Skip to main content
NCBI home page
Journal of Pediatric Psychology logoLink to Journal of Pediatric Psychology
. 2017 Aug 31;43(3):237–242. doi: 10.1093/jpepsy/jsx114

Topical Review: Pain in Survivors of Pediatric Cancer: Applying a Prevention Framework

Amanda L Stone 1,2,✉, Cynthia W Karlson 1, Lauren C Heathcote 3, Abby R Rosenberg 4,5, Tonya M Palermo 6
PMCID: PMC6454449  PMID: 29048571

Abstract

Objective

To apply a biopsychosocial framework to understand factors influencing pain in survivors of pediatric cancer to inform pain prevention efforts and highlight the need for interdisciplinary care.

Method

This topical review draws from both pediatric cancer survivorship research and chronic noncancer pain research to illustrate how components of a preventative model can be applied to pain in survivorship.

Results

Pain is a common experience among long-term survivors of pediatric cancer. The pain experience in survivorship can be conceptualized in terms of biological disease and treatment factors, cognitive and affective factors, and social and contextual factors. We review literature pertinent to each of these biopsychosocial factors and tailor an existing public health prevention framework for pain in survivors of pediatric cancer.

Conclusions

Classifying survivors of pediatric cancer into pain risk categories based on their daily experiences of pain, pain-related functional impairment, and distress could help guide the implementation of pain-related prevention and intervention strategies in this population. Future research is needed to establish the efficacy of screening measures to identify patients in need of psychosocial pain and pain-related fear management services, and interdisciplinary pediatric chronic pain management programs in survivors of pediatric cancer.

Keywords: cancer and oncology, chronic and recurrent pain, health promotion and prevention, pain

With continuing improvements in pediatric cancer treatment, increasing numbers of children are surviving cancer each year. In fact, 80% of children diagnosed with cancer survive >5–10 years (Phillips et al., 2015). Pain is a common experience among long-term survivors of pediatric cancer. Studies demonstrate up to 58.7% of survivors report pain and are four times more likely to experience pain compared with their siblings (Huang et al., 2013; Lu et al., 2011). Pain experienced by survivors can be acute (i.e., short-lasting pain occurring from a medical procedure or acute illness), episodic (i.e., recurrent experiences of pain separated by pain-free periods), or chronic (i.e., pain daily or almost every day for at least 3 months). One third to one half of survivors of pediatric cancer report experiencing at least mild pain-related impairment, and pain has been associated with elevated suicidal ideation and distress (Alessi et al., 2007; Recklitis et al., 2010). In addition to the emotional distress that can occur in response to a painful experience, pain symptoms may be additionally distressing for survivors because they raise concerns of recurrent disease or serious treatment-related medical complications (Zebrack et al., 2002). Although some survivors will experience pain, given the nature of their disease and treatment course, prevention efforts targeting pain-related distress and impairment in this population could improve overall quality of life.

This topical review aims to apply a biopsychosocial framework to first understand factors influencing pain in survivors of pediatric cancer. We then apply a public health prevention model (Kazak, 2006) to demonstrate how assessment and interdisciplinary intervention could be aimed at preventing and reducing pain, functional impairment, and pain-related distress within the broader context of interdisciplinary survivorship care to address physical late effects, psychological, social, and educational needs. Owing to Because of the limited empirical attention directed at episodic and chronic pain in survivors of pediatric cancer, we also draw on chronic noncancer pain research to illustrate components of a pain prevention model.

Disease and Treatment-Related Factors

Survivors of pediatric cancer may be vulnerable to experiencing acute, episodic, or chronic pain because of a number of disease and treatment-related factors. Several biological factors including specific types of cancer (e.g., osteosarcoma), cancer treatments (e.g., vincristine and platinum-based chemotherapy agents, central nervous system irradiation, and surgery), younger age at diagnosis, and female sex have been associated with increased rates of pain and poorer health-related quality of life in survivors of pediatric cancer (Huang et al., 2013). Chemotherapy-induced peripheral neuropathy and other pain-related symptoms resulting from cancer or cancer treatments may be progressive, increase in severity over time, or develop as a late effect (Argyriou, Bruna, Marmiroli, & Cavaletti, 2012). Regardless of the underlying origin of pain, the experience of pain represents a complex interaction of neural pathways often implicated in both the physical and emotional experiences of pain (Simons, Elman, & Borsook, 2014).

