Social support and childhood cancer survivors: A systematic review (2006–2022)

Research has indicated that social support may play a protective role in the face of stress and help children and adolescents cope with the demands and challenges they face on a daily basis during their cancer journey. However, social support tends to reduce over time as survivors overcome their illness despite its ongoing importance even years after treatment has finished. The current review aimed to systematically examine existing evidence on social support in child and adolescent cancer survivors.


| INTRODUCTION
Approximately 300,000 families worldwide annually receive a diagnosis of childhood cancer, 1 with children under the age of 15 accounting for up to 70% of this number. 2 However, survival rates for childhood cancer have dramatically improved over the last 40 years, from approximately 10% to approximately 80% and higher due to major improvements in cancer care and treatment over the years. 3 Nonetheless, survivors 1 and their families face many challenges on a daily basis, such as managing the disease, dealing with the associated treatment, understanding their illness, interacting with healthcare professionals on a regular basis, lengthy hospital stays, aggressive treatment methods and constant uncertainty. [5][6][7] Moreover, in comparison with healthy peers, child and adolescent cancer survivors have been shown to report higher levels of depression and anxiety. 8,9 Social support, despite being a widely used term in the literature, is often difficult to define and conceptualise. 10 However, it is generally agreed that social support is a multidimensional concept, referring to the interpersonal relationships we have with others. 11 Models of social support are generally divided into two categories: structural and functional. 11 Structural models of social support assess the availability and frequency of support from an individual's social network (i.e., direct and indirect social connections with others such as family, friends, neighbours), whereas functional models of social support assess the functionality or degree to which these networks provide social support to the individual. 12 Emotional and instrumental support are the most commonly valued supports by survivors. 13,14 A further distinction can be made between perceived social support and received social support. Perceived social support refers to an individual's own appraisal of their social support, while received social support refers to evaluations of actual levels of support received. 15 Theories of social support have distinguished between four categories of social support, namely, emotional support, instrumental support, informational support, and/or appraisal support. 16 Emotional support is concerned with expressions of empathy, love, care and understanding from others. 16 Instrumental support refers to tangible assistance from others. 16 Informational support is concerned with the sharing of information and advice, particularly during decision making and appraisal support is concerned with the availability of information for self-evaluation and affirmation. 16 Research among children and adolescents has indicated that social support may play a protective role in the face of stress and also in the process of coping with adverse events, such as cancer. 17,18 Likewise, lower levels of anxiety, depression and mood disturbances are generally found among those with greater levels of social support. 19 The two most prevalent social support models guiding research in this area are (1) the main effects model, and (2) the stress-buffering model. 20,21 Under the direct or main effect model, social support has a direct effect on psychological well-being and adjustment, regardless of stress, while the buffering model posits that social support buffers or cushions an individual's reaction to stress. 20,21 Strong social connections are often acknowledged as critical in the successful adjustment of cancer survivors, 22 with parents being acknowledged as the primary source of social support for childhood cancer survivors. 23 Peers have also been shown to be important sources of social support. 22 For example, a study by Ingersgaard and colleagues 24 found a positive relationship between perceived social support from peers and a number of psychological outcomes among adolescents diagnosed with cancer. Similarly, healthcare professionals, such as nurses, oncologists and doctors, play an important role in supporting survivors. 25 Healthcare professionals spend lengthy amounts of time with patients while they are undergoing treatment. 25 However, this support tends to diminish over time as survivors successfully combat their illness, despite the beneficial role social support plays on a number of psychological outcomes, thus highlighting the importance of having a number of strong social connections throughout and subsequent to the course of the cancer journey. 26,27 Research has also indicated that social support may have beneficial effects on quality of life and adjustment to child and adolescent cancer. 18,28 A number of reviews to date have undertaken investigations of social support in families of children with cancer, 29  Therefore, we aimed to synthesise and evaluate quantitative research exploring social support in childhood cancer survivors from the child and adolescents perspective and examine the relationship between social support and outcomes such as quality of life, wellbeing and stress. Furthermore, given the wide disparity in approaches to social support noted in Decker's review, 32 a secondary 820 -DEEGAN ET AL. aim of the review was to examine how social support in the above context is measured in the literature. Findings from this review may offer greater insight into social support in child and adolescents cancer survivors and may also inform future support interventions and provide recommendations to clinicians working in this area in relation to promoting adaptive social support approaches.

