Psychosocial functioning in pediatric cancer

OBJECTIVE: To describe the emergence of pediatric psycho-oncology and to summarize research on psychosocial aspects of childhood cancer and survivorship. METHODS: To review research into illness communication and informed consent, procedural pain, late effects, psychological distress, coping and adjustment, and special risk populations. Methodological challenges, appropriate methodology, and directions for future research are discussed. RESULTS: The past 30 years have seen change from avoidance of communication about cancer to an emphasis on straightforward discussion of diagnosis and prognosis. Behavioral research has led to interventions to reduce procedural distress. Late effects have been observed in social functioning. Although average levels of distress in survivors of pediatric cancer are typical, subsets of more vulnerable patients and family members exist. Factors predicting positive and negative coping have been identified. CONCLUSIONS: As the numbers of pediatric cancer survivors increase, psychosocial researchers will be better able to conduct longitudinal studies not only of adjustment and its predictors but also of the impact of the emerging medical treatments and interventions to ameliorate late effects of treatment. Additional funding, improving methodology, and multi-institutional cooperation will aid future pediatric psycho-oncology investigators.     

A multicenter, randomized clinical trial of a cognitive remediation program for childhood survivors of a pediatric malignancy

Survivors of childhood cancer whose malignancy and/or treatment involved the central nervous system may demonstrate a consistent pattern of neurocognitive deficits. The present study evaluated a randomized clinical trial of the Cognitive Remediation Program (CRP). Participants were 6- to 17-year-old survivors of childhood cancer (N = 161; 35% female, 18% Hispanic, 10% African American, 64% Caucasian, 8% other) who were at least 1 year off treatment and who manifested an attentional deficit. They were enrolled at 7 sites nationwide. Two thirds of the participants were randomly assigned to cognitive remediation. All participants were assessed using a battery of academic achievement/neurocognitive tests and parent/teacher measures of attention. The CRP resulted in parent report of improved attention and statistically significant increases in academic achievement. Effect sizes were modest but were comparable with those for other clinical trials of brain injury rehabilitation and for psychological interventions in general. The CRP is presented as a potentially beneficial treatment for many survivors of pediatric cancer. Long-term clinical significance remains unproven. Further work is needed to improve effect sizes and treatment compliance and to address the needs of other populations with pediatric brain injury.     

A review of individual psychotherapy outcomes for adult survivors of childhood sexual abuse

This review examines the current literature on individual psychotherapy outcomes with adult survivors of childhood sexual abuse. As compared to the literature on treatment with victims of sexual trauma in adulthood, fewer researchers have explored the field of psychotherapy outcome with this population, particularly with regard to individual treatments. For this reason, a review of psychotherapy outcome for adult survivors of child sexual abuse is needed. This review first presents issues salient to the study of treatment with this population, such as prevalence, short-term impact, and long-term sequelae of childhood sexual trauma. Next, the eight studies that have been published on individual psychotherapy for this population are presented and evaluated according to both efficacy and effectiveness criteria for a methodologically sound study. Last, directions for future research with this population include continued integration of efficacy and effectiveness methodology, use of multi-method/multi-rater assessment data, as well as further investigation of interpersonal variables such as the therapeutic alliance.     

Neurocognitive Dysfunction in Hematopoietic Cell Transplant Recipients: Expert Review from the Late Effects and Quality of Life Working Committee of the Center for International Blood and Marrow Transplant Research and Complications and Quality of Life Working Party of the European Society for Blood and Marrow Transplantation