Cognitive–Affective Factors

Cognitive Factors

In chronic noncancer pain, biopsychosocial models have proposed a strong influence of cognitive and affective factors on pain experience and pain-related impairment (Asmundson, Noel, Petter, & Parkerson, 2012). At the center of many biopsychosocial models of pain is the cognitive appraisal of pain as threatening. Heightened pain threat appraisal is proposed to drive fear and worry, hypervigilance (selective attending to pain), and negative interpretations of other ambiguous bodily information (e.g., stomach or muscle aches). These biased cognitive processes in turn fuel avoidance of activities and heighten pain perception. Expectations that one will experience pain and hypervigilance to the occurrence of pain can increase an individual’s perceived pain severity (Wiech, 2016). Despite well-described psychological models, to date pain-related cognition and affect have received little consideration within the context of pediatric cancer survival. The lack of research on pain-related cognition is surprising, given pain may be a sign of recurrence for many young survivors of cancer, and is therefore likely to be perceived as highly threatening and necessitating medical evaluation (Heathcote & Eccleston, 2017).

One cognitive factor that may significantly contribute to pain and impairment within the context of cancer survival is executive functioning. For example, the ability to effortfully control and flexibly shift attention is vital, given that attentional focus on pain may be heightened when pain is perceived as indicative of disease recurrence or highly threatening (Heathcote et al., 2017). On individual performance-based measures of cognitive functioning, survivors of pediatric cancer show impairments across multiple domains including attention, memory, and cognitive control (Annett, Patel, & Phipps, 2015). Given low attention control influences child pain experiences (Heathcote et al., 2017), studies are needed to investigate how neurocognitive impairments moderate pain experiences in survivors of pediatric cancer as well as whether interventions targeting both cancer-related cognitive impairment and cognitive appraisal of pain are warranted.

Affective Factors

Survivors of pediatric cancer experience varying levels of psychological distress over time (Zebrack et al., 2002; Zeltzer et al., 2009), yet how distress relates to the experience of pain has not been well-described. Although evidence suggests that rates of psychological distress in survivors of pediatric cancer are similar to the general population (Zeltzer et al., 2009), a subset may experience persisting, clinically significant distress, including higher levels of posttraumatic stress symptoms and depressive symptoms. Evidence from the Childhood Cancer Survivor Study suggests increased internalizing and externalizing symptoms in survivors of pediatric cancer who experience cancer-related pain compared with those who experience no pain (Brinkman et al., 2016). Thus, similar to chronic noncancer pain populations (Vinall, Pavlova, Asmundson, Rasic, & Noel, 2016), survivors who experience increased pain symptoms also are at increased risk for psychiatric comorbidities.

In sum, both cognitive and affective processes influence the perception and experience of pain. Increased difficulty in modulating attention and increased negative affect may interact with hypervigilance toward physical sensations following cancer treatment and increase the risk for pain in survivors of pediatric cancer. The relation between cognitive–affective factors and pain is likely bidirectional, as experiencing pain can also lead to heightened distress and difficulty modulating attention. Needed are studies investigating associations between acute, episodic, and chronic pain experiences and cognitive–affective factors in this population. Intervening on relevant cognitive–affective factors may in turn prevent or reduce pain-related interference and distress in survivors of pediatric cancer who experience persistent pain.

Social and Contextual Factors

A number of social and contextual factors including parent and family factors and sociodemographic risk factors may influence risk for pain and maintenance of functional impairment in survivors of pediatric cancer. Parent and family functioning affect children’s outcomes in both pediatric cancer and pediatric chronic noncancer pain populations (Kazak, Schneider, Didonato, & Pai, 2015; Palermo, Valrie, & Karlson, 2014). Multiple aspects of family functioning including greater cohesion, expressiveness, and support, as well as less family conflict, are associated with better child adjustment after receiving a cancer diagnosis (Van Schoors et al., 2017). In children with chronic noncancer pain, poorer family functioning and greater parental distress relate to increased pain-related impairment in youth (Chow, Otis, & Simons, 2016; Palermo et al., 2014). However, little is known about these factors in the context of daily or recurrent pain in survivors of pediatric cancer.

