhelpful coping activities Delaying emotional involvement. Parents employed various strategies to counter the emotional turmoil that they experienced from the early weeks of pregnancy. There was often a reluctance to share the news; some delayed telling even family or close friends until the third trimester.18,19,21– 23,25,28,30,31 Several studies reported that parents also tried to consciously ‘hold back’ from forming an attachment to
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Core concept The impacts of stillbirth or neonatal death on parents’ experiences of subsequent pregnancies are not fully recognised. Maternity care focuses on increased surveillance to minimise the risk of recurrence, but emotional and psychological support may be lacking. Additional targeted support may improve parents’ experiences and pregnancy outcomes.
their baby, in an attempt at self-protection in case of another loss.22–25,30,31 The impetus for these behaviours is understandable; however, there were also potential negative effects. Parents’ accounts demonstrated that secrecy deprived them of social support in the early weeks of pregnancy18,22 and attempts at emotional disengagement were counterproductive.18,25 Seeking control. Parents reported that they were more assertive with health professionals19,21–23,25,28,30,31 in seeking a more active role in the direction of their care than in previous pregnancies.18–22,24,25,28 Several women changed their obstetrician to access particular expertise in high-risk pregnancy.19,23,25,28–30 Seeing a new doctor, in a different hospital, was also motivated by a desire to avoid reawakening of traumatic memories and to clearly differentiate the current pregnancy from their previous experience.19,21,22,24–26,28
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Parents’ experiences of pregnancy after perinatal death
A number of women felt that they had missed warning signs related to the previous baby’s death and were more vigilant for development of any complication in this pregnancy.19–21,25,28,30 Although women derived some comfort from self-monitoring, these activities also had psychological drawbacks. Analysing the significance of every minor change in pregnancy signs or symptoms actively generated anxiety19,21,25,28,30 and frequently led to additional contacts with care providers to seek reassurance.19,21,25
Seeking reassurance through interactions Interactions with care providers. Although maternity care professionals were perceived as a crucial source of support by parents, the included studies consistently demonstrated that actual contacts often failed to meet expectations.18–31 Parents frequently encountered professionals who were unaware of their history, through lack of access to/or reading of notes before a consultation.18,20,23,26,30 Dismissive attitudes to fears and concerns18–20,23,24,26–31 and insensitive and inappropriate comments sometimes resulted.18–20,23,24,26–31 These often remained with parents long after the event. In contrast, emotional wellbeing was enhanced when care providers demonstrated empathy, listened to concerns and committed to a collaborative and supportive relationship.18,20,22–25,27–29,31 Parents valued direct acknowledgment of the baby who had died,22–24,28,29 including using his or her name.23,24,28–30 Flexible antenatal care including extra appointments, routinely or on request, was also welcomed. The lack of appropriate antenatal education and preparation for parenting were raised in several studies. Standard antenatal classes were often perceived as problematic; bereaved parents particularly expressed discomfort at the prospect of disclosing previous experiences and discussing their baby’s death. Concerns related not only to personal impact of reliving severe trauma, but also to potential effects on other parents-to-be, who were unlikely to have been exposed to poor outcomes.28–30 As a result, some women admitted they concealed the number of children they had in group discussions and several stopped attending sessions altogether.28,30 Reflecting the desire for a greater control over the direction of care, women often expressed a desire to plan the birth at an early stage of pregnancy. This occasionally led to conflict with professionals who preferred to delay making concrete arrangements until nearer term.22,29–31 Most women were offered and accepted induction of labour or elective caesarean delivery before term,18,19,30 a few decided to await spontaneous labour.29,30 Two women planned homebirths,28,30 one to avoid attending the same hospital where the stillbirth had occurred.30