Hematopoietic cell transplantation (HCT) is a potentially curative treatment for children and adults with malignant and nonmalignant diseases. Despite increasing survival rates, long-term morbidity after HCT is substantial. Neurocognitive dysfunction is a serious cause of morbidity, yet little is known about neurocognitive dysfunction after HCT. To address this gap, collaborative efforts of the Center for International Blood and Marrow Transplant Research and the European Society for Blood and Marrow Transplantation undertook an expert review of neurocognitive dysfunction after HCT. In this review we define what constitutes neurocognitive dysfunction, characterize its risk factors and sequelae, describe tools and methods to assess neurocognitive function in HCT recipients, and discuss possible interventions for HCT patients with this condition. This review aims to help clinicians understand the scope of this health-related problem, highlight its impact on well-being of survivors, and help determine factors that may improve identification of patients at risk for declines in cognitive functioning after HCT. In particular, we review strategies for preventing and treating neurocognitive dysfunction in HCT patients. Finally, we highlight the need for well-designed studies to develop and test interventions aimed at preventing and improving neurocognitive dysfunction and its sequelae after HCT.     

Evidence-based interventions for survivors of childhood cancer and their families

OBJECTIVE: To summarize existing empirical reports of psychological interventions for children with cancer and their families and to outline directions for future work. METHODS: Research accomplishments related to four areas were summarized: understanding and reducing procedural pain; realizing long-term consequences; appreciating distress at diagnosis and over time; and knowing the importance of social relationships. Recommendations for future research in this field were discussed in three domains: balancing competence and psychopathology; integrating research-based interventions into clinical care; future directions). RESULTS AND CONCLUSIONS: Interventions related to procedural pain and distress have strong empirical support. Although most survivors of childhood cancer and their families adjust quite well as they complete treatment, evidence-based interventions during and after treatment that address cancer-specific sequelae represent important growing areas of work. In the future, the development of interventions to address emerging survivorship issues will be vital, and theoretically grounded, evidence-based pragmatic interventions will be necessary.     

Risk of posttraumatic stress symptoms: a comparison of child survivors of pediatric cancer and parental bereavement

OBJECTIVE: To compare the risk of posttraumatic stress (PTS) symptoms and the mediating effect of perceived future threat on the risk of PTS symptoms among survivors of pediatric cancer and children who had a parent die. METHODS: Seventy-eight children (39 survivors of cancer, 39 bereaved) completed self-report measures of PTS symptoms, depression, anxiety, and perceived risk of future threat for the event they experienced. RESULTS: The children who lost a parent reported significantly more PTS symptoms than the survivors of cancer. The effect of group status (survivor of cancer vs. bereaved) on PTS symptomatology was partly mediated by the children's perceived risk of future threat. CONCLUSIONS: The rate of PTS symptoms was found to be higher among children who had lost a parent than among survivors of pediatric cancer. This difference may partly be explained by their perceived risk of a future threat. Clinical implications are discussed.     

Physical activity in child and adolescent cancer survivors: a review

Abstract

Childhood cancer survivors are at increased risk for future health problems. As such, physical activity (PA) has been targeted as a health promotion priority in child and adolescent cancer survivors. Research indicates that a large portion of pediatric survivors do not meet PA recommendations. Using Bronfenbrenner's ecological theory as a framework, this review presents a conceptual model to explain child and adolescent survivors' PA. The model considers predictors of PA across six domains: (1) demographic, (2) medical, (3) cognitive/emotional, (4) behavioural, (5) social/cultural and (6) environmental. A structured literature review found 14 empirical articles examining those predictors of PA among child and adolescent cancer survivors. Much existing research is cross-sectional, but suggests multiple factors work together to encourage or discourage PA among survivors of child/adolescent cancer. The conceptual model, which is based in empirical findings to date, can be used to understand the process through which PA is promoted and maintained, to inform the development of empirically-supported clinical interventions, and to guide future research objectives and priorities.     

Long-term outcomes of adults with features of VACTERL association

VACTERL association involves the presence of specific congenital, multi-organ malformations that tend to co-occur. Clinical and research efforts typically center on pediatric patients, and there is a scarcity of information in the literature regarding VACTERL-related issues and outcomes in adulthood. We describe here 11 adults with features of VACTERL association ascertained through our research study on the condition. In our cohort of adult patients, approximately 25% of medically significant malformations that are component features of VACTERL association, including 40% of vertebral, 50% of cardiac, and 50% of renal anomalies, were not identified during childhood. Additionally, medical sequelae of many of the primary malformations identified in infancy or early childhood persist or are first reported in adulthood. These sequelae can involve challenging medical and surgical management in adulthood. As most adults with VACTERL association are not specifically followed for VACTERL-related issues, a more uniform diagnostic work-up and a low threshold for investigation of medical sequelae of the primary disorder may enhance the quality of clinical management in these patients.     

