INTRODUCTION
When a child is diagnosed with a life-threatening illness, such as cancer, the entire family will be affected and face many challenges to retain or regain normality (Koocher & O'Malley, Reference Koocher and O'Malley1981; Bringager & Saeter, Reference Bringager and Saeter2014). Grief following the death of a significant person in one's life is acknowledged to be a complex process (Parkes, Reference Parkes1996; Thompson et al., Reference Thompson, Miller and Barrera2011). Sibling grief has been described as severe, intense, and prolonged (Nolbris et al., Reference Nolbris, Enskär and Hellström2014; Alderfer et al., Reference Alderfer, Long and Lown2010; Sveen et al., Reference Sveen, Eilegård and Steineck2014).
Siblings typically develop a strong bond with one another through shared experiences while growing up, making siblingship a major factor for normal child and adolescent development. Bereaved siblings must use their available resources and coping strategies to adapt in the best possible way (Woodgate, Reference Woodgate2006; Greeff et al., Reference Greeff, Vansteenwegen and Herbiest2011). Some siblings adjust well to a crisis, while others may lack the resilience to adapt to the new situation (Woodgate, Reference Woodgate2006). From both the clinical and scientific perspectives, it is important to gain insight into the resources associated with adjustment and adaptation of bereaved siblings.
Many studies have focused on how children and adolescents experience the loss of a sibling (Nolbris & Hellström, Reference Nolbris and Hellström2005; Eilegård et al., Reference Eilegård, Steineck and Nyberg2013a). Traditionally, much of the literature on childhood grief has been based on information by proxy and not on the voices of the adolescents themselves (Warnick, Reference Warnick2015). However, several recent studies have engaged bereaved children and adolescents who have lost a brother or sister to tell their story (Nolbris & Hellström, Reference Nolbris and Hellström2005; Eilegård et al., Reference Eilegård, Steineck and Nyberg2013b; Lövgren et al., Reference Lövgren, Bylund-Grenklo and Jalmsell2016; Sveen et al., Reference Sveen, Eilegård and Steineck2014). Nevertheless, evidence is scarce in pediatric palliative care research on how bereaved siblings perceive their brother's or sister's illness and death over time, especially the positive experiences. Insight into siblings' positive memories and experiences may be valuable in helping health professionals find ways to strengthen siblings' resilience during illness and death.
Many siblings of children with cancer experience psychosocial distress both during the brother's or sister's illness and after bereavement (Nolbris & Hellström, Reference Nolbris and Hellström2005; Woodgate, Reference Woodgate2006). Research on siblings' adjustment to childhood cancer has also focused on family support and functioning. In general, research has shown that family cohesion is a positive factor when it comes to coping (Franko et al., Reference Franko, Thompson and Bauserman2008). Cohesion bonds family members emotionally and appears to have a buffering effect on their psychological distress (Eilertsen et al., Reference Eilertsen, Hjemdal and Le2015).
There is little research on health promotion and the resilience of bereaved siblings. Resilience research has identified protective resources that are essential for adaptive coping in the face of adversity, such as an individual's characteristics and resources, family coherence, and social support (Hjemdal et al., Reference Hjemdal, Aune and Reinfjell2007; Eilertsen et al., Reference Eilertsen, Hjemdal and Le2015). Earlier studies investigating bereaved siblings' and parents' reactions and experiences have largely focused on psychopathology and rarely focused on a resource-oriented, health-promoting perspective (Greeff et al., Reference Greeff, Vansteenwegen and Herbiest2011; Norberg et al., Reference Norberg, Lindblad and Boman2005; Hjemdal et al., Reference Hjemdal, Aune and Reinfjell2007). Knowledge about positive and negative memories and the experiences of illness and death would help professionals in supporting and strengthening siblings' resilience, enabling them to create positive memories while buffering the negatives.
The aim of our study was therefore to explore bereaved siblings' positive and negative memories and experiences of their brother's or sister's illness and death.
METHODS
Our study is derived from a nationwide Swedish survey that focused on cancer-bereaved siblings' experiences of illness and death, as well as siblings' health and experiences of participation in research (Eilegård et al., Reference Eilegård, Steineck and Nyberg2013a).