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Interactions with the baby. Women reported being more aware of fetal behaviour during subsequent pregnancies25,27,28,30 and particularly vigilant in monitoring fetal movements, as tangible evidence of wellbeing.18–31 They also found it increasingly difficult to distance themselves from the pregnancy after the onset of fetal activity. Women experienced cycles of fear and relief, related to changes in the frequency of movements.18,19,21,25,28–30 Insomnia and disturbed sleep, often as a result of waking purposely to ensure movements were still present, were also common.18,25,30 Fathers’ accounts demonstrated that they also had a more watchful attitude compared with previous pregnancies, and sought regular reassurance of fetal wellbeing.25,27 Interactions with technology. Obstetric technologies were widely used to provide objective evidence of fetal wellbeing. Due to their high-risk status, women generally had more frequent ultrasound scans compared with their previous pregnancy.18–20,22,25,26,30,31 Despite increased anxiety before scans, normal findings were reassuring. Where the previous baby’s death was related to abnormalities or other features that could be visualised, normal ultrasound appearances increased optimism for a positive outcome.22 However, for many women reassurance derived from ultrasound was surprisingly short-lived.19,23,25–28 Several studies identified ultrasound examinations as a source of extreme anxiety for some women, because of the direct association with the confirmation of the death of their previous baby. The darkened environment of the room and the wait for information was central to their experience, and every subsequent scan reawakened traumatic memories.18,22,25,26,30 The experience of ultrasound was improved when scans were performed by a known professional, and by simple measures such as allowing parents to hear the fetal heartbeat at the start.22 Despite the reservations of professionals regarding efficacy and safety, several parents acquired hand-held Doppler probes enabling fetal heart auscultation at home.21,24,25,30 Many women also had regular cardiotocographic assessment in late gestation; extra monitoring was generally viewed positively.20,30 Specialist support. Nurse/Social Worker and Midwife-led specialist support programmes in the USA,22 Canada and Australia29 were evaluated in two studies. These provided additional support including telephone access to the co-ordinators, regular support group meetings, preparation for birth, parent education and postnatal support. The Special Delivery Service (SDS; Canada and Australia) included 24-hour telephone access to midwives, antenatal care and support during labour if required.29 Almost all participants in the US groups were women; men participated alongside
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their partners in the Canadian and Australian programmes. These groups were evaluated very positively, providing refuge to help parents ‘survive’ subsequent pregnancies. Peer support, empathy and the safe environment provided by groups were highly valued by parents. They were able to discuss experiences openly, referring to the baby who had died, which helped to work through ongoing grief. Group support was empowering and encouraged parents to be assertive with care providers to ensure their needs were met.22 Couples who attended together reported better understanding of how gender affected the expression and communication of emotions, and that this promoted dialogue enabling effective mutual support. The only reported disadvantage of participation in support groups was raised awareness of other complications associated with poor pregnancy outcomes, which was a potential source of anxiety for some.22
Line of argument synthesis In the final stage of analysis findings were combined to represent our interpretation, through a line of argument: Stillbirth or neonatal death profoundly alters the reality of subsequent pregnancies. To ‘survive’ the mire of anxiety and fears, parents frequently delay emotional engagement with their new pregnancy/baby. Misunderstanding of this self-protective behaviour and cultural misconceptions which underplay the lasting effects of perinatal loss deprive parents of support via traditional social networks. Well-intentioned health professionals attempt to meet needs by providing additional antenatal appointments and technological surveillance. However, normal antenatal findings provide only limited reassurance. Lack of understanding of the impact of perinatal death reduces the professional’s capacity to provide adequate emotional and psychological support during subsequent pregnancies. Targeted additional support was rated highly in delivering sensitive care.
programmes was perceived to have significant benefits. Our interpretation is outlined in the novel line of argument which emphasises the importance of access to high-quality psychosocial support to previously bereaved parents within maternity services.