| METHOD
This review followed the guidelines outlined by the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) framework. 33 The review was submitted and registered with PROSPERO (No. CRD42022314715). The review has been conducted in accordance with that protocol.

| Search strategy & eligibility criteria
Searches were conducted on the fifth of February 2022 on the following five electronic databases: PsycINFO, PubMed, CINAHL, EMBASE and Web of Science using search terms related to: social support, childhood cancer, cancer and child/adolescent. Mesh terms were used for social support, childhood cancer, cancer, child and adolescent where they existed, as well as free text terms to ensure a comprehensive search of the literature was conducted. A comprehensive search strategy for each database is provided in Supporting Information file S1.
Articles were included if they were full-text, peer reviewed journal articles published in English between 2006 and 2022. Only quantitative studies or mixed methods studies with a quantitative element that could be clearly separated were included. Perceived or received social support had to be described as a variable in the study and measured by a dedicated social support instrument. Included studies had to measure perceived or received social support from the perspective of the child or adolescent with cancer (i.e., not by someone else e.g., family member or friend) in any stage of survivorship from the timepoint of diagnosis on. Children and adolescents of all genders up to the age of 18 years old who were diagnosed with any type of cancer under the age of 18 were included. Studies evaluating a social support instrument were excluded.

| Data screening & extraction
The review was conducted with a team of two reviewers (AD & CB) and two supervisors (SD & PG). After searches were completed, the results were exported to the reference management system, Zotero, 34 where duplicates were removed before being exported to Covidence 35 where abstract/title and full text screening took place.
Each reviewer independently screened titles and abstracts of the retrieved studies for inclusion, while a third reviewer (SD or PG) was available to consult where disagreement occurred. In cases of disagreement, a consensus approach was taken, and the study was included in the full-text screening stage. At the full-text screening stage, studies that did not meet the eligibility criteria were excluded with the reason for exclusion recorded. Where the full text of a study was not available, the author was contacted and the reviewers independently screened the texts and, similarly, a third reviewer was available to consult for any disagreements. Data was extracted by the first author from each paper included in the review onto a standardised, prepared data extraction form that included general characteristics (e.g., authors names, year of publication, title, study design), unique demographic and clinical factors of interest (e.g., age, gender breakdown, setting, cancer diagnosis) and information and major findings relating to social support.

| Quality appraisal
Quality appraisal was conducted by both reviewers using the quantitative quality appraisal tool developed by Dunne and colleagues. 36 Items marked 'yes' received a score of 2, items marked 'partially' received a score of 1 and items marked 'no' received a score of 0.
Articles were scored out of a maximum of 24, with those scoring 17 or above considered high quality, those scoring between 9 and 16 considered adequate quality and those scoring below 9 considered poor quality.

| Synthesis of results
A narrative synthesis approach was selected to analyse the data due to the heterogeneity of social support definitions and measures included. Narrative synthesis is useful for integrating the results of studies to organise and identify patterns across studies from the data extracted. 37

| RESULTS
A total of 8682 abstracts across five databases were identified, 4959 after duplicates were removed, 171 for full-text screening and 10 met the inclusion criteria and were included in this review (see Figure 1). Of those included, seven studies employed quantitative analyses only and three studies employed mixed methods. Table 1 provides a summary of the sample, purpose and social support findings for the reviewed studies.

| Quality appraisal
All the included studies were deemed to have acceptable quality according to the quality appraisal tool, the details of which are DEEGAN ET AL.

