Disability, stress, and coping theories recognize that stressors, such as cancer, can pose a significant threat to child and family adjustment (Wallander and Varni 1992; Compas et al. 2012). The degree of perceived threat and controllability may influence an individual’s choice of coping strategies to manage the stressor. Research suggests that coping strategies focused on disengaging from the stressor (e.g., denial, wishful thinking) increase risk for difficulties, while active engagement strategies that aim to change the situation or adapt to it (e.g., problem solving, acceptance) tend to be associated with resilience (Compas et al. 2012; Connor-Smith et al. 2000). Thus, stressors, such as a cancer diagnosis, have important implications for coping and the emergence of psychopathology or successful adaptation in children and families.

Family systems and socio-ecological models emphasize that the stress of childhood cancer affects all members of the family, as individuals make adjustments to accommodate the illness and treatment in the family system (Alderfer and Kazak 2006). A child’s diagnosis occurs within the context of a family that has preexisting patterns of relationships and interactions. This climate, or the common values, rules, and beliefs within the family, provides a framework for how parents and children respond to one another about the challenges presented throughout treatment and thereafter. For example, family environment influences how openly the family speaks about the diagnosis, shares emotion and affect, as well as collaborates in decision making. Family members are interdependent, such that a person’s adjustment is influenced not only by intrinsic characteristics, but also by the significant others in his or her life. In other words, family functioning may set the stage for how the family will manage cancer-related stress as a system.

Consideration of child development is also important in the context of cancer. As a growing number of childhood cancer survivors mature into adults, we have gained a better understanding of the evolving and lasting impact of cancer on the developing child. Because the needs of children with cancer are ongoing and complex, research and care are optimized when considered within a life-span developmental perspective. This approach requires sensitivity to the dynamic context of the child and family’s illness experience over time (Holmbeck et al. 2010; Kazak 2001). Transitions between developmental periods are also important, as major changes in social roles and contexts can alter the course of physical and psychosocial well-being. For example, the child’s age or the timing of diagnosis and treatment are important with respect to risk for psychosocial effects and provision of appropriate supportive care. The developmental context of cancer has particular relevance for the child’s concept of illness and death, medical knowledge, and involvement in self-care and decision making into survivorship or at the end of life. Finally, there is evidence that childhood cancer can affect the transition between developmental periods after diagnosis, including the attainment of socially valued milestones (e.g., graduation, employment).

As many children with bone tumors are diagnosed in the second decade of life, it is helpful to acknowledge the primary developmental tasks of adolescence and emerging adulthood (i.e., ages 18–25). These years are characterized by numerous biological, psychological, and social changes (Feldman and Elliott 1993). A greater capacity for abstract thought, higher-order reasoning, perspective taking, and emotion regulation evolves. Socially, adolescents develop greater autonomy from parents and gravitate toward the peer group. Psychosexual development includes dating, sexual exploration, and more intimate emotional connections with romantic partners. At the same time, youth are also at higher risk for psychopathology, risky health behaviors, and nonadherence to medical regimens (Shaw 2001; Gibbons et al. 2012; Cicchetti and Rogosch 2002). Many health behaviors (e.g., nutrition, exercise) decline markedly in adolescence (Dumith et al. 2011; Lytle and Kubik 2003), and experimentation in risk-taking behaviors (e.g., alcohol use, unprotected sex) is common, further blurring the distinction between normative and abnormal functioning during this time (Cicchetti and Rogosch 2002).

During emerging adulthood, one gains increasing independence, acquires more responsibility, and forms an identity that will likely endure throughout adulthood (Arnett 2000). The decisions and actions that occur during this time can affect education levels, occupational attainment, and social status across the life span (Chisholm and Hurrelmann 1995). Typically, parent–child relationships improve in early adulthood as children become more independent, geographically distant (e.g., away at college), and similar to their parents with regard to roles and responsibilities (e.g., adult work or spousal roles) (Buhl 2007). Hallmark characteristics of this period are exploration and multiple life changes, such as dating different partners, moving frequently, seeking temporary or part-time employment, and pursuing various educational alternatives. Therefore, it is valuable to understand the challenges families face in balancing the demands of cancer with the normal developmental tasks of adolescence and emerging adulthood.

In general, children with cancer may have an increased risk for internalizing problems (e.g., depressed mood, anxiety) near diagnosis, but these symptoms tend to dissipate over the first year (Sawyer et al. 2000). Although most survivors are not at risk for severe psychopathology over the long-term, there is some risk for internalizing and for social problems, particularly among children with brain tumors or those who received central nervous system (CNS) directed therapies (Vannatta and Gerhardt 2003; Eiser et al. 2000). As evidence of enhanced functioning or lower rates of psychopathology has been found among children with cancer (Phipps et al. 2001; Noll et al. 1999), there has also been a growing emphasis on positive outcomes, such as benefit finding, posttraumatic growth, and the emergence of other competencies in response to stress (Barakat et al. 2006; Phipps et al. 2007).

