The impact of a sibling's life-limiting genetic condition on adult brothers and sisters
Abstract
It is estimated that rare diseases affect the lives of over three million people in the United Kingdom. Of these, a significant proportion are children and young people with genetic life-limiting or life-shortening conditions. This study used a qualitative approach with in-depth semi-structured interviews to explore the experiences of 10 adult siblings of a baby diagnosed with Trisomy 13 (Patau syndrome) or Trisomy 18 (Edward syndrome). Findings illustrate that parental grief from the time of their child's diagnosis onward is also experienced by siblings. Although young adults may have conflicting feelings as a bereaved sibling, there is evidence that the experience impacts on their world views and their attitudes about prospective and expectant parenthood. The study highlights the importance of providing siblings with short-term and long-term support from the time of their brother's or their sister's diagnosis onward and provides new understanding about benefit of professional and peer support in helping young adults develop resilience and coping strategies.
1 INTRODUCTION
The Department of Health UK Strategy for Rare Diseases (DHSSPS, 2013 ) estimates that rare diseases affect the lives of over three million people in the United Kingdom. A significant proportion are children and young people with genetic life-limiting conditions or life-shortening conditions (Together for Short Lives, 2016 ). In England and Wales between 2004 and 2011, there were 4,028 reported diagnosed cases of conceived babies with Trisomy 18 (Edward syndrome) and 1,662 reported cases of conceived babies diagnosed with Trisomy 13 (Patau syndrome) (Wu, Springett, & Morris, 2013 ). There is paucity of data concerning the average survival time for babies born with Patau or Edward syndrome (Nelson, Rosella, Mahant, & Guttmann, 2016 ). However, Wu et al's (2013) study found that one in five babies will survive 12 weeks or more and one in twelve babies will live for more than 12 months. Many of these surviving children will have complex health needs and significant disabilities (Janvier, Farlow, & Wilfond, 2013 ; Nelson et al., 2016 ). A study by Bruns (2015 ) highlighted a need for research focusing on those children who survive longer. Woodgate, Edwards, Ripat, Rempel, and Johnson (2016 ) recommended service providers and professionals engaged in research specifically focused on the unique experiences of siblings living with a brother or a sister with complex health needs.
Family members are interdependent. Anything that impacts on one person is likely to affect everyone in the family unit (Youngblut & Brooten, 2013 ). Brothers and sisters may be particularly at risk because parents are likely to experience anticipatory grief from the time of their baby's diagnosis (Barnett et al., 2003 ; Smith & Cheater, 2013 ). The focus of attention is on their affected child or the memory of their child who has died (Packman, Horsley, Davies, & Kramer, 2006 ).
Several authors purport the death of a sibling represents the severance of one of the most intimate relationships of a person's lifetime (Kim, McHale, Osgood, & Crouter, 2006 ; Pari, Carter, & Day, 2009 ). Siblings often share life experiences and they have been described as “comforters,” “friends,” and “antagonists” (Kim et al., 2006 ). The loss of this relationship may cause a young person to lose a sense of meaning and purpose (Brownlee & Oikonen, 2004 ; Kempson & Murdock, 2010 ). Siblings born into a family after a baby has died may still mourn the loss of what might have been (Youngblut & Brooten, 2013 ). Those who have grown up living with a brother or a sister are more likely to experience a sibling's death as major disruption to family stability and well-being (Giovanola, 2005 ). Others whose sibling died at birth or very soon after birth and those who were very young when their sibling was born are likely to have very few memories on which to draw in later life. This has been acknowledged in the literature which focuses on families with a child with a life-limiting/life-shortening condition and those with complex health needs as contributing to the distinctive nature of bereaved sibling experience (Donovan, Wakefield, & Russell, 2015 ; Youngblut & Brooten, 2013 ). Siblings who live with a brother or a sister with a life-limiting/life-shortening condition are likely to spend considerable time adjusting and re-adjusting to the situation (Axelsson, Granlund, & Wilder, 2013 ; Caplan, 2011 ; Woodgate et al., 2016 ). However, many value spending time with their brother or sister (Woodgate et al., 2016 ).
