The Self-Reported Experiences of Siblings Who Have a Brother or Sister Diagnosed with Childhood Cancer: a Systematic Review Protocol of Qualitative Evidence

Review question/objective The overall objective of this review is to promote a broader understanding of the experiences of siblings of a brother or sister diagnosed with childhood cancer. The specific objectives include to synthesize existing findings and to summarize the best available evidence to increase the overall understanding of what it is like to live with a brother or sister with cancer. The recommendations derived from this study may provide further information for clinical nursing to understand the needs of siblings who have a brother or sister diagnosed with childhood cancer, to offer evidence based recommendations toward providing appropriate care and support for these siblings of children with cancer, to promote their adaptation and mental health, and to provide the families of children with cancer care reference, to promote a family-centered care model. The review questions are: 1. What are the experiences of siblings who have a brother or sister diagnosed with a childhood cancer? 2. What are the experiences of siblings who have a brother or sister diagnosed with a childhood cancer regarding the impact on their lives? Background Cancer is the second most common cause of death, after accidents, in childhood and a significant public health problem in developed countries1. Owing to highly specific diagnostic procedures and the introduction and continuous improvement of multimodal treatment strategies, the past decades have led to dramatic improvements in survival rates for most types of childhood cancer with a marked rise in the probability of cure1, 2. Childhood cancer is a highly stressful experience that can challenge and disrupt the family dynamic and its members3. As cancer is a life threatening illness, when a child is diagnosed with cancer the whole family is affected4, 5. With the need to care for an ill child, a family is forced to change the normal daily life routines, whilst also trying to maintain a quasi-normal way of life5, 6. Many of the family stresses and changes accompanying childhood cancer have a severe impact on siblings7. The family is viewed as an interactional system. Family members are connected to each other, exhibit coherent behaviors, and share some degree of interdependence7, 8. Families are in constant interaction with their physical, social, and cultural environment9. A layering of subsystems is found within family systems, such as the spouse subsystem, the parent-child subsystem, the sibling subsystem, etc7, 9. Therefore the family is a complex, integrated whole. Within a family system, patterns of interdependence result in all members of the family system being impacted by change in one another7. When a child in the family is stricken with a serious disease, almost every family enters into a crisis situation8. The family must adapt to accommodate the ill child's needs. Thus, the family system must be dynamic, and there is often interdependence among its members. The sibling subsystem is an important family subsystem in which a child and his or her brothers and sisters interact as they grow up. The relationships of siblings serve as the first social-skill learning for children. Normally the sibling relationship helps a child learn how to share, support, become angry, negotiate, compromise, compete, cooperate, and imitate each other9, 10. The closeness that develops among them eventually leads to a very special, life-long relationship. However, when one child becomes ill, the other healthy siblings may suffer with confused feelings, as the relationship between them and the ill child changes4. When parents focus their attention on the child diagnosed with cancer, the parents may neglect the other child at home and healthy siblings may be confronted with decreased physical and emotional availability of their parents5. The impact on the healthy sibling/s include: (a) change in psychological status, i.e., increase in worry about life, anxiety about the disease, and negative self-concepts (b) change in family interaction, i.e., relationship with mother, relationship with father, relationship with other siblings (c) change in social relationship, i.e. hunger for companions, changing recreation9. Prchal and Landolt5 conducted a qualitative study to determine the experiences of siblings of pediatric cancer patients in different areas of life in the first half-year after the cancer diagnosis. They investigated five areas of the siblings' daily life: hospital, school, peers, family life, and ill child. Siblings were very much aware of the consequences of the illness and the treatment for the ill child. They observed their brothers' or sisters' appearance, suffering, behavior change, and their being the center of attention. Siblings described how family life after the cancer diagnosis was determined by the ill child's treatment protocol, resulting in their parents' regular absence and reduced family activities. Living together with a sick brother or sister also resulted in sibling jealousy and evoked intrusive worries about the ill child's health and prognosis. Interviewed siblings also expressed worries about their brother's or sister's altered behavior, and it was difficult for siblings to observe the ill child's changed appearance. If the child was hospitalized, siblings also reported being affected by other patients in the hospital. Witnessing other ill children suffering, observing their altered appearance, or even hearing about the death of a patient was difficult for siblings. Further difficulties emerged at school, with siblings reporting a decline in academic performance, and an aversion to taking part in everyday life during the time they felt the need to support the ill child. Siblings' peer activities were reduced