Journal of Pediatric Psychology Advance Access originally published online on August 14, 2007
Journal of Pediatric Psychology 2007 32(9):1055-1066; doi:10.1093/jpepsy/jsm060
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Adaptive Style in Children with Cancer: Implications for a Positive Psychology Approach
Division of Behavioral Medicine, St. Jude Children's Research Hospital, Memphis, TN
All correspondence concerning this article should be addressed to Sean Phipps, PhD, Member, Division of Behavioral Medicine, St. Jude Children's; Research Hospital, 332 N. Lauderdale, Memphis, E-mail: sean.phipps{at}stjude.org.
| Abstract |
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Objective To describe the adaptive style paradigm as a heuristic model for understanding the very positive psychosocial adjustment that has been observed in children with cancer, and to integrate findings regarding repressive adaptive style into a broader positive psychology framework. Method A selective review of the literature on adaptive style, and its relevance to outcomes of depressive symptoms, posttraumatic stress symptoms, somatic distress, and health-related quality of life in children with cancer. Results Studies have found children with cancer report low levels of psychological distress. Adaptive style is a much stronger predictor of psychosocial outcomes than is health history. Conclusion Children with cancer represent a flourishing population. A repressive adaptive style is one pathway to resilience in this population. Additional constructs from the domain of positive psychology are reviewed, and a positive psychology model is suggested as a framework for guiding future research in this area.
Key words: adaptive style; benefit finding; childhood cancer; optimism; positive psychology; repression; resilience.
Pediatric cancer has long been considered a highly stressful, burdensome, and even traumatic experience for those children who must face it, and understandably so (Kazak, 2005
This article introduces the adaptive style paradigm as a framework for understanding the positive adjustment of children with cancer. A selective review of the literature is presented, indicating that a preponderance of the available data supports a view of children with cancer as exceptionally well adjusted. We then describe the adaptive style paradigm and review studies that indicate a high prevalence of a repressive adaptive style in children with cancer. Studies that examine the relationship of adaptive style to outcomes of depression, posttraumatic stress, somatic symptoms, and quality of life are presented, which suggest lower levels of distress among those with a repressive adaptive style. Subsequently, ongoing work from our laboratory is presented, where preliminary findings suggest that there are not significant physiological costs of maintaining a repressive adaptive style. Repressive adaptation is thus presented as a pathway to resilience in children with cancer, who, as a group, are construed as a thriving or flourishing population. Adopting a positive psychology framework, repressive adaptation is presented as only one of several potential mechanisms for the psychosocial thriving observed in children with cancer. The article concludes with a brief review of other constructs from positive psychology, pointing to their potential relevance for children with cancer, and as targets for future research.
| Children with Cancer Appear Exceptionally Well-Adjusted |
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To illustrate the surprisingly positive adjustment of children with cancer, consider the outcome of depressive symptomatology. The majority of published studies have shown that children with cancer report low symptom levels, in many cases significantly lower than either test norms or healthy peer controls (Bennett, 1994
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In considering these findings, a fundamental question becomes—are the self-reports of low depression from these children "real," or might they be in some way distorted or reflective of "illusory mental health" (Shedler, Mayman, & Manis, 1993
| The Adaptive Style Paradigm |
|---|
The adaptive style paradigm, initially developed by Weinberger and colleagues (Weinberger, 1990
Canning and colleagues documented a high prevalence of repressive adaptive style in children with cancer, which explained, in part, the low levels of self-reported depressive symptoms in their cancer sample (Canning et al., 1992
). We replicated these findings in a large cross-sectional sample of children and adolescents with cancer, showing that the incidence of repressive adaptive style in the cancer group was double that of healthy controls (36–18%), and also conversely that the cancer group demonstrated low levels of a "high anxious" style relative to healthy controls (Phipps & Srivastava, 1997
). These findings have been replicated several times by our group and others, both in children with cancer and long-term childhood cancer survivors (Erickson & Steiner, 2001
; Jurbergs, Long, Hudson, & Phipps, 2007
; Phipps, Larson, Long, & Rai, 2006
; Phipps, Steele, Hall, & Leigh, 2001
).