According to research undertaken by Oltjenburns (2001 ), Ribbens McCarthy and Jessop (2005 ), Rostila, Saarela, and Kawachi (2012 ), sibling grief and distress after a brother or a sister dies can be prolonged and re-surface at times of young peoples’ transitions or changed life experiences. Rosenberg et al's (2015) study of the long-term psychosocial outcomes among bereaved siblings of children with cancer provided evidence that bereavement impacted on transitions in education and career choices.
Malone (2007 ) reported that the impact of sibling death is more likely to be greater in girls. However, due to a lack of control group, sample bias, and limited sample size in studies, the true impact of sibling loss remains unidentified (Bolton et al., 2016 ). Where studies have been undertaken, these focus almost exclusively on data from mothers reporting on the effects on their children when a sibling dies from cancer (Bolton et al., 2016 ; Eilegard, Steineck, Nyberg, & Kreicbergs, 2013 ; Herbeman, Fullerton, & Ursano, 2013 ; Rosenberg et al., 2015 ), neuro-degenerative conditions (Arias et al., 2011 ), cerebral palsy (Al Gamal, 2013 ), and spinal muscular atrophy (Yang, Mu, & Wang, 2016 ).
There is increasing reference in the literature to the fact that although the death of a brother or a sister can have catastrophic consequences for siblings, providing young people are supported in their families and they are able to access professional intervention, many develop resilience and coping strategies which are transferrable to adulthood (Brown, 2013 ; Portnoy & Stubbs, 2012 ). Research undertaken by Seiffe-Krenke (2000 ) suggests that young people bereaved after 15 years old are more likely to develop effective strategies for dealing with stress in adulthood. Rostila et al. (2012 ) recommend that siblings receive support at the time of their brother's or their sister's end of life in addition to long-term therapeutic care after the sibling's death. Whatever the circumstances, the death of a sibling is a deeply personal experience for young people and they need to be supported in coming to terms with their emotions publicly and privately.
1.1 Study aims
The purpose of the research was to explore the impact on the long-term well-being of adult brothers and sisters of a sibling diagnosed with the genetic life-limiting/life-shortening conditions Trisomy 18 or Trisomy 13. Objectives were to ascertain how being a bereaved sibling affects world-views and how this impacts on adult bereaved sibling attitudes about parenthood and support needs. To our knowledge, this is the first study of its kind in the United Kingdom which has enabled adult siblings to share their previously untold stories.
2 METHODS
The research used an exploratory qualitative approach which aimed, through examining siblings’ retrospective narrative, to reach a better understanding of bereaved sibling experiences, providing groundwork for future research.
Recruitment of the siblings was purposive. The team worked closely with the charity who acted as gatekeeper to identify potential participants. The charity featured an article in their Family Newsletter in relation to the proposed research, inviting siblings’ willingness to participate in a telephone interview with a member of the Research Team. Ten adult siblings aged 18>31 years expressed their interest in the research. These siblings were contacted by the research team to ensure their willingness to be involved and demographic information was collected, including gender, age, age at sibling's death, birth order of the interviewee, gender of the deceased brother or sister, numbers of living brothers and sisters in the family, and family ethnicity. A demographic form was developed and data recorded. Participants were recruited from England, Scotland, and Wales. Criteria for inclusion in the study was a person over the age of 18 years, bereaved of a sibling with a diagnosis of Edward syndrome or Patau syndrome. Eight of the siblings had been alive at the time of their brother's or their sister's diagnosis and two had been born into their family after their sibling's death (Table 1 ).
