Fatal fetal anomaly: Experiences of women and their partners

This study explored the care experiences of parents whose pregnancy was diagnosed with a fatal fetal anomaly following the legalisation of termination of pregnancy in 2019 in Ireland.


| INTRODUCTION
Approximately 2% of pregnancies are diagnosed with a congenital anomaly each year. 1,2 A proportion of these anomalies will be lethal or fatal, and congenital anomalies remain the leading cause of fetal death and child mortality. 2 The term fatal fetal anomaly (FFA) is used to describe anomalies that will lead to fetal or neonatal death and is used interchangeably with lethal and life-limiting terms. 3,4 While FFA is not a medical term, it gained popularity in Ireland during its use in the political campaigns to repeal the Eighth Amendment of the Irish Constitution. 5 The Eight Amendment prevented termination of pregnancy (TOP) for FFA in Ireland prior to repeal and the subsequent enactment of the Health (Regulation of TOP) Act, 2018, which legalised TOP in Ireland for the first time. 5 A diagnosis of FFA can result in an intense grief reaction for parents, 6,7 which can be exacerbated by inadequate care and support. [8][9][10][11][12] A variety of responses to the diagnosis of an FFA as well as coping strategies have been identified, demonstrating a spectrum of parental health care needs. These needs are multifactorial and often extend beyond routine care. 13 Following confirmation of an FFA, parents are faced with decisions concerning the remainder of the pregnancy, giving birth and seeing their new-born baby, or choosing TOP. 8,14,15 Following the introduction of legislation permitting TOP for FFA, 16 and the subsequent provision of abortion services, fetal medicine specialists reported stretched resources and challenges in the rapid introduction of a TOP service. 17,18 There is some evidence to suggest a lack of standardised care and service provision for TOP across maternity units in the Republic of Ireland. 19 An understanding of the care experiences of those who continue the pregnancy and those who terminate the pregnancy following an FFA diagnosis is essential to optimise new and developing services. 20 However, there is a lack of research regarding the lived experience of those who receive a diagnosis of an FFA, particularly in Ireland since TOP became legal in 2019. Therefore, the aim of this study was to explore the care experiences of parents whose pregnancy was diagnosed with an FFA following the legalisation of TOP for FFA in 2019.

| Study design
A qualitative study design was adopted. 21,22 Grounded in phenomenology, interpretative phenomenological analysis (IPA) was undertaken to facilitate an in-depth exploration of the subjective care experiences of individual parents whose pregnancy was diagnosed with an FFA. 23,24 The study is reported in line with the Standards for Reporting Qualitative Research. 25

| PARTICIPANTS
A purposeful sampling strategy was engaged to identify those parents whose pregnancies were diagnosed antenatally with an FFA in 2019 and 2020. 26 This time period follows the 2019 change in legislation allowing for the option of TOP for FFA in Ireland. Parents were required to be 12-24 months bereaved or six months following a diagnosis where the child was alive. 27

| RECRUITMENT
Recruitment was facilitated through voluntary organisations who provide support to parents who continue or terminate a pregnancy following an FFA diagnosis, and by the pregnancy loss team in a tertiary maternity hospital. Snowballing techniques were also utilised. 28 Upon being identified to the research team, parents were sent an electronic information sheet outlining the nature and purpose of the study and details of the interview process. Ten parents (six women and four of their partners) were recruited. Drawing on the concept of information power, recruitment was ceased at this point as it was perceived that the depth required to provide meaningful insight into parental experiences of an FFA diagnosis had been achieved. 29

| Knowledge-user engagement
A parent advocate and various healthcare professionals (HCPs) working in pregnancy loss services were consulted for feedback on the content, language, and appropriateness of the topic guide, information sheet, and consent process. This ensured the relevance, acceptability, and accessibility of the research. 30,31

| DATA COLLECTION
One-to-one semi-structured interviews were conducted, recorded, anonymised and transcribed verbatim. The interview was framed by a topic guide composed of open-ended questions. This method enabled the researcher to introduce topics for discussion but also to pursue topics important to parents, enabling a more intensive and detailed 554 engagement with individual cases. 32,33 Topics included the diagnosis of the FFA, decision making and care after the diagnosis, loss of the baby, supportive care, and follow-up. The average interview length was 80 min (range of 40-120 min). Field notes were made during and after each interview. 34 Due to the ongoing global COVID-19 pandemic, interviews were conducted virtually through the videoconferencing software Google Meet. 35 In the absence of in-person contact, videoconferencing facilitated the nonverbal communication that is a key element of the interview process. 36 Parents were sent a unique link to ensure only they could access the interview.