Contextual factors such as lower educational attainment, minority status, and lower socioeconomic status have been associated with increased risk for pain and psychological distress in survivors of pediatric cancer (Lu et al., 2011; Zeltzer et al., 2009). These associations could partly be because of systems factors (e.g., difficulty accessing health-care resources and pain treatment centers) as well as patient expectations and biases (e.g., perceived injustice), and provider characteristics (e.g., patient–provider communication; provider implicit biases and attitudes) (Green et al., 2003). Additional research is needed to understand the contribution of contextual factors to pain experiences to better inform prevention efforts for high-risk families.

Pain Prevention in Pediatric Cancer: Implications for Assessment and Intervention

Taking into account these biopsychosocial factors related to pain, a comprehensive preventative health model could help minimize and reduce the potential impact of pain in survivors of pediatric cancer. Existing public health prevention models have already been adapted to inform general psychosocial screening and intervention in pediatric cancer (Kazak, 2006). Existing models use a prevention framework to identify three different levels of care for youth and their families: (1) Universal (i.e., broadest level of care within an identified population), (2) Targeted/Selected (i.e., a number of risk factors are present and should be closely monitored), and (3) Clinical/Treatment (i.e., clinically meaningful symptoms are present). These levels correspond with distribution of resources in a cost-effective manner to maximize intervention delivery to those at highest risk while minimizing potential costs associated with providing resources and care to the entire population.

We propose tailoring such preventative health models to include relevant risk factors for the development of persistent pain, pain-related functional impairment, and pain-related distress (Figure 1). Given the risk for persistent pain, routine screening is recommended of all survivors of pediatric cancer at the Universal level performed by all members of the interdisciplinary care team. Brief tools capturing pain-related risk such as the Pediatric Pain Screening Tool (PPST) (Simons et al., 2015) or the PROMIS Pain Intensity and Pain Interference scales (Stone, Broderick, Junghaenel, Schneider, & Schwartz, 2016) could identify survivors with frequent and severe pain, impairment, and pain-related distress. Medical providers could then match survivors to either Targeted- or Clinical-level interventions. For pediatric cancer patients undergoing surgical interventions, using measures of both parent and child cognitive–affective factors presurgery (e.g., parent catastrophizing about the child’s pain and child anxiety) may help identify children who are at greatest risk for developing persistent postsurgical pain that extends into survivorship. Postsurgical patients would also benefit from close monitoring with pain scales such as the PROMIS, as well as early pain intervention (Rabbitts, Fisher, Rosenbloom, & Palermo, 2017). Additional tools already being used in pediatric cancer such as the Psychological Assessment Tool (Pai et al., 2008) are further recommended to provide information on social and family contextual risk factors potentially related to pain. Increased emphasis on providing optimal, interdisciplinary pain management (e.g., acute pain medication, child life intervention, relaxation strategies, and physical medicine and rehabilitation) for acute procedures and postoperative pain in pediatric cancer patients during and after treatment may help reduce the development of persistent pain during survivorship.

Figure 1.

Figure 1.

Open in a new tab

A preventive health framework applied to pain in survivors of pediatric cancer.

With increasing intensity and frequency of pain, survivors move from the Universal level to the Targeted level of intervention, meaning increased support and monitoring, and intervention is needed to prevent the escalation of pain, impairment, and affective distress. At the Targeted level are those survivors with ongoing pain but who do not yet have impairments in their overall quality of life and/or daily functioning. Interventions such as in-person or Internet-delivered pain education programs (Robins, Perron, Heathcote, & Simons, 2016; Stinson et al., 2013) could reinforce adaptive pain coping strategies to reduce the potential escalation of symptoms and prevent functional impairment. Additionally, the development of adjunctive interventions such as cognitive bias modification (Lichtenthal et al., 2017) in pediatric cancer populations could address attentional and cognitive biases to prevent the escalation of cognitive appraisals of pain as threatening.