The genesis of the adaptive style differences between children with cancer and healthy children is unclear. A reasonable explanation might be that it represents a contingent response to the stresses of illness, such that patients facing the threat of cancer mobilize a more defensive posture, which if successful, would also lower anxiety. Some evidence for this in adults comes from two studies of women pre- and post-diagnostic procedures for breast cancer, where only women subsequently diagnosed with a malignancy showed increases in defensiveness and repressive adaptation (Kreitler, Chaitchik, & Kreitler, 1993
; Zachariae et al., 2004
). On the other hand, adaptive style has been shown to be a fairly stable personality trait, and, in children with cancer, the increased prevalence of a repressive adaptive style has been demonstrated within 2–3 weeks of diagnosis, remaining stable across the first year of treatment (Phipps et al., 2001
). Because of the low incidence of childhood cancer, a definitive answer to the question of the genesis of cancer–control differences in adaptive style may not be feasible. However, the higher prevalence of repressive adaptation in children with cancer appears well established, and thus examining the implications of adaptive style for patient adjustment, quality of life and other health outcomes in this population is crucial.
| Adapative Style and Depressive Symptoms |
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Study of the effects of adaptive style on patient adjustment began with the outcome of depression (Canning et al., 1992
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| Adaptive Style and PTSS |
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The low levels of depressive symptoms observed in children with cancer prompted a move away from traditional measures of psychopathology, which were thought to be inadequate to capture the specific and persistent experiences of children in this context (Kazak, 2005
The effect of adaptive style on patient reported PTSS was examined by our group in a cross-sectional study of pediatric cancer patients, survivors, and young adult survivors. Although we found that time elapsed from diagnosis was a significant determinant of PTSS, with recently diagnosed and on-treatment patients reporting higher symptoms levels than survivors, we also found that adaptive style was the most powerful determinant of PTSS outcomes (Phipps et al., 2006
; Phipps, Long, Hudson, & Rai, 2005
). Using the UCLA PTSD Index for DSM-IV (Pynoos, Rodriguez, Steinberg, Stuber, & Frederick, 1998
), we found that patients identified as repressor and low anxious report significantly less PTSS than do the high anxious or defensive high anxious patients. Although repressors reported the lowest level of symptoms, they did not differ significantly from the low anxious patients. Thus, it appears that for PTSS outcomes, trait anxiety level is the primary factor accounting for the observed effects, and defensiveness plays a lesser role (Phipps et al., 2006
). These findings have been replicated in a follow-up study that included a healthy control group. In that study, we found that children with cancer reported significantly fewer symptoms of intrusion/re-experiencing than did their healthy peers, while also reporting significantly greater symptom levels of numbing/avoidance (Phipps, Jurbergs, & Long, 2007
). However, there were no differences in total PTSS between cancer and control groups, who obtained almost identical mean scores. In contrast, large and significant effects of adaptive style were found on total PTSS and all symptom cluster subscales (Fig. 1B). In sum, our findings are consistent with a preponderance of the existing literature, suggesting that PTSS is simply not elevated in children with cancer. In addition, PTSS outcomes are much more dependent on preexisting personality traits such as adaptive style than on health history. The low levels of PTSS observed provides further evidence that predisposition to pathology is low in this population, or conversely, that children with cancer are resilient against the stresses of their illness.
| Adaptive Style and Somatic Symptoms |
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The relationship of adaptive style to affective distress outcomes, such as depression and PTSS raises the question of whether these effects extend to other domains, such as physical or somatic symptoms. Given the tendency of repressors to report low levels of psychological distress, one might predict they would report lower levels of somatic distress as well. However, studies in adults have documented that repressors exhibit heightened levels of physiological reactivity to laboratory stress, and that they appear to be at higher risk for a number of adverse health outcomes, such as tension and migraine headaches, ulcers, allergies, and hypertension (Schwartz, 1990
The absence of cancer–control differences in somatic symptoms suggests underreporting of physical symptoms in cancer survivors, influenced in part by adaptive style. Although some patients may refuse to acknowledge certain symptoms as a means of avoiding anxiety, it is unlikely that these children are deliberately denying symptoms of which they are aware, i.e., they are not "faking good." Rather, it appears they believe they are reporting in a generally honest manner, but several factors may promote a response bias that minimizes symptoms. One issue is "framing," whereby children who have experienced severe, life threatening symptoms in the past may tend to overlook or minimize more common and less severe physical symptoms, which are deemed unimportant. Alternately, the children may be reporting accurately based on their awareness of their physical state, but that awareness may be somewhat attenuated, as a function of adaptive style. Some have hypothesized that the attentional processes of repressors are altered, such that they are less likely to attend to threatening stimuli, and are poorly attuned to their physiological state (Schwartz, 1990
; Weinberger, 1990
). Thus, when children with cancer appear to underreport symptoms, they may either be genuinely unaware of a symptom, or have a limited awareness such that even when they acknowledge symptoms, they minimize their severity or impact.