| Gender | Age range | Number and age-range of living siblings in family | Age at affected sibling's birth | Age range at sibling's death | Pre-natal diagnosis | Life-span of baby/child | Place of death |
|---|---|---|---|---|---|---|---|
| Females | 18>30 years | 6 siblings 3>36 years | Unborn>17 years | Unborn>sibling living | 5 diagnosed 2 uncertain | Stillborn>living | 1 hospice 4 hospital 1 home 1 unknown |
| Males (3) | 19>31 years | 6 siblings 15>33 years | Unborn | Unborn>30 years | 1 diagnosed 2 uncertain | 2 day>7 years | 2 hospital 1 hospice |
A profile was created for each participant as recommended by Braun and Clark (2006 ). To determine sensitivity, appropriateness, and clarity of questions, the interview schedules were piloted with parents of two bereaved sibling teenagers who did not meet the age criteria for inclusion in the study. (These parents had requested their family should be involved in the project if possible). Some minor revisions were made. In-depth telephone interviews were undertaken with each participant, lasting between 1 hr and 2 hr. Open-ended questioning enabled data to be collected on sibling personal histories, perspectives, and experiences, taking into account the sensitive nature of the topics to be explored (Babbie, 2007 ). Interview questions were structured in a way that took participants on a journey from diagnosis through to bereavement. Initial questions encouraged siblings to recount their experience of living in a family with a baby diagnosed with a genetic life-limiting/life-shortening condition. Questions then focused on how siblings perceived the experience had shaped their adult lives and in particular their views as either prospective parents or as parents themselves. Siblings born into their family after a brother or a sister had died were asked how their families had recounted their experience of a baby diagnosed with a Trisomy and whether this had affected their childhood and adult life. Interview questions to the sole participant in the study with a living sibling focused on her 19 years as a sister. Gentle probing was used to encourage siblings to elaborate on their answers. Interviews were audio-recorded and transcribed verbatim. A sample of interview transcripts was checked against the audio recordings and verified by a member of the Research Team.
2.1 Ethics and governance
All supporting documentations including the research proposal, letters of invitation, participant information sheets, and consent forms were submitted for ethical review to the University Ethics and Governance Committee. The project was categorized as being low-risk and full approval was granted. No participants were named in project data and all interviews were undertaken and transcribed verbatim by a member of the research team not involved in the interviews and the transcripts were stored on encrypted files. Written consent was obtained from all participants. In accordance with the UK NHS Research Ethics Guidelines, participants were provided with a Participant Information Sheet and the research process was fully explained before they were asked to give their consent.
At the time of interview each participant was reassured that their anonymity would be safeguarded and that they could stop the conversation at any time. Agreement was sought from each interviewee to use anonymized quotes in the final research findings.
Data generation took into account the sensitive nature of the project focus which has involved participants describing their emotional responses from the baby's diagnosis onward. The researcher who carried out the interviews had longstanding experience interviewing vulnerable adults and children in addition to being a qualified bereavement counselor. Participants were given the details of the charity's Family Support Helpline should they need to talk to a person post interview.
2.2 Data analysis
Each interview transcript was read through several times in order for the researchers to familiarize themselves with the content. Thematic statements were identified within each participant's story in relation to the diagnosis of their brother's or their sister's condition; family life; the child's death; the funeral; and bereavement; the impact of the experience on their long-term well-being and professional support for the family. The interview with the sibling who had a living sister, focused on the diagnosis of the condition, family life, and her perceived impact of the experience. Notes were made about why the statements were considered to be important. Each statement was examined in terms of the study purpose and sub themes were highlighted and clustered (Braun & Clark, 2006 ; Smith & Firth, 2011 ). Quotes were selected to support findings within each of the six themes, highlighting similarities and differences. Field notes were also made in relation to general findings. This approach is particularly pertinent to the life story or biographical context of what people say and the ways in which they organize and make sense of events in their lives (Bryman, 2004 ; Roberts, 2002 ). Field notes also revealed that although questions invited participants to tell their stories sequentially from diagnosis onward, memories of their experiences were often interwoven.
3 RESULTS
The findings are reported under the five broad themes identified.
3.1 Diagnosis
We knew the diagnosis at 33 weeks. It was horrible because the doctors were pushing for my mother to end the pregnancy − but we had decided on her name − we had a cot − we had everything for her. We had watched her move − how could we have contemplated terminating the pregnancy then? We decided together − it was a unanimous decision.