| Analysis
Taking an idiographic approach, the analysis followed the six-stage process of IPA outlined in Smith and Larkin (2009). This began with close reading and re-reading of a transcript to begin to enter the participant's world. 24 This was followed by initial detailed noting by hand on the right side of the margin with descriptive, linguistic, and exploratory comments on the content. 24 These comments were analyzed to identify themes; this meant focussing on discrete sections of the interview while concurrently recalling the interrelationships, connections, and patterns in the initial noting of the whole interview. 24 Once completed, the next cases were analyzed in the same fashion. 24 Finally, themes were sought across the interviews.
From these observations, a framework of superordinate themes and subordinate themes was structured. 24 The IPA was led by the primary author, a medical student, while the multidisciplinary research team was involved in ongoing discussion regarding their interpretations and reflections of the data to enhance theme development and reflexivity. 42,43 The multiple participant perspectives included in the study were carefully considered within the analysis and a consensus overlap of patterns allowed a structure and narrative to be constructed with the divergence and convergence of experiences highlighted within each theme.

| FINDINGS
Ten parents (six women and four of their male partners) participated in the research study. Three women were recruited from each arm of recruitment with both arms of recruitment recruiting parents who continued and those who terminated. Table 1

| Theme one: Attachment and Coping
Different coping strategies and responses to the FFA diagnosis were evident in all the parents, and these subsequently shaped their care needs. Parents each developed their own level of connection to the baby, with differing layers of attachment. Subordinate themes constructed were "Coping and Response" and "Baby's Personhood".

| Response to diagnosis and coping strategies
Parents described a sense of normality ahead of the ultrasound scan, which they looked forward to as an opportunity to see their baby as highlighted by Rachel (Terminated the pregnancy) (TOP) who said that she had "No idea the rug was going to be pulled from under us". An abnormal ultrasound finding that suggested that an FFA caused deep shock and parents reported experiencing a period of heightened grief and anguish, Michaela (TOP) described how "days were really long, and really torturous". Revealing the shock she felt, Judy (Continued the pregnancy) (CONT) : felt that news of the abnormal finding "was like eating sawdust". It was interpreted that part of the shock parents faced was realising the need to readjust their expectations and hopes for the future of the pregnancy. A variety of responses to the diagnosis of an FFA This showcased how by sharing the news Rob (CONT) and Liz (CONT) were trying to accept "reality". It displayed how sharing their story with family and on social media was important in removing any "secret" or idea that their baby should not be celebrated like any other baby. In contrast, those who terminated their pregnancy told fewer people. Instead, they benefitted, as did those who continued, from the support of parents with similar experiences, who they met through voluntary organisations working with parents following an FFA diagnosis. Michaela (TOP) valued being able to "talk in an environment where you're not judged," revealing the perceived societal judgment she felt regarding abortion care. Parents were looking for others who they felt they could connect with, a "tribe", who would understand their grief, as Judy (CONT) described "I kind of knew I was in this club that nobody wants to be in but okay, so there was really good support". This highlights the support most parents felt from others who had experienced pregnancy loss and revealed how it made them feel slightly less isolated. In response to the diagnosis, some parents, as their pregnancies progressed, focused on fostering a narrative that encompassed a sense of identity for the baby. This was illustrated by both Liz (CONT) and Judy (CONT) referring to "celebrating" the baby's life. Tara (CONT) described how her baby "became this person and so we were really excited about meeting them but really sad" revealing how a sense of identity and journey of the pregnancy was developed which simultaneously coexisted with intense grief. This conceptualisation by parents of celebrating and creating this journey for the baby was interpreted as giving a sense of meaning and closure to parents. Some who terminated chose not to engage with this creation of a journey, as Rachel (TOP) illustrated "Oddly enough I was okay. As I said, maybe I had kind of detached myself a little bit from it, but I was fine". Similarly Jess (TOP) shared how a bereavement nurse came in to see her and "She kept talking about the baby, and when you deliver, and to be honest, it all seems so foreign to me because I didn't even really feel like I was that pregnant". A level of detachment is evident here and this was interpreted as being necessary for parents coping as focussing on the baby's development and identity was counterintuitive to their coping process.