| Adaptive Style and HRQL |
|---|
Another outcome that has grown in importance in pediatric oncology settings over the past decade is health-related quality of life (HRQL, Feeny, Furlong, Mulhern, Barr, & Hudson, 1999
The impact of adaptive style on self-reported HRQL outcomes was examined in our laboratory in a study assessing a cross-sectional population of children with cancer (N = 199) and a healthy acquaintance control group (N = 108). Children completed the Children's Health Questionnaire (CHQ), and one parent of each child also completed the CHQ parent report form (Landgraf, Abetz, & Ware, 1999
). By patient report, significant cancer–control differences were observed on the physical health domains of the CHQ (physical functioning, body pain, and general health perceptions), with, as expected, the cancer group reporting poorer health than controls (Russell, Hudson, Long, & Phipps, 2006
). However, children with cancer and healthy children did not differ significantly on the psychosocial health domains (mental health, self-esteem, general behavior) of the CHQ. In contrast, differences in self-reported HRQL outcomes as a function of adaptive style were seen on all CHQ outcomes (Jurbergs, Russell, Long, & Phipps, in press
). The effect of adaptive style was smaller on the physical health domains of HRQL, where effects were much greater for health status (cancer versus control), whereas adaptive style effects were much larger on the psychosocial domains, with children identified as repressors reporting the best HRQL regardless of health status (Fig. 1D).
This study also examined the relationship between parent and child-reported HRQL as an effect of child adaptive style. Although the consistency of parent and child reports was good, as evidenced by moderate correlations between parent and child scores and relatively small mean differences, adaptive style was nonetheless significantly predictive of discrepancies between parent and child report of HRQL on the psychosocial domains of the CHQ (Jurbergs et al., 2007b). Repressor and low anxious children reported better HRQL than did their parents, while high anxious children reported poorer HRQL than their parents, regardless of health status. Adaptive style was not predictive of parent–child differences on the physical health domains.
To summarize, adaptive style appears to be a significant determinant on many of the most common self-report outcomes in pediatric psychooncology research, including symptoms of depression, PTSD, somatic distress, and HRQL. Child health status (cancer versus healthy) appears to be unrelated, or only minimally related to the most common psychosocial outcomes, although it is a significant determinant of the physical health domains of HRQL. That expected differences between children with cancer and healthy children on physical health outcomes are, in fact, observed, suggests that children with cancer are reporting reasonably accurately on more objective, readily observed outcomes i.e., they are not just "faking good." This highlights the importance of appreciating the the nature of the self-reported outcome being examined, in terms of the relative level of subjectivity.
In considering HRQL, for example, items asking children to rate their mood, hopefulness about the future, and how able they are to have fun, which load onto the psychosocial domains, are clearly more subjective than items asking about concrete, functional aspects of HRQL that are part of the physical domains, such as how difficult it is to walk up a flight of stairs, bend, or bathe themselves. The effects of adaptive style are more likely to be observed as the subjectivity of the measure increases. One key issue may be the construal of "distress," which, by definition, implies a subjective component. In some contexts, it may be desirable to reduce subjectivity in measurement of certain HRQL outcomes, but typically, it is neither possible nor desirable to eliminate all subjectivity in HRQL measures. Thus, clinicians and researchers should be aware of the influence of adaptive style on subjectively assessed outcomes.