My parents told me and they left some leaflets around but I kind of thought if I read them that it might tempt fate so I didn't do any finding out until he was born and we knew for certain he had Edward's.
More than half of the siblings had learned about a brother or a sister who had died, by overhearing a conversation or finding literature which parents had put out of sight. Others were aware that a brother or a sister had died but the birth and the death of the baby had never been spoken about. These siblings had chosen to respect their mother's or their father's right to secrecy even though this has caused them emotional confusion and anguish. In all cases, diagnosis seemed to be a water-shed between two different lifestyles—the pre-diagnostic “normal” life and post-diagnostic life. Both parents and siblings had to adjust. Older siblings often cared for younger siblings and in several cases, they spoke about undertaking extra domestic responsibilities because parents were exhausted. Many of the interviewees spoke about the emotions that family members’ experienced as recurrent rather than sequential. One sibling referred to her feelings as “coming and going like the waves of the ocean.” Siblings who knew about the baby's diagnosis before birth described how they had experienced sadness and wishing things were not as they were.
How families coped with the stress seemed to vary. The type of Trisomy, severity of the condition, and visibility of physical impairment all played their part. Siblings reported being particularly aware of their brother's or their sister's physical appearance including cleft lip, small body size, and clenched fingers.
There are several phases of adaptation that siblings appeared to experience including shock, yearning, confusion, and helplessness (Table 2 ). Notwithstanding, all of the siblings who had lived with a brother or a sister diagnosed with Trisomy felt their lives had been enriched by the experience and that “this outweighed the tragedy of the situation.”
| Shock on hearing the news of the baby's prognosis. This was often accompanied by feelings of denial and disbelief. |
| Yearning for the normality of life before the news was heard. This longing for “normality” was often accompanied by crying, anger, and overwhelming sadness. |
| Confusion especially when parents had to make choices about whether to continue the pregnancy after the diagnosis had been given. Siblings expressed feelings of disbelief that a baby could be born with a condition from which they would die. |
| Helplessness accompanied by feelings of isolation, loss of purpose, low mood/depression, and anxiety. Siblings struggled to come to terms with powerful emotions and to understand the situation in which they found themselves. |
3.2 The child's death
Occasionally I think that I should ask what happened, but it seems like an intrusion on my parent's life. I had a brother and a sister born later on and I don't think that either of them knows anything about the “lost” baby.
There were times when I had had enough; every day we thought, is he going to die today? I suppose we must have known that his passing was inevitable but as a young child who had not really grasped a sense of time, the waiting seemed interminable.
We used to go and sit all day at the funeral directors and my mum became convinced that sometimes she could see my sister moving.
3.3 The funeral and bereavement
My mother carried her coffin in her arms and I remember this sinking feeling in my stomach − like when you fail an exam − but it was one hundred times worse. I never knew my stomach could sink so low.
Involvement in planning the service and playing a part in the ceremony was important to siblings. Years after their brother's or their sister's death, siblings are able to recall the funeral in detail.
We were in the crematorium and we were expecting the coffin to go behind the curtains but it went down and it was an enormous shock. My mother dived towards the coffin and my other mother pulled her back − my big sister's legs gave way and when I saw her falling as well it was as if both my sisters were going down together − I picked my sister up although she is 16 years old and I put her on my lap and held her like a baby. We were all sobbing − we couldn't move off our seats and everyone else just left the crematorium.
Siblings who had not attended the funeral, spoke about “unfinished business,” “feeling left out,” or “strangely removed from the reality of the death.” One of these siblings had decided on the way to the crematorium that she did not want to go to the service, a decision she had later regretted.
Teachers played a pivotal role in helping children to express their emotions and two siblings recalled how staff had attended their brother's or their sister's funeral or sent a letter of condolence to the family. Support from peers was highly valued and some siblings had discovered “true and lasting friendships’ which had often continued into adult life.