Because survivors appear to have limited psychopathology, research has focused on more subtle indicators of functioning, such as the attainment of socially valued outcomes or developmental milestones (e.g., graduation rates, employment, parenthood). Survivors of childhood cancer may not reach certain developmental milestones, or they may have delays in achieving life goals, such as completing their education and finding employment (Stam et al. 2006; Gurney et al. 2009; Lund et al. 2010). A recent meta-analysis (de Boer et al. 2006) reported that adult survivors of childhood cancer were disproportionately likely to be unemployed relative to controls. There is some evidence that survivors of childhood cancer may experience early social difficulties, as well as delays in marriage and parenthood relative to peers (Stam et al. 2006; Gurney et al. 2009; Lund et al. 2010). However, this risk is primarily accounted for by deficits among brain tumor survivors.

With respect to health behaviors, children with cancer demonstrate reduced physical activity and a lower capacity for exercise both during treatment and long-term (Tillmann et al. 2002; Warner et al. 1998). Adherence to sun protection has been noted as the least-frequent health behavior practiced by survivors (Tercyak et al. 2006). Furthermore, screening behaviors, such as breast or testicular self-examination, have been suboptimal among survivors (Yeazel et al. 2004). Experimenting in high-risk or health-compromising behaviors (e.g., sexual activity, substance use) is especially problematic for survivors of childhood cancer and may increase vulnerability to further health problems or secondary malignancies. Although children and adolescents with cancer may be protected from some high-risk behaviors, especially during treatment, participation in these activities at rates similar to peers is still of concern (Hollen and Hobbie 1996; Thompson et al. 2009; Klosky et al. 2012).

Research specific to children with bone tumors is sparse, with reports primarily focused on long-term functional or quality of life outcomes, as well as developmental milestones. Often, this work comes from the Childhood Cancer Survivor Study (CCSS) (Robison et al. 2009). Comparison samples vary across studies, including normative samples, sibling controls, other malignancies, or within-group comparisons based on amputation and limb salvage procedures. One review suggests that survivors of childhood bone tumors enjoy a favorable quality of life and have psychosocial outcomes similar to the general population (Mosher and McCarthy 1998). Another found no differences in outcomes between amputee and limb salvage survivors, but a third review concluded that there is some evidence that bone tumor survivors may fare less well relative to other cancer survivors (Langeveld et al. 2002).

Self-reported global functioning and psychological adjustment are generally good compared to normative samples or epidemiological controls (Felder-Puig et al. 1998; Teall et al. 2013; Nagarajan et al. 2009). However, Felder-Puig (1998) found that bone tumor survivors had lower rates of marriage, living independently, and parenthood. Survivors diagnosed in adolescence also had more social problems compared to those diagnosed in childhood or early adulthood. In other reports that compared bone tumor survivors to siblings, survivors were more likely to have some functional difficulty and employment disability (Nicholson et al. 1992). Those with amputations had greater deficits in education, employment, and health insurance coverage (Nagarajan et al. 2003). Although Zeltzer et al. (2008) found more psychosocial difficulties and poorer quality of life among bone tumor survivors than siblings, mean scores fell in the normal range.

Relative to survivors of other malignancies, survivors of bone tumors may have a higher likelihood of reporting adverse health status in at least one domain (e.g., activity limitations) (Nagarajan et al. 2011; Hudson et al. 2003). Interest has also focused on the relative superiority of limb salvage vs. amputation. This research tends to support some physical complications from limb salvage but generally similar quality of life and psychosocial functioning across groups (Postma et al. 1992; Nagarajan et al. 2002, 2004). However, quality of life may be lower for survivors who are female, have less education, and are older (Nagarajan et al. 2004). A final study found similar psychological functioning between groups, but survivors with amputations were more likely to have negative effects on marriage and work (Christ et al. 1996).

While many individual and family factors can contribute to the development of psychopathology more generally, often proximal factors (e.g., parental depression, family conflict) are the most common contributors to a child’s risk in the context of cancer (Robinson et al. 2007; Drotar 1997). This finding mirrors the developmental literature indicating the two primary factors that buffer the impact of stress on children are often intelligence and having a warm and consistent caregiver (Masten 2001). For example, both parental depression and anxiety have been positively associated with distress in children (Brennan et al. 2002; Langrock et al. 2002; Whaley et al. 1999).