| Is There a Cost for the Low Levels of Distress in Children with Cancer? |
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The literature documents low levels of distress with adequate to exceptional adjustment in children with cancer, and cancer–control differences in adaptive style help to explain some of these findings. This raises the question of whether there might be a cost, or "price to pay" for the avoidance of distress in children with cancer, particularly those with a repressive adaptive style? Research done in healthy adult populations suggests that repressors show increased physiological reactivity to stress, and are at higher risk for a number of adverse health outcomes (Schwartz, 1990
Study one used a case-control design, enrolling a cohort of children who had completed cancer therapy (N = 120), and a group of healthy children (N = 120) matched on age, race, gender, and SES. All children completed measures of adaptive style and underwent three verbal stress tasks, while measures of heart rate, heart rate variability, blood pressure, skin conductance, muscle tension, and salivary cortisol were obtained. Although good physiological reactivity to all stress tasks was obtained, correlations between physiologic indices, self-reported stress, and adaptive style were poor. Analyses completed thus far examining the effects of adaptive style on physiological reactivity have revealed mostly null findings. A few findings that approached or reached significance have been in the opposite direction of our a priori hypotheses, i.e., with high defensive children showing lower levels of physiological reactivity during the stress tasks. Study two enrolled a cohort of children with newly diagnosed cancer (N = 137). These patients completed the same battery of measures and laboratory assessments as in study one, and then were followed longitudinally to examine whether adaptive style was predictive of medical outcomes (number of hospital admissions, days in hospital, narcotic and antiemetic medication usage, and specific toxicities) during the first 6 months of treatment. Preliminary analyses have failed to show a significant association between patient adaptive style and these medical outcomes. In regards to hospitalization data, findings were null, but there were trends observed that were again opposite of the predicted direction—children identified as repressors experienced the lowest number of admissions and the fewest days in hospital.
These findings are inconsistent with our a priori hypotheses and what has been previously reported in adults. However, they do begin to address one of our primary questions—is there a physiological price to pay for the low distress reported by those children with a repressive adaptive style? Based on these preliminary findings, the answer appears to be a tentative "No." The results suggest that children identified as repressors do not appear to experience adverse effects of this coping style, as manifest either in increased physiological reactivity or poorer medical outcomes. Since children identified as repressors report lower levels of distress and generally better psychosocial outcomes without apparent adverse effects, we have begun to revise our thinking about this construct, viewing it as a potential pathway to resilience in children with cancer.
| Repressive Adaptative Style as a Pathway to Resilience in Children with Cancer |
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The notion of a repressive adaptive style as a pathway to resilience has been described previously in the context of caregiver bereavement in adults (Bonanno, 2004
The theoretical and empirical work addressing resilience in children has been focused largely on children facing chronic and global social stressors, such as poverty, neglect, and violence (Garmezy, 1991
; Luthar, Cicchetti, & Becker, 2000
). In contrast, much of the work in adult resilience has addressed relatively circumscribed traumatic events (such as bereavement) that occur in the context of otherwise normal or adequate social circumstances (Bonanno, 2005
). Childhood cancer presents an opportunity to extend childhood resilience research into new contexts, and to incorporate additional models from the global domain of positive psychology. The past decade has seen a shift in focus away from psychopathology towards positive psychology and a study of human strengths, with a dramatic increase in research in this area (Linley & Joseph, 2004
; Seligman & Csikszentmahalyi, 2000
; Snyder & Lopez, 2002
). A few constructs from this domain that might be particularly relevant to children with cancer include dispositional optimism, hope, benefit finding, posttraumatic growth, spiritual coping, and the assessment of positive affect. Many of these variables are likely to have overlap with the construct of adaptive style.
| Positive Psychology Models and Childhood Cancer |
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Dispositional optimism, defined as having positive expectations for the future (Scheier & Carver, 1985
Another area that would appear ripe for further study relates to the constructs of posttraumatic growth and benefit finding. Although these might be best considered as distinct constructs, there has been some conceptual confusion in the literature, and the terms are often used interchangeably. Regardless, the onset of a serious illness has been recognized as a potential catalyst for positive change, and this has been a burgeoning area of research (Kangas, Henry, & Bryant, 2002
; Sears, Stanton, & Danoff-Burg, 2003
). Many cancer patients report gains, such as a deepened sense of purpose, closer relationships with family and friends, a reappraisal of their life's priorities, and an enhanced spiritual life (Affleck & Tennen, 1996
; Carver & Antoni, 2004
; Cordova, Cunningham, Carlson, & Andrykowski, 2001
; Tomich & Helgeson 2004
). Again, most of the research has been conducted with adult populations and research in children lags behind, partly due to lack of appropriate child measures. Recently, two new measures have been developed specifically in pediatric cancer populations, one addressing the construct of posttraumatic growth (Barakat, Alderfer, & Kazak, 2006
), and the other, from our laboratory addressing the more circumscribed construct of benefit finding (Phipps, Ogden, & Long, in press
). The development of such measures will allow for additional research to proceed in this potentially fruitful area.