Survivors who screen high on pain-related functional impairment and affective distress are classified in the Clinical level, meaning treatment is indicated to improve functioning and reduce the burden of chronic pain. Survivors and their families who screen high on measures of affective distress may particularly benefit from psychosocial interventions. Optimally, an interdisciplinary treatment team would include some combination of the following providers: oncologists, nurses, psychologists, social workers, child life specialists, occupational therapists/physical therapists, and physical medicine and rehabilitation physicians. An ideal team approach would involve collaboratively working toward minimizing the experience of chronic pain and improving psychological factors associated with chronic pain in survivors of pediatric cancer. Although interdisciplinary pain treatment programs are effective in reducing pain-related impairment in individuals with chronic noncancer pain (Stahlschmidt, Zernikow, & Wager, 2016), research is needed to determine the components of interdisciplinary programs that are most effective for survivors of pediatric cancer.

Conclusions

Historically, pain in children with cancer has been a focus of acute management during painful medical procedures and acute complications from treatment (e.g., neuropathic pain). Burgeoning research within the past 10 years suggests pain is an ongoing concern for many survivors of pediatric cancer, and preventative models of pain care are needed. This review applies the biopsychosocial model to conceptualize episodic and chronic pain, and pain-related distress in survivorship in terms of biological disease and treatment factors, cognitive and affective factors, and social and contextual factors to tailor a preventative model for pain in survivorship. Applying existing preventative health models to pain in survivors of pediatric cancer may prove useful for identifying individuals in need of intervention, matching the level of intervention needed and ultimately minimizing and reducing the potential impact of pain.

Funding

T. M. Palermo is supported by NIH K24HD060068.

Conflicts of interest: None declared.