Transmission of distress between family members may be accounted for or modified by family environment. In general, children in families high in conflict are more prone to difficulties (Hammen et al. 2004; Holmes et al. 1999), while children in a positive family environment are more likely to adjust better to stress (Drotar 1997; Ivanova and Israel 2006; Varni et al. 1996). Varni and colleagues (1996) found that in families of children with newly diagnosed with cancer, cohesion and expressiveness were associated with fewer child internalizing problems. Thus, a positive family environment may be protective for children with cancer.

Interestingly, parents of children treated for cancer may be at greater risk for adverse psychological outcomes than children themselves. A meta-analysis found that parents of children receiving treatment for cancer, particularly mothers, have greater distress than comparison samples (Pai et al. 2007). While many parents do not report clinical levels of distress, a subgroup of parents may be at risk for difficulties, particularly internalizing symptoms. Single mothers, and those with fewer socioeconomic resources, may be at the highest risk for internalizing symptoms and benefit the most from clinical assistance (Dolgin et al. 2007). Distress tends to be higher near diagnosis and during treatment but usually declines over the first couple of years. However, elevations in posttraumatic stress symptoms have been reported for parents both during and after their child’s cancer treatment (Kazak et al. 2005).

Little research has focused on the experience of parents near the end of their child’s life. Caring for a seriously ill child can have deleterious effects on parental quality of life, mood, sleep, and fatigue, with fear of the child’s death and physical symptoms as frequent concerns (Klassen et al. 2008; Carter 2003; Gedaly-Duff et al. 2006; Theunissen et al. 2007). About half of parents of children with advanced cancer have been found to have high rates of distress (Rosenberg et al. 2013). These outcomes may be worse for parents of children with poorer health status, more intense treatment, less time since diagnosis, and more economic hardship (Klassen et al. 2008; Rosenberg et al. 2013). In fact, parents whose children have a “difficult death” or unrelieved pain, anxiety, and sleep disruption may have a higher risk for internalizing symptoms and poorer quality of life 4–9 years after the death (Kreicbergs et al. 2005; Jalmsell et al. 2010). Not surprisingly, bereaved parents are at risk for depression, anxiety, guilt, posttraumatic stress symptoms, and anger (Rosenberg et al. 2012). They routinely score worse on most scales of adjustment, especially internalizing problems, relative to norms and controls (Rosenberg et al. 2012). Although parental grief is more prolonged than in other types of loss, over time, some parents can also recognize personal growth and positive outcomes in response to a child’s illness and death (Hogan and Schmidt 2002; Helgeson et al. 2006).

Siblings are often overlooked in research, and most work is qualitative in nature (Sharpe and Rossiter 2002). In many cases, there are several years of treatment during which much of the family’s attention and resources are directed toward the ill child (Wilkins and Woodgate 2005). Older siblings are often caregivers for their brother or sister with cancer and can assume other adult roles in the home (Martinson and Campos 1991; Gaab et al. 2014). This stress, coupled with parents who are less available or impaired by the child’s illness and/or death, poses significant risk for siblings.

A meta-analysis found siblings of children with chronic illness are at risk for multiple difficulties (Sharpe and Rossiter 2002), but a recent review suggests only a subset of siblings of children with cancer experience symptoms of posttraumatic stress, emotional distress, or reduced quality of life (Alderfer et al. 2010). When a child dies, bereaved siblings have been noted to have lower social competence and more internalizing and externalizing problems relative to norms or controls within 2 years of the death (Birenbaum et al. 1989; Hutton and Bradley 1994; McCown and Davies 1995). Grief symptoms can resurface years later as children mature and reflect on the loss from a different perspective. However, there is also evidence of positive growth, such as having a better outlook on life, being kinder, and more tolerant of others (Hogan and Greenfield 1991; Hogan and DeSantis 1996).

Providing supportive care that is sensitive to the context of child development and the family system is important throughout cancer treatment, survivorship, and/or end of life. Currently, comprehensive standards for psychosocial care in pediatric psycho-oncology are under development. Interdisciplinary psychosocial services that include access to chaplains, child life, creative arts therapies (e.g., art and music therapy), school intervention, social work, psychologists, and psychiatrists are ideal. However, resources are limited, and the availability of clinical services varies both between and within centers. Recommendations for interdisciplinary care of children with cancer include assistance with the practical and financial burdens of treatment and rehabilitation; communication about diagnosis and treatment decisions between the family and healthcare providers; routine screening for risk and protective factors that may contribute to overall adjustment; cognitive behavioral strategies for coping with symptoms, procedural distress, adherence, and emotional and behavioral adjustment; and school reintegration services to sustain normal academic and social functioning (Noll and Kazak 2004). Support for adjustment among parents and siblings is also important, but often challenging with respect to visitor restrictions or access in the case of siblings.