Strengths and weaknesses The focus on English-language studies meant that relevant papers published in other languages might have been missed. However, only one non-English-language abstract was identified and following translation it was apparent that the study did not meet the inclusion criteria. None of the included studies was conducted in the UK. Ten of the fourteen were based in the USA, where maternity care is more highly medicalised, even in comparison with other high-resource settings.33 Obstetric dominance, the lesser role of midwives as primary caregivers and a greater reliance on technological interventions may have influenced the findings of this synthesis. Nevertheless, there was considerable resonance in issues raised across studies; Canada and Australia have publicly-funded care with similarities to Europe. Generalisabilty may also be restricted by the lack of low-resource settings and black and minority ethnic participants. Cultural norms and practices significantly impact on bereavement experiences,34 but none of the studies directly addressed ethnic diversity. A further limitation could be the range of losses included; some parents had early miscarriages rather than stillbirths or neonatal death. It seems plausible that grief reactions intensify with advancing gestation, reflecting increased attachment to the fetus.35 However, evidence for a direct correlation between gestation and the level of maternal psychological distress is equivocal.35,36 Reactions to bereavement are acknowledged to be highly personal, therefore experiences of early loss may precipitate similar responses to late losses for some individuals.37
Interpretation
Discussion Main findings This metasynthesis was conducted to enhance understanding of parents’ experiences of pregnancy after stillbirth or neonatal death through exploration of the qualitative literature. As life events, pregnancy and childbearing are generally associated with positive and affirmative emotions.32 In contrast, pregnancy after perinatal death was perceived by parents as ‘a trial to be endured’.30 Emotional stress was exacerbated by a lack of support through informal networks of family and friends or formal structures. Parents regarded contacts with health professionals as their central source of reassurance; but experiences often fell short of expectations. Participation in tailored support
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This metasynthesis highlights the significant impact of previous perinatal loss on parents during subsequent pregnancies. The concept of ‘perinatal bereavement’, encapsulating the unique experiences of parents immediately following pregnancy loss or the death of an infant, was only recognised relatively recently by professionals.38 This phenomenon describes a complex emotional response, most commonly expressed through grief, with no prescribed timeframe.37 Individual experiences are shaped by a multiplicity of internal and external factors and emotions are often intensified or reawakened in subsequent pregnancies.39 Although parents suffer similar anxieties and fears, fathers tend to suppress outward expression, as social norms dictate that their primary role should be to support the mother.40 Misinterpretation of paternal behaviour as
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Parents’ experiences of pregnancy after perinatal death
indifference is a recognised source of discord between bereaved couples. The synthesis also emphasised how common misconceptions, such as the belief that a new pregnancy helps to ‘get over’ grief for a lost child, effectively isolated parents from social support networks, increasing reliance on external and professional intervention. Antenatal appointments and assessments of fetal wellbeing were eagerly anticipated; however, discontent was expressed with aspects of care received. Parents felt ill-prepared for the anxiety generated in advance of appointments, the short duration of reassurance gained from tests and the lack of empathy demonstrated by some professionals. These findings may relate predominantly to US maternity care, the particular framing of birth as potentially pathological and emphasis on technological management.33 However, UK women pregnant after preterm birth, many of whom had experienced perinatal loss, also expressed significant anxiety surrounding investigations.41 Taken together, these findings caution against an over-reliance on technology to provide reassurance in pregnancy after perinatal death and emphasise the need for regular contact and sustained psychological support. Tailored support programmes appeared to improve experiences of subsequent pregnancy for participants. These included several components and the contribution of individual parts to overall success of the intervention was unclear. Continuous access to experienced professional facilitators, who provided multidimensional support through individual and group contacts, was considered important. In addition, benefits were derived from building relationships with other bereaved parents. Peer support in these groups appeared to foster many of the therapeutic factors described by Yalom and Leszcz42 as underlying the success of group approaches. Despite these obvious benefits, a ‘one size fits all’ approach is unlikely to meet the needs of all parents; support groups are most frequently used by affluent and better-educated individuals43 who were over-represented in the included studies. Rajan and Oakley44 identified improved postnatal emotional wellbeing in UK women who received additional one-to-one midwife contact in pregnancy after perinatal loss.
Conclusion This metasynthesis has important implications for professionals providing maternity care to families who have experienced perinatal bereavement. Provision of sensitive and effective care in subsequent pregnancies is dependent on understanding and acknowledging the impact of previous loss. Ensuring adequate access to and opportunities for reading of case notes before consultations would alert professionals to parents’ history and promote appropriate communication. Continuity of care and access
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to known professionals between scheduled visits appears to be particularly important and should be facilitated wherever possible. Professionals should be mindful that women may delay accessing care in an attempt to self protect through emotional disengagement. Given the high levels of psychological distress reported in this group, provision of support in pregnancy after perinatal loss within UK maternity services requires urgent examination. Extending the evidence base could support the development of interventions to reduce psychological morbidity and other adverse outcomes.