Late morbidity and mortality in adult survivors of childhood glioma with neurofibromatosis type 1: report from the Childhood Cancer Survivor Study

PurposeNeurofibromatosis type 1 (NF1) is associated with tumor predisposition and nonmalignant health conditions. Whether survivors of childhood cancer with NF1 are at increased risk for poor long-term health outcomes is unknown.MethodsOne hundred forty-seven 5+ year survivors of childhood glioma with NF1 from the Childhood Cancer Survivor Study were compared with 2629 non-NF1 glioma survivors and 5051 siblings for late mortality, chronic health conditions, and psychosocial, neurocognitive, and socioeconomic outcomes.ResultsSurvivors with NF1 (age at diagnosis: 6.8 ± 4.8 years) had greater cumulative incidence of late mortality 30 years after diagnosis (46.3% [95% confidence interval: 23.9–62.2%]) compared with non-NF1 survivors (18.0% [16.1–20.0%]) and siblings (0.9% [0.6–1.2%]), largely due to subsequent neoplasms. Compared with survivors without NF1, those with NF1 had more severe/life-threatening chronic conditions at cohort entry (46.3% [38.1–54.4%] vs. 30.8% [29.1–32.6%]), but similar rates of new conditions during follow-up (rate ratio: 1.26 [0.90–1.77]). Survivors with NF1 were more likely to report psychosocial impairments, neurocognitive deficits, and socioeconomic difficulties compared with survivors without NF1.ConclusionsLate mortality among glioma survivors with NF1 is twice that of other survivors, due largely to subsequent malignancies. Screening, prevention, and early intervention for chronic health conditions and psychosocial and neurocognitive deficits may reduce long-term morbidity in this vulnerable population.     

Parsing the Heterogeneity and Increasing Specificity: A Commentary on "Neurocognitive Functioning in Bipolar Disorder"

"Neurocognitive Functioning in Bipolar Disorder" is a comprehensive examination of the literatures examining neurocognitive and neuroimaging studies in pediatric and adult bipolar disorder. Based upon this review, the present commentary highlights four important areas of future investigation: increasing specificity of cognitive measures, evaluating the specificity of neurocognitive abnormalities to bipolar disorder, modeling heterogeneity of neurocognitive functioning within bipolar disorder, and searching for core neurocognitive processes more closely linked to the dysfunctional behavior seen in bipolar disorder. In addition to greater phenotypic clarity, attention to these four areas will be crucial for advancing the field. The present commentary provides recommendations for addressing these areas to facilitate progress in neurocognitive and neurobiological research of bipolar disorder.     

The Child in the Family (Book)

Reviews outcome literature on survivors of extracorporeal membrane oxygenation (ECMO), who have experienced severe cardiorespiratory disease with potential central nervous system sequelae. Long-term follow-up has been reported on approximately 350 survivors representing less than 5% of cases. Several consistent findings are noted from a review of the literature: (a) Most infant developmental scales are more accurate predictors short-term outcome when integrated with neurological exams or neuroimaging findings; (b) most ECMO survivors (70% to 80%) fall in the normal range on development and intelligence measures; (c) 20% to 30% of ECMO survivors have poor outcomes, with 50% to 100% of these usually having associated chronic lung disease and/or brain disease/injury; and (d) 20% to 80% of those with normal outcomes and 10% to 35% of those with normal outcomes qualify for special services. Patterns of neurological and assessment outcome are discussed in terms of location of brain injury and neuropsychological theory.     