and difficult to maintain because of the changed situation in the family. However, not all the experiences of siblings were negative. Siblings accounts indicated that supportive relationships with peers were resources that helped them cope with the situation. Siblings also reported increased closeness and a strengthening of bonds between family members after the experience of acute illness in the family. This finding of positive experiences is consistent with results of earlier studies6, 11. In a study by Iles12, five healthy school age siblings of children with cancer were interviewed. The findings indicated themes which included the change of interpersonal relationships, the change of self concept, and loss and growth. The loss experience of healthy siblings included disruption of interpersonal relationships (especially with parents, siblings and peers), physical dysfunctions, and disturbances in the routine of family life. These circumstances provided opportunities for the emotional growth of the siblings involved. Siblings perceived some positive effects of the cancer experience, such as increased empathy for parents needs. A literature review by Wilkins and Woodgate13 on the childhood cancer experience from the perspective of siblings examined 27 articles from 1979 to 2004. In this study, the most frequently used research design was descriptive exploratory. The sampling methods included convenience, theoretical purposive, and random. Most studies employed interviews for data collection. The most commonly used qualitative data analysis procedures were the constant comparative method and content analysis. The number of sibling participants across studies ranged from one to 254. Sixty-three percent of the studies included both brothers and sisters. Siblings' ages ranged from five to 40 years, with the majority between the ages of seven and 18 years. The findings were categorized into three themes: (a) Siblings perceived tremendous change in family relationships and their family dynamics and routines, including losses (e.g. losses of parental attention, normal family roles, sense of self) and gains (e.g. increased family closeness, independence, maturity, empathy); (b) intense feelings (e.g. sadness, loneliness, rejection, anxiety, anger, jealousy, guilt), and (c) unmet sibling needs (e.g. needs for open and honest communication, more information and involvement in the treatment process, and greater support maintaining personal interests and activities). These finding highlights the fact that siblings missed their own and their family's usual activities and routines, and they often felt neglected. It also highlighted that it was important for siblings to receive information about the ill child's cancer and treatment14. Furthermore, siblings perceived intense feelings, these results, like other studies have demonstrated that siblings of children with cancer experience negative emotional reactions in themselves and family members15, 16, such as worry about their ill brother or sister17, increased sibling rivalry, anger and frustration, rejection and guilt6; feelings of hate, jealousy and isolation11. To understand the experience of a sibling who has a brother or sister diagnosed with cancer, it is important to allow them to tell the stories of their experience in their own words5. Approaching the study of siblings' experiences from the qualitative research paradigm provides valuable insights into the changes siblings encounter, the intensity of their feelings and the resources that they find helpful13. Qualitative meta-synthesis is a useful method for bringing together studies that explore the same or closely related phenomena. The goals of qualitative meta-synthesis are theory development, higher level abstraction, and generalizability in order to make qualitative findings more accessible for application in practice18, 19. Qualitative meta-synthesis increases the usefulness of constituent study findings by creating more comprehensive views of phenomena19. Although there is qualitative research available that discusses the experience of siblings of children with cancer, synthesizing the data into a systematic review may provide a better understanding of the overall experiences of siblings of children with cancer. This may facilitate the development of knowledge that would be useful to pediatric nurses and those involved in care of the children. Furthermore, knowledge about siblings' perspective of the childhood cancer may provide insight into the experience of the family system. The study's findings may highlight the philosophy that nursing care needs to be family-centered. An initial search of CINAHL, MEDLINE/PubMed, Eric, and The Joanna Briggs Institute Library of Systematic Reviews was conducted to identify any previous systematic review on this topic. There was a systematic literature review by Wilkins and Woodgate13 on the childhood cancer experience from the perspective of siblings. The review focused on the contributions of qualitative research in advancing our understanding of the childhood cancer experience from the perspective of siblings. The focus of the research was to review qualitative studies on the broad topic of siblings of children with cancer, siblings' ages ranged from five to 40 years. However, the present review differs from Wilkins and Woodgate13 in several significant ways. Firstly, the review aims to promote overall understanding of the experience of siblings' of a brother or sister diagnosed with childhood cancer. Secondly, the review will be conducted on the experiences of siblings aged from six to 20 years, who have a brother or sister diagnosed with a childhood cancer and their voice that describes the experience. Thirdly, this review aims to cover a wider range of databases and cultures than the previous review, and to review literature in both the Chinese and English languages.