References

  1. Alessi D., Dama E., Barr R., Mosso M. L., Maule M., Magnani C., Pastore G., Merletti F. (2007). Health-related quality of life of long-term childhood cancer survivors: A population-based study from the Childhood Cancer Registry of Piedmont, Italy. European Journal of Cancer, 43, 2545–2552. doi:10.1016/j.ejca.2007.07.026 [DOI] [PubMed] [Google Scholar]
  2. Annett R. D., Patel S. K., Phipps S. (2015). Monitoring and assessment of neuropsychological outcomes as a standard of care in pediatric oncology. Pediatric Blood and Cancer, 62, S460–S513. doi:10.1002/pbc.25749 [DOI] [PubMed] [Google Scholar]
  3. Argyriou A. A., Bruna J., Marmiroli P., Cavaletti G. (2012). Chemotherapy-induced peripheral neurotoxicity (CIPN): An update. Critical Reviews in Oncology/Hematology, 82, 51–77. doi:10.1016/j.critrevonc.2011.04.012 [DOI] [PubMed] [Google Scholar]
  4. Asmundson G. J. G., Noel M., Petter M., Parkerson H. A. (2012). Pediatric fear-avoidance model of chronic pain: Foundation, application and future directions. Pain Research and Management, 17, 397–405. [DOI] [PMC free article] [PubMed] [Google Scholar]
  5. Brinkman T. M., Li C., Vannatta K., Marchak J. G., Lai J.-S., Prasad P. K., Kimberg C., Vuotto S., Di C., Srivastava D., Robison L. L., Armstrong G. T., Krull K. R. (2016). Behavioral, social, and emotional symptom comorbidities and profiles in adolescent survivors of childhood cancer: A report from the Childhood Cancer Survivor Study. Journal of Clinical Oncology, 34, 3417–3425. doi:10.1200/jco.2016.66.4789 [DOI] [PMC free article] [PubMed] [Google Scholar]
  6. Chow E. T., Otis J. D., Simons L. E. (2016). The longitudinal impact of parent distress and behavior on functional outcomes among youth with chronic pain. The Journal of Pain, 17, 729–738. doi:10.1016/j.jpain.2016.02.014 [DOI] [PMC free article] [PubMed] [Google Scholar]
  7. Green C. R., Anderson K. O., Baker T. A., Campbell L. C., Decker S., Fillingim R. B., Kalauokalani D. A., Lasch K. E., Myers C., Tait R. C. (2003). The unequal burden of pain: Confronting racial and ethnic disparities in pain. Pain Medicine, 4, 277–294. [DOI] [PubMed] [Google Scholar]
  8. Heathcote L. C., Eccleston C. (2017). Pain and cancer survival: A cognitive-affective model of symptom appraisal and the uncertain threat of disease recurrence. Pain, 158, 1187–1191. [DOI] [PubMed] [Google Scholar]
  9. Heathcote L. C., Lau J. Y. F., Mueller S. C., Eccleston C., Fox E., Bosmans M., Vervoort T. (2017). Child attention to pain and pain tolerance are dependent upon anxiety and attention control: An eye‐tracking study. European Journal of Pain, 21, 250–263. [DOI] [PubMed] [Google Scholar]
  10. Huang I.-C., Brinkman T. M., Kenzik K., Gurney J. G., Ness K. K., Lanctot J., Shenkman E., Robison L. L., Hudson M. M., Krull K. R. (2013). Association between the prevalence of symptoms and health-related quality of life in adult survivors of childhood cancer: A report from the St Jude Lifetime Cohort Study. Journal of Clinical Oncology, 31, 4242–4251. doi:10.1200/JCO.2012.47.8867 [DOI] [PMC free article] [PubMed] [Google Scholar]
  11. Kazak A. E. (2006). Pediatric Psychosocial Preventative Health Model (PPPHM): Research, practice, and collaboration in pediatric family systems medicine. Families, Systems and Health, 24, 381. [Google Scholar]
  12. Kazak A. E., Schneider S., Didonato S., Pai A. L. (2015). Family psychosocial risk screening guided by the Pediatric Psychosocial Preventative Health Model (PPPHM) using the Psychosocial Assessment Tool (PAT). Acta Oncologica, 54, 574–580. doi:10.3109/0284186x.2014.995774 [DOI] [PubMed] [Google Scholar]
  13. Lichtenthal W. G., Corner G. W., Slivjak E. T., Roberts K. E., Li Y., Breitbart W., Lacey S., Tuman M., DuHamel K. N., Blinder V. S., Beard C., Blinder V. S. (2017). A pilot randomized controlled trial of cognitive bias modification to reduce fear of breast cancer recurrence. Cancer, 123, 1424–1433. [DOI] [PMC free article] [PubMed] [Google Scholar]
  14. Lu Q., Krull K. R., Leisenring W., Owen J. E., Kawashima T., Tsao J. C., Zebrack B., Mertens A., Armstrong G. T., Stovall M., Robison L. L., Zeltzer L. K. (2011). Pain in long-term adult survivors of childhood cancers and their siblings: A report from the Childhood Cancer Survivor Study. Pain, 152, 2616–2624. doi:10.1016/j.pain.2011.08.006 [DOI] [PMC free article] [PubMed] [Google Scholar]
  15. Pai A. L., Patiño-Fernández A. M., McSherry M., Beele D., Alderfer M. A., Reilly A. T., Hwang W. T., Kazak A. E. (2008). The Psychosocial Assessment Tool (PAT2.0): Psychometric properties of a screener for psychosocial distress in families of children newly diagnosed with cancer. Journal of Pediatric Psychology, 33, 50–62. doi:10.1093/jpepsy/jsm053 [DOI] [PMC free article] [PubMed] [Google Scholar]