History of psycho-oncology: overcoming attitudinal and conceptual barriers

The formal beginnings of psycho-oncology date to the mid-1970s, when the stigma making the word "cancer" unspeakable was diminished to the point that the diagnosis could be revealed and the feelings of patients about their illness could be explored for the first time. However, a second stigma has contributed to the late development of interest in the psychological dimensions of cancer: negative attitudes attached to mental illness and psychological problems, even in the context of medical illness. It is important to understand these historical underpinnings because they continue to color contemporary attitudes and beliefs about cancer and its psychiatric comorbidity and psychosocial problems. Over the last quarter of the past century, psycho-oncology became a subspecialty of oncology with its own body of knowledge contributing to cancer care. In the new millennium, a significant base of literature, training programs, and a broad research agenda have evolved with applications at all points on the cancer continuum: behavioral research in changing lifestyle and habits to reduce cancer risk; study of behaviors and attitudes to ensure early detection; study of psychological issues related to genetic risk and testing; symptom control (anxiety, depression, delirium, pain, and fatigue) during active treatment; management of psychological sequelae in cancer survivors; and management of the psychological aspects of palliative and end-of-life care. Links between psychological and physiological domains of relevance to cancer risk and survival are being actively explored through psychoneuroimmunology. Research in these areas will occupy the research agenda for the first quarter of the new century. At the start of the third millennium, psycho-oncology has come of age as one of the youngest subspecialties of oncology, as one of the most clearly defined subspecialties of consultation-liaison psychiatry, and as an example of the value of a broad multidisciplinary application of the behavioral and social sciences.     

Lessons in pediatric neuropsycho-oncology: what we have learned since Johnny Gunther

OBJECTIVE: This article provides a commentary on the progression of research over the last six decades into the neurobehavioral outcomes of children treated for intracranial tumors. METHODS: Published studies and literature reviews are surveyed, with illustrations from the author's ongoing longitudinal study. RESULTS: Research on late effects in pediatric brain tumors continues to increase in both volume and quality. Samples are larger and more differentiated, scope of outcome measurement has increased, and more powerful developmental research designs are being used. Particularly promising recent developments are described, including research on: imaging-behavior relationships, improved modeling of dose-volume heterogeneity in radiation therapy, improved late effects measurement, and treatments for neurobehavioral sequelae. CONCLUSIONS: There is now a large body of scientific evidence of increasing sophistication regarding the outcomes of patients receiving the most toxic treatments. It is argued that more research into the neuropsychological effects associated with "benign" tumors of childhood is needed.     

Cancer survivors in the United States: a review of the literature and a call to action

Background: The number of cancer survivors in the U.S. has increased from 3 million in 1971, when the National Cancer Act was enacted, to over 12 million today. Over 70% of children affected by cancer survive more than 10 years, and most are cured. Most cancer survivors are adults, with two-thirds of them 65 years of age or older and two-thirds alive at five years. The most common cancer diagnoses among survivors include breast, prostate and colorectal cancers. This review was conducted to better appreciate the challenges associated with cancer survivors and the opportunities healthcare providers have in making a difference for these patients.Methods: Comprehensive review of literature based on PubMed searches on topics related to cancer survivorship, and associated physical, cognitive, socio-economic, sexual/behavioral and legal issues.Results: At least 50% of cancer survivors suffer from late treatment-related side effects, often including physical, psychosocial, cognitive and sexual abnormalities, as well as concerns regarding recurrence and/or the development of new malignancies. Many are chronic in nature and some are severe and even life-threatening. Survivors also face issues involving lack of appropriate health maintenance counseling, increased unemployment rate and workplace discrimination.Conclusions: Advances in the diagnosis and treatment of cancer will lead to more survivors and better quality of life. However, tools to recognize potentially serious long-lasting side effects of cancer therapy earlier in order to treat and/or prevent them must be developed. It is incumbent upon our health care delivery systems to make meeting these patients'' needs a priority.     