Disclosure of interests The authors confirm that they have no interests to declare.
Contribution to authorship TAM and TL conceived the idea for the metasynthesis. CR and AC performed the searches, reviewed the papers, conducted the synthesis and contributed to drafting the article. TAM reviewed the papers, conducted the synthesis and drafted the final article. TL, AEH and MW conducted the synthesis and revised the article for important intellectual content.
Details of ethics approval This was a literature review; therefore no ethical approval was necessary.
Funding This study was funded by Tommy’s.
Acknowledgement The authors would like to thank Claire Storey for her comments and suggestions.
Supporting Information Additional Supporting Information may be found in the online version of this article: Table S1. Grading criteria. Table S2. Themes and subthemes. Table S3. Characteristics of studies included after quality review. &
References 1 Office for National Statistics. Births and Deaths in England and Wales, 2010. Newport, UK: ONS, 2011. 2 Barkway P. Perinatal Death: A Phenomenological Study of Bereaved Parents’ Experience. Adelaide, SA, Australia: Flinders University of South Australia, 1997. 3 Hughes PM, Turton P, Evans CD. Stillbirth as risk factor for depression and anxiety in the subsequent pregnancy: cohort study. BMJ 1999;318:1721–4.
949
Mills et al.
4 Armstrong D, Hutti M. Pregnancy after perinatal loss: the relationship between anxiety and prenatal attachment. J Obstet Gynecol Neonatal Nurs 1998;27:183–9. 5 Mulder EJ, Robles de Medina PG, Huizink AC, Van den Bergh BR, Buitelaar JK, Visser GH. Prenatal maternal stress: effects on pregnancy and the (unborn) child. Early Hum Dev 2002;70:3–14. 6 Blackmore ER, Cote-Arsenault D, Tang W, Glover V, Evans J, Golding J, et al. Previous prenatal loss as a predictor of perinatal depression and anxiety. Br J Psychiatry 2011;198:373–8. 7 Warland J, O’Leary J, McCutcheon H, Williamson V. Parenting paradox: parenting after infant loss. Midwifery 2011;27:e163–9. 8 Turton P, Badenhorst W, Hughes P, Ward J, Riches S, White S. Psychological impact of stillbirth on fathers in the subsequent pregnancy and puerperium. Br J Psychiatry 2006;188:165–72. 9 Bhattacharya S, Prescott GJ, Black M, Shetty A. Recurrence risk of stillbirth in a second pregnancy. BJOG 2010;117:1243–7. 10 Getahun D, Lawrence JM, Fassett MJ, Strickland D, Koebnick C, Chen W, et al. The association between stillbirth in the first pregnancy and subsequent adverse perinatal outcomes. Am J Obstet Gynecol 2009;201:e1–6. 11 Royal College of Obstetricians and Gynaecologists. Late Intrauterine Fetal Death and Stillbirth (Green-top 55), 1st edn. London: RCOG, 2010. 12 Jensen LA, Allen MN. Meta-synthesis of qualitative findings. Qual Health Res 1996;6:553–60. 13 Cooke A, Smith D, Booth A. Beyond PICO: the SPIDER tool for qualitative evidence synthesis. Qual Health Res 2012;22:1435–43. 14 Shaw RL, Booth A, Sutton AJ, Miller T, Smith JA, Young B, et al. Finding qualitative research: an evaluation of search strategies. BMC Med Res Methodol 2004;4:5. 15 Noblit GW, Hare RD. Meta-Ethnography: Synthesizing Qualitative Studies. Thousand Oaks, CA: Sage, 1988. 16 Walsh D, Downe S. Appraising the quality of qualitative research. Midwifery 2006;22:108–19. 17 Downe S, Finlayson K, Walsh D, Lavender T. ‘Weighing up and balancing out’: a meta-synthesis of barriers to antenatal care for marginalised women in high-income countries. BJOG 2009;116:518–29. 18 Morton PA. Perinatal Loss and the Replacement Child: The Emotional Limits of Reproductive Technology. New York, NY: City University of New York, 1996. 19 Sun HL, Sinclair M, Kernohan GW, Chang TH, Paterson H. Sailing against the tide: Taiwanese women’s journey from pregnancy loss to motherhood. MCN Am J Matern Child Nurs 2011;36:127–33. 20 Cote-Arsenault D, Donato KL. Restrained expectations in late pregnancy following loss. J Obstet Gynecol Neonatal Nurs 2007;36:550–7. 21 Cote-Arsenault D, Donato KL, Earl SS. Watching and worrying: early pregnancy after loss experiences. MCN Am J Matern Child Nurs 2006;31:356–63. 22 Cote-Arsenault D, Freije MM. Support groups helping women through pregnancies after loss. West J Nurs Res 2004;26:650–70. 23 Cote-Arsenault D, Marshall R. One foot in-one foot out: weathering the storm of pregnancy after perinatal loss. Res Nurs Health 2000;23:473–85.