One of the conceptual issues critical in the shift to a positive psychology focus is recognition of positive and negative affect as relatively independent constructs and not opposite ends of a single continuum (Watson & Clark, 1997
). The pediatric psychology and psychooncology literature, as virtually all of clinical psychology, has been largely focused on negative affect, mood disturbance and distress, to the exclusion of any measure of positive emotions. The importance of positive emotions is now being recognized, and the independence of positive and negative emotions has several implications. For example, positive and negative psychological states can occur contiguously or even simultaneously, and positive psychological states can occur in the midst of highly stressful circumstances such as terminal illness or bereavement (Folkman & Moskowitz, 2000
). Further, the experience of positive psychological states can serve to promote emotional and physical well-being, both by undoing the deleterious effects of negative states and serving as a buffer against future stress and negative experience (Fredrickson, 2001
; Tugade, Fredrickson, & Barrett, 2004
).
Fredrickson's (2001
) "broaden and build theory of positive emotions" posits that negative emotions are associated with specific action tendencies that require autonomic activation to prepare for the activity (e.g., fight, flight). Positive emotions are not associated with any specific action tendencies, and as such, can serve to reduce autonomic activation, undoing the physiological preparation for action, and returning the body to a resting equilibrium. In this way, positive emotional states can serve as an antidote to the effects of negative emotions (Fredrickson, 2001
; Tugade et al., 2004
). According to this theory, the experience of positive emotions results in an increment in enduring personal resources, both physical and psychological, that can be drawn upon later, in other contexts and other emotional states. Many of the positive outcomes associated with dispositional optimism may reflect the fact that optimists are more likely to experience positive emotions (Affleck & Tennen, 1996
). In a similar vein, Folkman's research has indicated that people coping successfully with severe stress seek naturally to create positive emotional states, both to gain relief from stress and gather strength to face adversity (Folkman & Moskowitz, 2000
; Moskowitz, Folkman & Acree, 2003
). A key finding from her studies is that positive emotional experiences need not be intense nor prolonged to produce their beneficial effects (Moskowitz et al., 2003
).
These lines of thinking have yet to be explored in pediatric oncology. Certainly, our adaptive style work might lead to the hypothesis that those children with a repressive adaptive style are more likely to experience positive emotional states. But beyond adaptive styles, there may be aspects of the childhood cancer experience in general that are associated with an increased frequency of positive affects, even if these are transitory, and embedded in a context that is also threatening. Researchers in this area are encouraged to include measures of positive affect in their study designs, and to attend to aspects of the patients milieu that might be associated with fluctuations in daily emotional experiences.
In sum, since the introduction of self-report methodologies, children with cancer have demonstrated surprising adjustment, faring as well as, and often better than, healthy peers on outcomes such as depression, anxiety, posttraumatic stress, and quality of life. The adaptive style paradigm has provided a heuristic model for understanding these findings. Children with cancer consistently demonstrate higher levels of a repressive adaptive style, which serves to explain in part, their low levels of distress/pathology and excellent adjustment. Research by our group suggests that the positive adjustment of children with cancer in general, and those with a repressive adaptive style, in particular is not reflective of illusory mental health, nor is it associated with significant risks or adverse health outcomes. Thus, we have come to view children with cancer as a flourishing population, and repressive adaptation as one pathway to resilience in this population. Additional pathways and mechanisms for promoting and maintaining resilient outcomes are likely to be found, and constructs from positive psychology, such as optimism, posttraumatic growth, benefit finding, and discrete assessment of positive emotions provide fruitful starting points. Incorporating a positive psychology framework opens the door wide for range of research questions. We hope that this discussion will stimulate new research efforts on these and other positive psychology constructs in the future.
| Acknowledgment |
|---|
Supported in part by a grant from the National Institutes of Health (R01 CA82378) and by the American Lebanese-Syrian Association Charities (ALSAC).
Conflict of Interest: None declared.
Received January 28, 2007; revision received June 27, 2007; accepted July 9, 2007
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