Barriers to fertility preservation among pediatric oncologists

Objective: Limited research has been conducted on the extent to which fertility preservation is discussed by pediatric oncologists with patients and less is known about the discussion content. This study sought to examine factors that may influence the discussion with pediatric cancer patients and families. Methods: Qualitative data were collected using open-ended, in-depth interviews with 24 pediatric oncologists in Florida at 13 children's cancer centers, representing 87% of the centers in the state. Results: Providers practiced between 0.5 and 35 years, treated most types of pediatric cancers, and most were board certified in pediatric hematology/oncology. The main factors associated with a discussion of fertility preservation options were: (1) physician factors that contribute to communication issues with FP discussion (e.g., awareness and sense of comfort in discussing issues, perceptions of the priority); (2) parent factors (e.g., receptiveness and cultural background); (3) patient factors (e.g., receptiveness and age); and (4) institutional factors (e.g., referral sites and practice guidelines). Conclusions: Pediatric oncologists may benefit from education about fertility preservation options for pediatric cancer patients, particularly females. Practice implications: Training on how to address parents' and patients' emotions, culture, and other factors may also promote the discussion. Although guidelines have been established, barriers related to availability and affordability of resources must also be addressed.     

Consequences of vanishing twins in IVF/ICSI pregnancies

BACKGROUND: Spontaneous reductions are a possible cause of the increased morbidity in IVF singletons. The aim of this study was to assess incidence rates of spontaneous reductions in IVF/ICSI twin pregnancies and to compare short- and long-term morbidity in survivors of a vanishing co-twin with singletons and born twins. METHODS: We identified 642 survivors of a vanishing co-twin, 5237 singletons from single gestations and 3678 twins from twin gestations. All children originated from pregnancies detected by transvaginal sonography in gestational week 8. By cross-linkage with the national registries the main endpoints were prematurity, birth weight, neurological sequelae and mortality. RESULTS: Of all IVF singletons born, 10.4% originated from a twin gestation in early pregnancy. Multiple logistic regression analyses adjusted for maternal age, parity and ICSI treatment showed for birth weight <2500 g an odds ratio (OR) of 1.7 [95% confidence interval (CI) 1.2-2.2] and for birth weight <1500 g OR 2.1 (95% CI 1.3-3.6) in singleton survivors of a vanishing twin versus singletons from single gestations; corresponding figures were seen for preterm birth. This increased risk was almost entirely due to reductions that occurred at >8 weeks gestation. We found no excess risk of neurological sequelae in survivors of a vanishing co-twin versus the singleton cohort; however, OR of cerebral palsy was 1.9 (95% CI 0.7-5.2). Furthermore, we observed a correlation between onset of spontaneous reduction, i.e. the later in pregnancy the higher the risk of neurological sequelae (r = -0.09; P = 0.02). Adjusted OR of child death within the follow-up period was 3.6 (95% CI 1.7-7.6) in the survivor versus the singleton cohort. CONCLUSIONS: One in 10 IVF singletons originates from a twin gestation. Spontaneous reductions that occur at >8 weeks gestation are one of the causes for the higher risk of adverse obstetric outcome in IVF singletons.     

Patient-centered care and breast cancer survivors' satisfaction with information

Satisfaction with information is an important patient outcome and may be related to the physician's ability to elicit the patients' concerns, to consider the patients' psychosocial needs, and to involve patients in treatment decision making; these communication techniques have been described under the umbrella of "patient-centered." The aim of this study was to examine the relationship between patient-centered care and satisfaction with information among women with a history of breast cancer. We administered a questionnaire to 182 women who had completed treatment for breast cancer. Our findings suggest that, while breast cancer survivors are highly satisfied with information related to treatment, they are less satisfied with information related to the long-term physical, psychological, and social sequelae of the disease and its treatments. In multivariate analysis, patients' perception of patient-centered behaviors was strongly associated with patients' satisfaction with information. These results provide support for the theory that patient satisfaction is improved when physicians incorporate patient-centered behaviors into their care.