| Study characteristics
All of the studies were exploratory and cross-sectional in design.
Studies were conducted in the United States (n = 3), Turkey (n = 2), China (n = 1), the United Kingdom (n = 1), Canada (n = 1), Japan (=1) and Poland (n = 1). Nine studies recruited survivors from paediatric hospitals and centres, while one study recruited participants through a summer camp for children with cancer. 40      Finally, Gilliam and colleagues 47 reported that male survivors were physically more active than females (p = 0.04).

| Age differences relating to social support
Three studies examined age differences relating to social support in childhood cancer survivors, where mixed findings were reported.
Conrad and Altmaier 40 found no significant differences between cancer survivors aged 9-12 years and older survivors aged 13-

| Relationship of social support to other outcomes
The relationship of social support to other outcomes was examined in eight of the reviewed studies. Ekim and Ocakci 44 found a strong positive relationship between perceived social support and posttraumatic growth total scores (p = 0.00). Moreover, there was a significant negative correlation between extending the duration of treatment and total perceived social support scores. Soejima and colleagues 51 found that social support from peers and teachers was positively associated with school re-entry.
Three studies found evidence to support a positive relationship between social support and physical activity levels. 45  Family and friends were reported as the major sources of social support for childhood cancer survivors in the included studies. 38,44,48 Friends become increasingly more important during middle to late childhood, while family support is important at all stages of childhood. 54 Parents were indicated as the most important sources of social support by survivors in two of the reviewed studies. 44,48 This is not surprising, as previous research has indicated that family members, particularly parents, are major sources of social support for children/adolescents with cancer. 23,55 Family members typically live in the same household as the child and, therefore, may spend considerably more time with the child than anyone else. Furthermore, children who are ill tend to have restricted social support systems and/or networks due to their illness and treatment despite having greater social support needs than healthy children. 56 As a result, ill children may be highly dependent on the support they receive from their family and friends even more so than healthy children.
Mixed findings were found in relation to gender differences.
While some studies reported no differences between genders, 42,44 others found that girls tend to report more social support than boys. 40,44 Additional studies are needed using larger and more evenly dispersed samples to elucidate the mixed and inconsistent findings in relation to gender differences for social support among childhood cancer survivors.
Similarly, mixed findings were found in relation to age related differences. Only three of the 10 reviewed studies explored potential age-related differences of included children and adolescents. 30,45,47 These studies differentiated between younger children (8-11 and 9-12 years) and older children/adolescents (12-  Ocakci 44 the support children and adolescents receive tends to decline over time as they successfully manage their illness, however this does not mean that the social support they receive should be halted or given less of a priority than as before. As such, children and adolescents should be provided with opportunities for social interaction at all stages of the cancer journey. 31 It is also likely that social support will be more important at certain stages of a survivor's life; for example, when transitioning from primary to secondary school, as there can be huge gaps in school attendance between survivors and their peers making the transition more difficult for survivors. 58 This highlights the importance of having strong social support during this transition. Likewise, the cancer experience and related late effects can have an impact on the development and maintenance of romantic relationships in young adulthood and beyond, again highlighting the importance of continued support beyond the end of treatment. 59 Moreover, all of the studies were cross-sectional in nature, limiting the ability to make conclusions about the pattern and long-term impact of social support throughout the child's cancer experience.

| Clinical implications
Future research should prioritise longitudinal research design studies that examine social support over time.

| Study limitations
Despite following recommended practice for conducting systematic reviews, a number of limitations were inherent in this review.

| CONCLUSION
The systematic review offers greater insight into the current state of the literature on social support among childhood cancer survivors aged 18 or younger. The evidence from the review highlights that social support, particularly from family and friends, is highly valued by cancer survivors. The findings also demonstrate that social support is related both positively and negatively to a number of psychosocial variables (i.e., posttraumatic growth, psychological stress, physical activity etc). However, mixed findings in relation to gender, age and group differences indicates the need for more research elucidating these findings further. Like previous reviews conducted by Woodgate 13  -831 longitudinal research designs, studying social support as the primary variable of interest and using appropriately developed social support instruments for children and adolescents.