All children who were diagnosed with cancer before the age of 17 and died before the age of 25 between 2000 and 2007 were traced through the Swedish Childhood Cancer Registry (all pediatric deaths caused by cancer in Sweden can be found in this registry). From this cohort, 187 fulfilled the inclusion criteria. Siblings were then located through the Swedish Population Registry (every permanent resident in Sweden has a personal identification number and can be found in this registry) and were included in our study if they were between the ages of 12 and 25 when their brother or sister died and between 19 and 33 at the time of follow-up in 2009. To be eligible, they also had to be born in one of the Nordic countries, speak and understand Swedish, and have a listed phone number and address. Of a total of 271 siblings, 240 were eligible for this study, and 174 participated. An introductory letter was sent in 2009 to eligible siblings, briefly explaining the aim of the study. A research assistant phoned the siblings 3 to 5 days later and asked if they were willing to participate. An anonymous study-specific questionnaire with a separate response letter was mailed to the siblings who agreed to participate.
The study-specific questionnaire about the siblings' experiences of losing a brother or sister to cancer was developed using a stepwise approach described by Charlton (Reference Charlton2000) and others (Eilegård et al., Reference Eilegård, Steineck and Nyberg2013a; Grenklo et al., Reference Grenklo, Kreicbergs and Valdimarsdottir2013; Kreicbergs et al., Reference Kreicbergs, Valdimarsdottir and Onelov2004). The development of the questionnaire and the pilot data collection have been described in detail (Eilegård et al., Reference Eilegård, Steineck and Nyberg2013a; Reference Eilegård, Steineck and Nyberg2013b). The final questionnaire comprised ~200 questions about the siblings' health and their experiences of bereavement. The following open-ended statements from the questionnaire were used for our study: “Please write down any positive memories or experiences from your siblings' illness and death that you might have” and “Please write down any negative memories or experiences from your siblings' illness and death that you might have.”
The regional ethics committee of Karolinska Institutet in Stockholm approved the study (no. 2007:862–31).
Data Analysis
The responses to the open-ended statements were analyzed thematically and using systematic stepwise text condensation (Malterud, Reference Malterud2012). Analysis was performed independently by three members of the research group (M.E.B.E., M.L., A.E.W.) and then discussed. This was important to create a wider analytic space (Malterud, Reference Malterud2012). In the next step, we started by systematically reviewing a transcript line-by-line to identify meaning units and then coded them. In the third step, the transcript was reduced to a decontextualized selection of meaning units sorted as thematic code groups. The data were organized and reduced to a few code groups containing meaning units and sorted into fewer subcategories with the capacity to reveal aspects of participants' positive and negative memories and experiences. In the fourth step of the analysis, the data were reconceptualized and subcategories sorted into main categories. In the final step of the analysis, the data were reviewed as a whole.
RESULTS
Approximately 70% of the 176 bereaved siblings responded to both open-ended statements, which focused on bereaved siblings' positive and negative memories and experiences of their brothers' or sisters' illness and death. The responses were subcategorized into four main themes, with contrasting terms:
1. endurance versus vulnerability
2. family cohesion versus family conflicts
3. growth versus stagnation
4. professional support versus lack of professional support
Endurance versus Vulnerability
Bereaved siblings expressed that they had many happy memories and experiences of their sick brother or sister, emphasizing the sick siblings' strong willpower until their death and how the brother or sister lived each day of their life to the fullest. Others described happy memories because of their sick brother's or sister's ability to keep smiling and be happy even in what the sibling knew was perhaps the darkest moment in the course of their illness: “He lived until he died.” Many also described that it was a good memory and experience for them to be with the sibling at home or at the hospital during treatment and when their sibling was dying: “She was peaceful at death”; “He is my hero.” Some reported that the sick sibling matured from child to adult, becoming calmer, making life around them better.
However, many others reported having had a difficult time. It was hard to think positively when the person who was perhaps closest to them was suffering, sick, and dying, possibly because of sadness around their brother's or sister's illness, which caused pain, anxiety, and suffering. The physical and mental changes during the cancer trajectory and at death appear to have had an influence on their memories and experiences. As one sibling expressed it: “My brother became another person. We had no control over the disease.”
Family Cohesion versus Family Conflicts
Many siblings expressed that being together as a family, especially with the sibling, strengthened the bonds between them: “We fought together and became closer.” Moreover, siblings emphasized that it was a positive experience that the family cared more for each other and grew closer through their brother's or sister's illness and death. Siblings pointed out that love and relations became more important for them and that being supported by relatives was a positive memory and experience for them: “We have wonderful parents.” It may also appear that sharing experiences and being with the sick sibling gave the healthy sibling a positive memory from the time of the illness and after death. A great majority of siblings described these experiences through the illness in terms of spending time together, just being with the sick sibling, or actively engaging in activities together, such as going to concerts, traveling, dogsledding, getting tattoos, painting together, just having a good time with them, or even being at the hospital together. One respondent got a tattoo to show their sick sibling that he would not be forgotten: “We became closer after the illness”; “We had a cool time together”; “It is difficult to say what is positive, but I had a good time with my sister the last day before she died.”