Eventually we started to go out but it was hard because we live in a very small town where everyone knows everybody. We felt different because I was the girl whose sister had died and my mother was the person who had lost a daughter − they were really hard times.
Every night my mother asked me to look at my brother's picture and to kiss it before I went to bed. I went along with what was required of me but I used to dread that.
Anniversaries and special times were poignant for all of the siblings. Significant dates always brought to mind the grief which brothers and sisters continued to feel, but marking occasions helped them keep the child's memory “alive.”
3.4 Impact on the long-term well-being of siblings
I can genuinely say that having a sister with Trisomy has made an enormous difference to the way we have lived our lives but my mum and I would not change the situation for the world. Although my mum has had to fight my sister's corner for her on many occasions and she has had to do this alone, we are very close as a family. But I do worry about the strain on my mum.
The way in which parents and siblings communicated with each other appears to have been critical in determining how well individual family members adjusted and continued to live their lives. How parents shared their emotions also appears to determine how well siblings felt included and whether they felt that their grief was validated. Two of the siblings had grown up in a single parent family and they perceived that the impact of parental grief had resulted in their parents separating or divorcing. Siblings talked about their perception of their father needing to “stay in control” and how their mothers had been “overwhelmed” by emotions.
My parents kept my brother's bedroom like a shrine and every night we used to go and say “goodnight” to a photograph of him. It was weird really and I found myself really hating this requirement my parents placed on us.
Having the experience has helped me empathise more with other people and it has definitely moulded who I am.
Two of the siblings had been born after their brother or their sister had died. In these circumstances, they seemed to have incorporated the experience into their memories of growing up and they remarked that it had seemed quite “normal” because they had never known any different.
I think being a sibling has made it difficult for me as I associate having kids with a lot of stress and that's put me off if I'm honest. I can't imagine combining my life with a child's life because I've only experienced a child taking over my parent's life.
I worry that I might be the exception and have an affected baby. But if this happened I would go ahead with the pregnancy because I think I could cope.
3.5 Professional support
Where professionals had given information honestly and in straightforward language this was valued. Siblings who were included by professionals in discussions seemed to adjust more easily to the reality of the situation. Indeed, where professionals had taken time to include siblings in decisions this was very helpful.
I remember going to the hospital and hearing a woman scream. The nurse told me that my mother was very upset because she had just been given the news by the doctor that my brother's condition was “incompatible with life.” In the end he lived for a while but the insensitivity of the doctor scarred my mother and I believe that she suffered post-traumatic stress as a result.
In the end we put a notice on the door which said ‘NO VISITORS TODAY PLEASE. We just needed to space to be ourselves, even though the midwife was wonderful. She just sat with us and didn't ask us how we felt. She knew how awful it was for us all.
Throughout this interview I have realised that it wasn't really the big gestures that help most − it was the small things. They weren't grand deeds but they really made a difference.
I remember my dad saying that the yearly gathering was the only time he ever had a chance to talk to other fathers. For me, most of all I remember the activities and the enormous cake we shared at teatime. I grew up looking forward to staying in a hotel for the weekend. I am not sure what my parents did while I was having fun but I got the feeling that it was positive.
4 DISCUSSION
4.1 Limitations
The project provided an insight into the lived experience of siblings of a brother or a sister diagnosed with Edward syndrome or Patau syndrome. However, there is an obvious limitation in that the sample size is small and the findings may not be replicated in a larger study or one involving siblings of a brother or a sister with a different diagnosis. Furthermore siblings who were very young, or those born after their brother's or their sister's death rely largely on family accounts of events rather than their own memories. Events reported decades later may have been altered by cognitive maturity or other significant life experience. Participation in the research was reliant upon a third party for inclusion and all the participants had a longstanding relationship with the charity which referred them to the research team. This may have led to bias in opinions since many of the families had been reliant on the charity for support from the time of their baby's diagnosis onward. Families who had not continued a relationship with the charity for emotional or social support or altruistic reasons may have different experiences. Additionally the findings reflect the views of families from White ethnic backgrounds and it is possible that the experiences and opinions of individuals from Black, Asian Minority Ethnic (BAME) communities might differ (Table 3 ).
