Discussion
The aims of the current study were to evaluate group differences in social adjustment across three groups of children (survivors of ALL, survivor siblings, controls, control siblings), to examine disease-related predictors of social adjustment in survivors, and to explore executive functioning as a potential mediator of these relationships. Results suggested that survivors of ALL had greater social withdrawal (i.e., poorer social adjustment) compared to controls and the siblings of those controls. This finding was consistent with our hypothesis and with previous literature that has identified social adjustment difficulties among survivors of pediatric ALL.6-8 Importantly, when we examined the frequency of those reporting social difficulties within the clinical range (i.e., >60) 26% of survivors did report social withdrawal within the clinical range compared to only 10% of controls. Thus, the social adjustment of survivors of ALL warrants greater clinical and research attention.
Importantly, our hypothesis that executive functioning would be associated with social adjustment among survivors as proposed by the Social Competence Model,9 was supported by our second aim. The current study provides preliminary evidence for the application of the Social Competence Model to survivors of pediatric ALL. What is unique about the current study from previous literature, however, is that none of our current sample of survivors of ALL had received radiotherapy. This finding suggests, therefore, that despite a move towards less toxic therapy for survivors of ALL, social adjustment difficulties remain.
The story does appear to be slightly more complex, however, among survivors of ALL. Although executive function emerged as a significant predictor of social adjustment among survivors of ALL, executive function did not explain differences in social withdrawal between survivors, siblings and controls. This has important implications for the Social Competence Model as well as for our understanding of social adjustment difficulties in survivors of ALL. First, our findings strengthen the applicability of the Social Competence Model by confirming the relationship of executive function to social adjustment, irrespective of acquired brain injury. In other words, this relationship holds among clinical and healthy populations. In order to understand the difference in social adjustment scores between survivors and healthy controls, therefore, consideration of treatment-related variables may be key. For example, it might be important to consider chemotherapy dosage. In addition, little research has explored the indirect effects of a diagnosis of ALL and the consequences of the several years of treatment required to cure ALL and the implications this might have on social development.
The literature exploring non-treatment factors associated with social adjustment in survivors of pediatric ALL is more variable. Some evidence suggests that demographic variables such as ethnicity, marital status, parent education, and household income may play a role in social adjustment outcomes.34-37 The literature examining sex and social adjustment outcomes in survivors of ALL has revealed inconsistent findings.22,37,38 Individual and family factors, such as survivor and parent mental health (e.g., mood, anxiety, posttraumatic growth),39,40 and parenting stress,36 have been associated with social adjustment in this population and require further investigation. Importantly, given our current findings, it would be critical to also explore how such family factors may be related to sibling social adjustment. Knowledge about both treatment and non-treatment factors associated with social adjustment are important to inform risk-based screening guidelines and targets for intervention.
One of the most surprising findings from the study was that survivors did not differ in their social adjustment compared to their siblings. This finding was not consistent with our hypotheses. Certainly, the siblings of pediatric cancer patients are not immune to the experience of cancer within the family and have been shown to exhibit increased levels of emotional distress and behavioral problems.41,42 In addition, previous research among a heterogenous sample of pediatric cancer survivors found no differences between survivors and siblings with respect to social adjustment.43 It is important to keep in mind, however, our population of survivors in the acute survivorship phase (2-7 years post treatment). Knowing the extensive literature documenting the delayed onset of cognitive deficits, so too might we expect to see the social adjustment of survivors worsen the further off treatment they might be.
This study was not without limitations. To begin, we had small samples to test for these effects, specifically for the survivor sibling group. This study is limited by its cross-sectional design which precludes any conclusions to be made about causal factors of social adjustment difficulties. Mediation models typically imply a sequence of events occurring over time; in the current study we were not able to capture potential ‘true’ mediation given this design limitation. Longitudinal research is needed to explore the trajectory of social adjustment difficulties for survivors of pediatric ALL and to being to explore mechanisms that may explain this relationship. In addition, although the control groups were a relative strength of the study, we did not compare survivors of ALL to survivors of cancer who do not receive toxic therapies directed at the CNS. Survivors of childhood cancer who did not receive toxic therapies directed at the CNS would have provided a more conservative control group as they have both experienced a traumatic cancer diagnosis and been treated with chemotherapies that lead to socially limiting side effects (e.g., hair loss and compromised immunity limiting their ability to be in public places or school environments). However, without having received therapies believed to be centrally neurotoxic, the effects of CNS-directed treatment on social adjustment could have been more clearly differentiated in survivors of ALL. In addition, while we were able to examine outcomes of survivors based on disease risk status, we did not have specific dosage information for therapies known to be neuro-toxic, including methotrexate and intrathecal chemotherapies. Finally, the study did not explore non-treatment-related factors that might also contribute to social functioning in this population. Future research should aim to explore the other factors that might be related to social function among this population with a specific focus on both treatment- and non-treatment-related factors.
In conclusion, survivors of pediatric ALL are at risk of more social difficulties compared to non-cancer controls. Predictors of social adjustment among survivors included executive functioning and time off treatment. However, executive functioning did not explain any of the observed group differences in social adjustment. This is an important finding among a sample of survivors 2-7 years from the completion of therapy who did not receive CRT and highlights the potential persistent toxicity of therapy for pediatric ALL despite efforts to reduce treatment-related toxicity. Based on the current findings, survivors of pediatric ALL presenting to follow-up programs should be screened for difficulties with social adjustment. Future research should aim to more comprehensively examine the potential multiple factors that may contribute to poorer social adjustment outcomes in order to identify modifiable targets for interventions.
Acknowledgements: This work was supported by the Alberta Children’s Hospital Research Institute 2015-16 ARC Award in Family Centered Care.
Conflict of Interest Statement
None to disclose.
Data Availability Statement
The data that support the findings of this study are available from the corresponding author upon reasonable request.
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