On the other hand, anxiety, sadness and, grief in the family led to arguments and frustration even though they needed to stay together. The memories and experiences expressed by many siblings showed that the brother's or sister's illness and death was a terrible period in their lives, with a great deal of turbulence and fear: “It was like living in a bubble.” A great many siblings highlighted the physical and mental strain on the parents, as well as the sadness around the illness and death as a negative experience for them. Communication also appeared to have a role in the siblings' negative memories: “We closed ourselves in as a family so no one else could get in”; “We could not communicate as a family before the illness, and it just got worse after the death.”
Growth versus Stagnation
Many siblings emphasized that being acknowledged by both family and health professionals was important, and it became even more so over the course of the brother's or sister's illness and death. Many siblings felt that they grew as individuals during this process, where others highlighted the physical and mental distress they experienced over this time period.
Through the course of the illness and after death, many siblings reported that they had learned to appreciate each day of life, to live in the moment, and that everyday problems became less important. They often pointed out that they tried to make the best of the situation, which they described as being extreme, and learned that life was too short to throw away: “I try not to dwell on the small problems in life”; “I look at life with new eyes.” In this extreme experience, they often expressed that they learned a great deal about themselves, thereby maturing and developing as a person: “I learned to take care of the ‘small things' in life and enjoy life”; “I learned a lot about life and what happens in a hospital”; “Contrasts in life make life more exciting.” Moreover, other siblings stated that they had become more open and had a more relaxed attitude toward death and dying, which they perceived as a positive experience.
Conversely, some siblings felt forgotten, like ghosts, and always in the way, even though they now realized that it was not like that. It appears that the healthy siblings' need for acknowledgement became even more acute during the course of the sick siblings' illness and death. Many siblings expressed it as feeling that they were priority number two and were relegated to the sidelines. They felt forgotten, not part of the team, in the way, and not valued as a sibling: “I was bullied over a long period of time, and no one helped me.” Other siblings reported that everything seemed unreal and went by so fast: “Life was like a roller coaster.” They expressed feelings of uneasiness, anxiety, panic attacks, grief, emptiness, sadness, and constant worry until the time of their siblings' death.
Professional Support versus Lack of Professional Support
Siblings experienced and remembered positive aspects of professional support, as well as a lack of such support. However, most siblings perceived the physicians and staff at the hospital as always being warm, kind, honest, and close. They felt as if the professionals cared about their ill brother or sister. The respondents emphasized that they received good information from the staff and that things were explained well. Siblings were impressed by the way they were received by professionals, who appeared calm, efficient, and easy to talk with. They experienced most members of the staff as friendly, supportive, helpful, understanding, providing honest information about the illness, and answering all their questions and communicating well with the school: “They [nurses and doctors] helped so [that] my sister could die at home.”
Nonetheless, some siblings remembered having experienced unfriendly staff, too many different doctors, and a lack of information: “As a sibling, I was the last to get information, [and] poor information”; “Some doctors were emotionally disengaged.” Several siblings felt as if no one cared—they did not receive any support and pointed out that the school was of no help in their situation.
DISCUSSION
The results from this nationwide study show that bereaved siblings have many positive memories and experiences, even though the death of a brother or sister is a distressing and grievous situation. Moreover, and most importantly, our results show a positive buffering effect in terms of the bereaved siblings' own endurance and personal growth in a stressful and sorrowful life situation, as well as the effect on family cohesion and social support. It appears that the sick siblings, as well as the parents, played important roles in the shaping of their healthy siblings' experiences.
Our results suggest an interconnection between ill brothers' or sisters' physical and mental condition (their symptoms and reactions) and how their healthy siblings experience the support they receive from their own family or healthcare professionals. The ill sibling's condition functions as a buffer, yielding positive experiences or intensifying the negative experiences. Moreover, siblings and parents are closely entwined and can consequently influence each other's grieving processes (Warnick, Reference Warnick2015), both positively and negatively.