| Participant | Participant's gender | Participant's age at interview | Participant's age at sibling's birth | Participant's age at sibling's death | Pre-natal diagnosis of trisomy 13/18 | Life-span of baby or child | Place of death |
|---|---|---|---|---|---|---|---|
| 1 | Female | 18 years | 17 years | 17 years | √ | 50 days | Children's hospice |
| 2 | Male | 19 years | 18 months | 18 months | √ | 2 days | Hospital |
| 3 | Female | 23 years | Not born | Not born | √ | 18 months | Hospital |
| 4 | Female | 29 years | 10 years | 10 years | Unknown | Stillborn? | Unknown |
| 5 | Male | 26 years | Not born | 24 years | x | 5 days | Hospital |
| 6 | Female | 24 years | 4 years | 11 years | √ | 7 years 4 months | Home |
| 7 | Female | 30 years | 11 years | 11 years | √ | Stillborn | Hospital |
| 8 | Female | 22 years | 3 years | Sibling living | Unknown | n/a | n/a |
| 9 | Male | 31 years | 7 years | 14 years | Unknown | 7 years 9 months | Children's hospice |
| 10 | Female | 29 years | 20 months | 20 months | √ | 1 day | Hospital |
Despite some limitations, the interviews demonstrate much in common with the existing literature in relation to the impact on families of the diagnosis of a child with a life-limiting/life-shortening condition and those with complex care needs (Caplan, 2011 ; Donovan et al., 2015 ; Woodgate et al., 2016 ; Youngblut & Brooten, 2013 ). Notwithstanding, there are also some innovative findings in relation to sibling experiences which can inform professionals working in bereavement services, health, nursing, and education. In particular, siblings drew the researchers’ attention to the importance they placed on professionals listening to their stories and they viewed such opportunities as therapeutic.
Theorists of childhood bereavement have advocated that open and honest communication between parents and children is pivotal to bereaved children's long-term psychological wellbeing and development (Freud, 1984 ; Kubler Ross, 1983 ; Worden, 2004 ). The siblings reflected on the benefit of being included in conversations with their parents and with professionals from the time of diagnosis, bereavement, and beyond. Brothers and sisters also spoke about the importance of being proactively involved by parents in decisions about their life-threatened sibling's care. Brown's (2007 ) study of life-limited children and their families underlined the importance of providing children with age-appropriate information and this has been endorsed in this research. It seems likely that even if young children do not fully comprehend the implications of a life-limiting/life-shortening condition or the meaning of death, where they have been given an explanation which has avoided the use of euphemisms and metaphors, this has been helpful.
Education has provided siblings with “time out” away from the emotionally charged atmosphere at home and opportunities that can contribute to their holistic development. Previous research (Brown, 2007 ; Davis, 1999 ) has reported a prevalence of bullying in school where bereaved young people are perceived as “different” but this research has described peers as being particularly supportive.
Ribbens McCarthy and Jessop (2005 ) purported that bereavement contributed to a decline in young peoples’ academic performance. Although this topic was not explored in this research, five of the siblings interviewed had been to university and one was currently undertaking training as a GP. Additionally four of these graduates were employed in caring professions which has raised the question as to whether their life experience has influenced their educational goals and career choice, a fact previously reported by Caplan (2011 ) and Rosenberg et al. (2015 ). Certainly there is strong evidence in this study that being a bereaved sibling has shaped the world-view of the young adults who perceive themselves as empathetic toward the needs of other people.