  16. Palermo T. M., Valrie C. R., Karlson C. W. (2014). Family and parent influences on pediatric chronic pain: A developmental perspective. The American Psychologist, 69, 142–152. doi:10.1037/a0035216 [DOI] [PMC free article] [PubMed] [Google Scholar]
  17. Phillips S. M., Padgett L. S., Leisenring W. M., Stratton K. K., Bishop K., Krull K. R., Alfano C. M., Gibson T. M., de Moor J. S., Hartigan D. B., Armstrong G. T., Robison L. L., Rowland J. H., Oeffinger K. C., Mariotto A. B. (2015). Survivors of childhood cancer in the United States: Prevalence and burden of morbidity. Cancer Epidemiology, Biomarkers and Prevention, 24, 653–663. doi:10.1158/1055-9965.epi-14-1418 [DOI] [PMC free article] [PubMed] [Google Scholar]
  18. Rabbitts J. A., Fisher E., Rosenbloom B. N., Palermo T. M. (2017). Prevalence and predictors of chronic postsurgical pain in children: A systematic review and meta-analysis. The Journal of Pain, 18, 605–614. doi:10.1016/j.jpain.2017.03.007 [DOI] [PMC free article] [PubMed] [Google Scholar]
  19. Recklitis C. J., Diller L. R., Li X., Najita J., Robison L. L., Zeltzer L. (2010). Suicide ideation in adult survivors of childhood cancer: A report from the Childhood Cancer Survivor Study. Journal of Clinical Oncology, 28, 655–661. doi:10.1200/jco.2009.22.8635 [DOI] [PMC free article] [PubMed] [Google Scholar]
  20. Robins H., Perron V., Heathcote L., Simons L. (2016). Pain neuroscience education: State of the art and application in pediatrics. Children, 3, 43. [DOI] [PMC free article] [PubMed] [Google Scholar]
  21. Simons L. E., Elman I., Borsook D. (2014). Psychological processing in chronic pain: A neural systems approach. Neuroscience and Biobehavioral Reviews, 39, 61–78. doi:10.1016/j.neubiorev.2013.12.006 [DOI] [PMC free article] [PubMed] [Google Scholar]
  22. Simons L. E., Smith A., Ibagon C., Coakley R., Logan D. E., Schechter N., Borsook D., Hill J. C. (2015). Pediatric Pain Screening Tool (PPST): Rapid identification of risk in youth with pain complaints. Pain, 156, 1511–1518. doi:10.1097/j.pain.0000000000000199 [DOI] [PMC free article] [PubMed] [Google Scholar]
  23. Stahlschmidt L., Zernikow B., Wager J. (2016). Specialized rehabilitation programs for children and adolescents with severe disabling chronic pain: Indications, treatment and outcomes. Children, 3, 33. [DOI] [PMC free article] [PubMed] [Google Scholar]
  24. Stinson J. N., Jibb L. A., Nguyen C., Nathan P. C., Maloney A. M., Dupuis L. L., Gerstle J. T., Alman B., Hopyan S., Strahlendorf C., Portwine C., Johnston D. L., Orr M. (2013). Development and testing of a multidimensional iPhone pain assessment application for adolescents with cancer. Journal of Medical Internet Research, 15, e51. [DOI] [PMC free article] [PubMed] [Google Scholar]
  25. Stone A. A., Broderick J. E., Junghaenel D. U., Schneider S., Schwartz J. E. (2016). PROMIS fatigue, pain intensity, pain interference, pain behavior, physical function, depression, anxiety, and anger scales demonstrate ecological validity. Journal of Clinical Epidemiology, 74, 194–206. doi:10.1016/j.jclinepi.2015.08.029 [DOI] [PubMed] [Google Scholar]
  26. Van Schoors M., Caes L., Knoble N. B., Goubert L., Verhofstadt L. L., Alderfer M. A. (2017). Systematic review: Associations between family functioning and child adjustment after pediatric cancer diagnosis: A meta-analysis. Journal of Pediatric Psychology, 42, 6–18. [DOI] [PubMed] [Google Scholar]
  27. Vinall J., Pavlova M., Asmundson G. J., Rasic N., Noel M. (2016). Mental health comorbidities in pediatric chronic pain: A narrative review of epidemiology, models, neurobiological mechanisms and treatment. Children, 3, 40. [DOI] [PMC free article] [PubMed] [Google Scholar]
  28. Wiech K. (2016). Deconstructing the sensation of pain: The influence of cognitive processes on pain perception. Science, 354, 584–587. [DOI] [PubMed] [Google Scholar]
  29. Zebrack B. J., Zeltzer L. K., Whitton J., Mertens A. C., Odom L., Berkow R., Robison L. L. (2002). Psychological outcomes in long-term survivors of childhood leukemia, Hodgkin’s disease, and non-Hodgkin’s lymphoma: A report from the Childhood Cancer Survivor Study. Pediatrics, 110, 42–52. [DOI] [PubMed] [Google Scholar]
  30. Zeltzer L. K., Recklitis C., Buchbinder D., Zebrack B., Casillas J., Tsao J. C., Lu Q., Krull K. (2009). Psychological status in childhood cancer survivors: A report from the Childhood Cancer Survivor Study. Journal of Clinical Oncology, 27, 2396–2404. [DOI] [PMC free article] [PubMed] [Google Scholar]

Articles from Journal of Pediatric Psychology are provided here courtesy of Oxford University Press

RESOURCES