| Baby's personhood
The identity of the baby was central to most parent's pregnancy experience and consequently their experience of care as illustrated by Evan (CONT) who gave consideration to termination as a means of stopping "future pain". Evan (CONT) shared "As soon as we went in for  noted across all participants. Tara (CONT) illustrated this as she described how "There was always somebody there to answer a question, and to take the time, and to be with us and to listen to us and that goes from consultants and doctors to the nurses, to the bereavement nurses, to like everybody." Some parents, both those who continued and terminated, shared how important and comforting it was to continuously meet the same HCPs, who were familiar with the couple's situation.
However, some women felt that they were left alone for too long with a lack of contact from HCPs during their time in the maternity hospital. Two women shared their discontent upon lack of care during their TOP and having to birth their baby alone with one woman's birth occurring alone in the toilet on an inpatient ward.

| DISCUSSION
This qualitative study used in-depth semi-structured interviews and IPA to better understand the lived care experiences of parents whose baby was diagnosed with an FFA. Research with service users can promote holistic in-depth understanding of complex health issues improving the overall care experience. 44 The findings of this study indicate that the experience of an FFA diagnosis and subsequent care has a lasting impact on women and men.
Women do not attend an antenatal appointment with a neutral mindset. 8,45 Parents in the study expected that everything would be fine with their own baby and were unprepared for when an anomaly was detected, thus compounding shock. 8 The delivery of "bad news" holds the potential for severe perinatal stress and to shatter expectations of a routine pregnancy as was demonstrated in our study. 45 The profound emotional impact of intense shock and grief experienced by parents in this study is consistent with the literature 46 76 Implementation of new services and managing change in healthcare is a complex process. 77 This study reveals stronger dissatisfaction with and greater inconsistency in the aspects of care among parents who chose to terminate. These parents reported a lack of continuation of care after TOP and shared greater feelings of isolation and a lack of support. Parents who terminate should receive the same consistency, continuity, and empathy in their care and support as those who continue pregnancy after an FFA diagnosis.
A critical reflection on the strengths and limitations of this study is included in Table 2.

| CONCLUSION
The findings of this study provide a unique insight into the experience of parents whose pregnancy was diagnosed with an FFA, helping us to further understand their care needs at a time of development of a new national TOP service. Parents require care that is sensitive and individually adapted to meet their needs to support them to cope with the loss of their baby. 4,54-57 Our study highlighted parents need for consistent, well-communicated, and comprehensive care, which encourages a perinatal palliative care approach that is individual to the parents. Our study, among others, suggested inconsistencies across care provision. [17][18][19] Thus, further studies are needed to expand on these conclusions, better understand parental experiences of FFA, and systematically inform improvements in care pathways nationwide and internationally.

ACKNOWLEDGEMENT
The authors would like to express particular thanks to the parents who took part in the study. The authors are also grateful to the voluntary support organisations and HCPs who facilitated recruitment for the study. Special thanks are to be expressed to the various individuals who were consulted on the study materials as part of the knowledge user engagement. Open access funding provided by IReL.
T A B L E 2 Strengths and limitations of the study � The IPA methodology is a strength, as it allowed the researcher to gain an in-depth insight into the lived experience of these pregnancies. The study is supported by a multidisciplinary team with expertise in qualitative research and pregnancy loss. � This is the first study to explore the care experiences of those who continue the pregnancy and those who terminate a pregnancy since the legislation of TOP for FFA in Ireland. � The sample contained experiences of men and women, and it was balanced between those who continued and terminated. This gave an equal voice to a variety of parents. � Trisomies 18 and 13 were the most common FFA diagnoses in the study, so this study lacks an insight into other FFA diagnosis. � As with all qualitative research, the insights and experiences are not wholly generalisable to the wider population. However, they are in concordance with previous literature and are likely to have transferrable commonalities with other settings.