950
24 Cote-Arsenault D, Morrison-Beedy D. Women’s voices reflecting changed expectations for pregnancy after perinatal loss. J Nurs Scholarsh 2001;33:239–44. 25 O’Leary J. The Meaning of Parenting During Pregnancy: A Descriptive Phenomenological Study of Parenting a Subsequent Baby Following a Perinatal Loss. Minneapolis, MN: University of Minnesota, 2002. 26 O’Leary J. The trauma of ultrasound during a pregnancy following perinatal loss. J Loss Trauma 2005;10:183–204. 27 O’Leary J, Thorwick C. Fathers’ perspectives during pregnancy, postperinatal loss. J Obstet Gynecol Neonatal Nurs 2006;35:78–86. 28 Thebert-Wright C. Women’s Experiences with Coping After Late Pregnancy Loss: A Qualitative Study. Kalamazoo, MI: Western Michigan University, 2002. 29 Caelli K, Downie J, Letendre A. Parents’ experiences of midwife-managed care following the loss of a baby in a previous pregnancy. J Adv Nurs 2002;39:127–36. 30 Keith PM. Womens’ Experiences of Livebirth After Stillbirth. Vancouver, BC, Canada: University of British Columbia, 2005. 31 Simmons H. The Experience of ‘High-Risk’ Pregnancy Following Perinatal Loss. Halifax, NS, Canada: Dalhousie University, 2007. 32 Geller PA. Pregnancy as a stressful life event. CNS Spectr 2004;9:188–97. 33 van Teijlingen E, Wrede S, Benoit C, Sandall J, DeVries R. Born in the USA: exceptionalism in maternity care organisation among high income countries. Sociol Res Online 2009;14:1–19. 34 Hebert MP. Perinatal bereavement in its cultural context. Death Stud 1998;22:61–78. 35 Klier CM, Geller PA, Ritsher JB. Affective disorders in the aftermath of miscarriage: a comprehensive review. Arch Womens Ment Health 2002;5:129–49. 36 Brier N. Grief following miscarriage: a comprehensive review of the literature. J Womens Health (Larchmt) 2008;17:451–64. 37 Fenstermacher K, Hupcey JE. Perinatal bereavement: a principle-based concept analysis. J Adv Nurs 2013;69:2389–400. 38 Kennell JH, Slyter H, Klaus MH. The mourning response of parents to the death of a newborn infant. N Engl J Med 1970;283:344–9. 39 Cote-Arsenault D. Pregnancy after loss. J Obstet Gynecol Neonatal Nurs 1999;28:13–14. 40 de Montigny F, Beaudet L, Dumas L. A baby has died: the impact of perinatal loss on family social networks. J Obstet Gynecol Neonatal Nurs 1999;28:151–6. 41 O’Brien ET, Quenby S, Lavender T. Women’s views of high risk pregnancy under threat of preterm birth. Sex Reprod Healthc 2010;1:79–84. 42 Yalom I, Leszcz M. The Theory and Practice of Group Psychotherapy, 5th edn. New York, NY: Basic Books, 2005. 43 De Frain J. Learning about grief from normal families: SIDS, stillbirth, and miscarriage. J Marital Fam Ther 1991;17:215–32. 44 Rajan L, Oakley A. No pills for the heartache. The importance of social support for women who suffer pregnancy loss. J Reprod Infant Psychol 1993;11:75–87.
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