Earlier studies investigating bereaved siblings' and parents' reactions and experiences have largely focused on psychopathology and rarely on a health-promoting perspective (Greeff et al., Reference Greeff, Vansteenwegen and Herbiest2011; Norberg et al., Reference Norberg, Lindblad and Boman2005; Hjemdal et al., Reference Hjemdal, Aune and Reinfjell2007). Our study emphasizes bereaved siblings' positive memories and experiences, something we believe is important in increasing our knowledge about the subject of siblings' grief, as well as helping professionals to support, strengthen, and promote bereaved siblings' resilience.
Endurance versus Vulnerability
Our results suggest that the ill brothers' or sisters' strong willpower, good mood, and stamina in their difficult situation influences and has a positive buffering effect on the healthy siblings' memories. Moreover, involvement and shared time together with the ill brother or sister strengthens these positive experiences and memories both in the bereaved sibling and within the family. A study by Woodgate (Reference Woodgate2006) showed that siblings of a brother or sister with cancer perceived that they had minimal involvement in their ill brother's or sister's care and experienced stress and helplessness when faced with their sibling's symptoms. The sibling's lack of involvement in their brothers' or sisters' illness and death could potentially have long-term impact on their coping and bereavement, considering that sibling relationships are among the most enduring relational contexts and affect individual development throughout life (Branje et al., Reference Branje, van Lieshout and van Aken2004; Lövgren et al., Reference Lövgren, Bylund-Grenklo and Jalmsell2016). Nonetheless, individual strengths and resources may influence psychological distress reactions and affect the mental health of healthy siblings' (Eilertsen et al., Reference Eilertsen, Hjemdal and Le2015). In our study, siblings' involvement with their ill brother or sister seems to influence their positive experiences and memories, possibly strengthening the healthy sibling and thus affecting their mental health.
Family Cohesion versus Family Conflicts
The feeling of loss that comes with the death of a sibling can have long-term effects on the well-being of surviving siblings (Warnick, Reference Warnick2015; Sveen et al., Reference Sveen, Eilegård and Steineck2014). This is especially important given the close and long-lasting nature of a sibling relationship. Therefore, family cohesion may contribute toward strengthening healthy siblings' positive experiences. In general, research has shown that family cohesion is a positive factor for coping (Franko et al., Reference Franko, Thompson and Bauserman2008; Grenklo et al., Reference Grenklo, Kreicbergs and Valdimarsdottir2013). This was also emphasized in our study, where siblings expressed the significance of being together as a family unit, having a common goal in supporting the sick sibling, and thereby strengthening the bonds between surviving family members.
Family cohesion acts to bond family members emotionally and may buffer the effects of psychological distress. It may also contribute toward meeting healthy siblings' needs to be acknowledged and not feel invisible, ultimately contributing to better mental health in the bereaved sibling. Eilertsen et al. (Reference Eilertsen, Hjemdal and Le2015) showed that family cohesion is a significant predictor of general mental health for mothers of children who survive acute lymphoblastic leukemia. This may also be true for siblings whose brother or sister has cancer, as shown in other studies (Woodgate, Reference Woodgate2006; Spuij et al., Reference Spuij, van Londen-Huiberts and Boelen2013; Nolbris et al., Reference Nolbris, Enskär and Hellström2014). These studies found that enhanced family support resources can result in more positive outcomes for siblings, making this an important factor for health professionals in promoting family-centered care. Furthermore, Eilertsen et al. (Reference Eilertsen, Eilegård and Steineck2013) reported that most bereaved siblings stated that their family was the most common source of social support during the illness and after death, and that a lack of family support was associated with increased anxiety. Moreover, a small study by Spuij et al. (Reference Spuij, van Londen-Huiberts and Boelen2013) employing a combination of a cognitive behavioral therapy and parental counseling sessions found that prolonged grief disorder symptoms in siblings decreased. In our study, siblings mentioned both positive and negative aspects of experiencing a death in the family, ranging from strengthening the bonds between family members to the hardships of family conflicts. Furthermore, it appears that parents' grief and conflicts between parents may have accentuated healthy siblings' negative memories. However, most siblings emphasized the importance of support from family members.
Growth versus Stagnation
Even though it has been found that bereaved siblings do not report feeling more mature or more self-confident than non-bereaved peers (Eilegård et al., Reference Eilegård, Steineck and Nyberg2013b), most of the siblings in our study reported that they experienced personal growth over the course of their sick sibling's illness and death. Moreover, siblings appear to have gained personal strength and to have experienced positive memories when the bonds between family members grew tighter, as described in the theme on family cohesion.