Empirical research (Edgar-Bailey & Kress, 2010; Ribbens McCarthy & Jessop, 2005 ; Rostila et al., 2012 ) has concluded that bereaved sibling grief is likely to re-surface in adulthood at times of transition. This research has demonstrated that although young people may experience conflicting feelings in relation to their loss, the experience of being a bereaved sibling is likely to impact on young adult views of both prospective and expectant parenthood. Other studies conclude that young peoples’ emotional health may be at particular risk where they have experienced a significant loss (Ribbens McCarthy & Jessop, 2005 ). Conversely, Luthar and Cicchetti (2000 ) and Grotberg (2001 ) purport that providing young people receive adequate support they may adapt positively to bereavement. The findings of this study would endorse this view.
Recent research by Rostila et al. (2012 ) which focused on whether there was an increased likelihood of morbidity in bereaved siblings compared with non-bereaved siblings, recommended siblings receive support before and after their brother's or their sister's death. This research focusing on siblings known to a charity supporting families of Trisomy 13 and Trisomy 18 children has contributed new understanding about the importance of altruistic support through long-standing friendships formed at the charity family weekends and in particular siblings perceived benefits in contacting other bereaved siblings via social media, a fact previously reported by Janvier et al. (2013 ) in their research with parents of surviving children diagnosed with Trisomy 13 or Trisomy 18. Siblings from families who had either ceased relationships with the charity or who had never received support, might have different experiences.
According to Edgar-Bailey and Kress (2010 ) participating in rituals can “provide young people with an opportunity to engage in the present moment by creating meaningful behavior that reframes their tragedy in light of a significant purpose” (p. 170). Seven of the siblings had attended their brother's or their sister's funeral and this was reported as beneficial providing siblings knew what to expect. Some young adults who had actively participated in the funeral ceremony recalled the event in great detail. Most of these siblings continued to attend family occasions and anniversaries which marked their sibling's life and death. This was in sharp contrast to others who for various reasons had not attended the funeral, a situation which was perceived by siblings as being “unhelpful” or in the case of not being given a choice of whether to attend or not, as “unfair.” Where bereaved brothers and sisters had engaged in rituals this seemed to support the siblings in sustaining an ongoing bond with their deceased brother or sister. The participant with a living sibling recounted how celebrating birthdays and significant family events with her sister “helped build a treasure chest of positive memories.”
Malone (2007 ) noted that females may be more likely to be affected by sibling death than males. Although only two males participated in this study, there were some apparent gender differences in respect of the impact of sibling bereavement. Data suggest that females may include more descriptions of emotional aspects of their experiences while males placed greater emphasis on disruption of daily routines and practical issues in relation to their sibling's care.
5 CONCLUSIONS
The experience has taught me so much about life and death. In her short life my sister left her mark on my family and on everyone who met her. If you ever re-tell the story I am telling you, let people know that my sister was a fighter first − she had Edward's Syndrome second, because her condition did not define her.
Given that long-term survival of children with Trisomies is greater than previously thought (Nelson et al., 2016 ), professionals and service providers need to be aware of the importance of providing holistic family support from diagnosis to death and beyond. This includes identifying potential factors that may impact on sibling long-term well-being. The impact of long-term holistic family support needs to be evaluated to provide robust evidence. Notwithstanding, the results of this study demonstrate whether parents and professionals provide age-appropriate information to brothers and sisters and include them in open and honest discussions about their sibling's diagnosis as early as possible this is helpful. The experience of living with a sibling of a brother or a sister diagnosed with a life-limiting condition has been shown to impact positively, for example, in developing empathic attitudes and resilience which are carried into adulthood. Where families provide siblings with choices about whether they attend and participate in their brother's or their sister's funeral the study has shown this to be beneficial. Additionally, opportunities for siblings to sustain memories, for example, through participating in shared events which celebrate their brother's or their sister's life this has been positive. Providing opportunities to tell their stories enables bereaved brothers and sisters to integrate their past experiences into their present and future lives.
The charity that commissioned the study has used the findings to inform their service development and is currently sharing the outcomes with statutory and third-sector organizations and commissioners involved in providing holistic support to families with a child diagnosed with a life-limiting/life-shortening condition.
ACKNOWLEDGMENTS
The authors would like to thank all the participants for sharing their experiences and the charity that commissioned the research for their assistance in recruitment.