Healthy siblings' needs to be acknowledged and not feel that they have been forgotten by parents and significant others through the course of their sick sibling's illness and death constituted an especially important finding of our study. However, anxiety, sadness, and grief in the family could also lead to arguments and frustration, even when closeness was desperately needed. Parents' grief and/or conflicts between spouses often appeared to be transferred to the siblings, accentuating healthy siblings' negative memories. A study by Wallin and coworkers (Reference Wallin, Steineck and Nyberg2016) found that bereaved siblings who avoided talking with their parents had a 3 to 4 times higher risk of anxiety. Many siblings emphasized the physical and mental strain their parents experienced, as well as the sadness around the illness and death, as a negative experience.
Professional Support versus Lack of Professional Support
Contributing to a positive experience of an otherwise negative situation is a challenge for the sibling themselves, their family, and the professionals working with and around them. Several studies (Clemente, Reference Clemente2007; Knapp & Contro, Reference Knapp and Contro2009; Pedro et al., Reference Pedro, Galvão and Rocha2008) have emphasized the nurse's essential role in providing social support as well as information to the whole family during the trajectory of a child's illness. Other studies also emphasized siblings' information needs (Havermans & Eiser, Reference Havermans and Eiser1994; Sloper, Reference Sloper2000). However, in a study by Nolbris and Hellström (Reference Nolbris and Hellström2005), all siblings of a child with cancer complained about a lack of information, particularly during the illness. They felt that doctors and nurses did not recognize them as people who needed to be informed routinely and continuously. Moreover, a study by Lövgren et al. (Reference Lövgren, Bylund-Grenklo and Jalmsell2016) highlighted that siblings wanted to be included in the care process from diagnosis until years after bereavement, either because they wanted to be practically involved in their sick siblings' care or because they wanted information and support for themselves.
Bereaved siblings expressed that they felt involved in the terminal phase but unprepared for the fact that the sick child would die, underlining a lack of professional support, and resulting in strong feelings of anxiety that sometimes persisted for years after bereavement (Nolbris & Hellström, Reference Nolbris and Hellström2005). The presence of social support—perceived and actual—may determine how well a sibling and other family members cope (Kupst et al., Reference Kupst, Natta and Richardson1995), possibly also influencing their stamina and endurance, as well as decreasing their experience of loneliness and vulnerability during the grieving process. Siblings' needs for information, as well as the need to be recognized, may also have influenced their experience of professional support.
STRENGTHS AND LIMITATIONS OF THE STUDY
The strength of this study is that it was nationwide in scope, adding to our knowledge about health promotion for siblings of children with cancer throughout the cancer trajectory and during bereavement. However, our study also has potential limitations, one being that the two open-ended statements requesting a description of bereaved siblings' experiences and memories were short and sometimes difficult to interpret. Another is the risk that the siblings' memories may have changed over time, either accentuating or underestimating their positive and negative memories.
CLINICAL IMPLICATIONS
Although the death of a sibling is a profoundly distressing situation and one that will have a life-long impact on the healthy sibling and their family, our results indicate that the risks may be reduced if healthcare professionals gain insight into and focus on a resource-oriented and health-promoting perspective.
In order to give the best possible help and support to siblings of children with cancer, it is important for healthcare and non-healthcare professionals to acknowledge healthy siblings as individuals, focusing on their resources and strengths (Hjemdal et al., Reference Hjemdal, Aune and Reinfjell2007; Eilertsen et al., Reference Eilertsen, Hjemdal and Le2015), and help them become involved in their ill and dying brother's or sister's care. This may in turn promote strong family cohesion, thus contributing by helping bereaved siblings to create more positive experiences with and memories of their sick sibling. Helping siblings in this way could possibly strengthen their resilience and aid their personal growth during the process of their sick sibling's illness and possible death, and also give them the chance to more deeply appreciate the life they are living.
CONCLUSIONS
Our study shows that bereaved siblings also have positive memories and experiences of their brother's and sister's illness and death. The significance of the positive buffering effect on the bereaved siblings' own endurance, personal growth, family cohesion, and social support should be noted. This knowledge can be valuable in showing healthcare professionals the importance of supporting the siblings of children with cancer throughout the cancer trajectory and afterward during bereavement.
FUNDING
Our study was supported by grants from the Swedish Childhood Cancer Foundation, the Swedish Research Council, the Swedish Cancer Society, the Karolinska Institutet, the Gålö Foundation, Ersta Sköndal Bräcke University College–Sweden, and the Norwegian University of Science and Technology. The funding bodies had no involvement in any part of the study.
DISCLOSURES
The authors hereby declare that they have no conflicts of interest to disclose.
