End-of-Life Symptom Burden among Patients with Cancer Who Were Provided Medical Assistance in Dying (MAID): A Longitudinal Propensity-Score-Matched Cohort Study.

Cancer is the primary underlying condition for most Canadians who are provided Medical Assistance in Dying (MAID). However, it is unknown whether cancer patients who are provided MAID experience disproportionally higher symptom burden compared to those who are not provided MAID. Thus, we used a propensity-score-matched cohort design to evaluate longitudinal symptom trajectories over the last 12 months of patients' lives, comparing cancer patients in Alberta who were and were not provided MAID. We utilized routinely collected retrospective Patient-Reported Outcomes (PROs) data from the Edmonton Symptom Assessment System (ESAS-r) reported by Albertans with cancer who died between July 2017 and January 2019. The data were analyzed using mixed-effect models for repeated measures to compare differences in symptom trajectories between the cohorts over time. Both cohorts experienced increasing severity in all symptoms in the year prior to death (ß from 0.086 to 0.231, p ≤ .001 to .002). Those in the MAID cohort reported significantly greater anxiety (ß = -0.831, p = .044) and greater lack of appetite (ß = -0.934, p = .039) compared to those in the non-MAID cohort. The majority (65.8%) of patients who received MAID submitted their request for MAID within one month of their death. Overall, the MAID patients did not experience disproportionally higher symptom burden. These results emphasize opportunities to address patient suffering for all patients with cancer through routine collection of PROs as well as targeted and early palliative approaches to care.

Making sense of the cancer journey: Pediatric cancer survivors' and their parents' autobiographical memories.

PURPOSE: Youth diagnosed with acute lymphoblastic leukemia (ALL) and their caregiver's experience a myriad of challenges in all domains of health that extend beyond treatment. Yet, little is known about how the cancer experience, and recollections associated with the experience, impact survivorship. We explored pediatric ALL survivors' and their caregivers' autobiographical memories of the cancer experience from diagnosis onwards. METHODS: Survivors of ALL, and one of their caregivers, were recruited through a local clinic. Survivors and their caregivers completed a demographic survey and semi-structured, private, one-on-one interviews. Demographic information were analyzed using descriptive statistics. Interviews were transcribed verbatim and analyzed using reflexive thematic analysis at the level of the individual and dyad. RESULTS: Insights from survivors (N = 19; Mage = 15.3 years) and their caregivers (n = 19; Mage = 45.4 years) were captured. Analyses generated two themes contingent on role (i.e., survivor or caregiver): (1) It is hard to recall my cancer experience and (2) We did as much as we could to manage our child's cancer experience and two unified themes (present in both survivors and their caregivers): (3) It took a village to get through the cancer experience and (4) The cancer diagnosis and experience has had a lasting impact. CONCLUSIONS: Findings highlight the varied and long-lasting ways cancer impacts survivors of pediatric ALL and their caregivers. Survivors had difficultly remembering their experience or felt that information was withheld and were acutely aware of their caregiver's distress. Caregivers were cautious and intentionally limited the information they shared. IMPLICATIONS FOR CANCER SURVIVORS: Survivors desired to be included within, or told about, decisions related to their healthcare and were acutely aware of their caregiver's distress. Efforts should be made to communicate with survivors (from diagnosis onward) openly and to consider strategies to minimize the short- and long-term impacts of pediatric ALL among survivors and their caregivers.

Bedtime digital media use, sleep and fatigue among survivors of childhood cancer, their siblings and healthy control sibling pairs.

Bedtime digital media use (BDM) is linked to poor sleep and fatigue in many populations. Pediatric cancer patients have been observed to engage in BDM in clinical settings, but it is unknown whether BDM rates are higher in this population or how this impacts their sleep and fatigue during treatment and into survivorship. The goal of this study was to evaluate patterns of BDM and its relationship with sleep and fatigue in a sample of pediatric cancer survivors and to compare these patterns with children from their own family (i.e. siblings) and children from unaffected families (i.e. healthy matched controls and siblings of controls). Ninety-nine children (4 groups: 24 acute lymphoblastic leukemia survivors, 13 survivor siblings, 33 controls, 29 control siblings) ages 8-18 were recruited from a long-term survivor clinic at a large children's hospital and via community advertisements. Survivors were 2-7 years post-treatment (M = 4.80 years). Children's BDM was parent-reported. Children completed 7 consecutive days of sleep actigraphy and the PedsQL Multidimensional Fatigue Scale. Most survivors (66.67%) engaged in BDM; smartphones were the most common medium. BDM patterns were equivalent across survivors, their siblings, controls, and control siblings. Statistical trends suggested that BDM was associated with fewer minutes of sleep and greater fatigue for all children; these relationships were equivalent across groups. BDM was common among survivors, but usage was not different from their own siblings or compared to healthy control children and sibling pairs. This study underscores the importance of assessing bedtime digital media use in childhood cancer survivors, although other factors impacting sleep should be explored. Clinicians should emphasize established recommendations for healthy media use and sleep habits in pediatric oncology settings.

Health related quality of life in children with sickle cell disease: A systematic review and meta-analysis.

This review had three aims: 1) describe the measures used to assess health-related quality of life (HRQL) in pediatric patients diagnosed with sickle cell disease (SCD); 2) document the biopsychosocial factors related to HRQL in pediatric patients diagnosed with SCD; and 3) complete a meta-analysis comparing HRQL in pediatric patients diagnosed with SCD to healthy controls. Included studies were published in English, quantitatively assessed HRQL as a primary aim, in both SCD and controls, and included participants between 0 and 21 years of age. The final review included 66 articles, with a total of 8642 participants with SCD, 4 months-21 years of age, and 62,458 controls, 5-27 years of age. HRQL was predominately measured using the Pediatric Quality of Life Inventory Generic Core and Sickle Cell Disease Module. Meta-analyses revealed children with SCD had significantly worse HRQL compared to healthy controls (standardized mean difference = -0.93, 95% CI = -1.25, -0.61, p < 0.00001). Worse HRQL was associated with more severe SCD, female sex, and pain. The findings indicate that children with SCD are at risk for worse HRQL compared to their healthy peers and their HRQL may be impacted by several biopsychosocial factors. Future research is needed to examine how sociocultural factors uniquely impact this population and their overall quality of life.

Symptom Burden and Complexity in the Last 12 Months of Life among Cancer Patients Choosing Medical Assistance in Dying (MAID) in Alberta, Canada.

BACKGROUND: In 2019, cancer patients comprised over 65% of all individuals who requested and received Medical Assistance in Dying (MAID) in Canada. This descriptive study sought to understand the self-reported symptom burden and complexity of cancer patients in the 12 months prior to receiving MAID in Alberta. METHODS: Between July 2017 and January 2019, 337 cancer patients received MAID in Alberta. Patient characteristics were descriptively analyzed. As such, 193 patients (57.3%) completed at least one routine symptom-reporting questionnaire in their last year of life. Mixed effects models and generalized estimating equations were utilized to examine the trajectories of individual symptoms and overall symptom complexity within the cohort over this time. RESULTS: The results revealed that all nine self-reported symptoms, and the overall symptom complexity of the cohort, increased as patients' MAID provision date approached, particularly in the last 3 months of life. While less than 20% of patients experienced high symptom complexity 12 months prior to MAID, this increased to 60% in the month of MAID provision. CONCLUSIONS: Cancer patients in this cohort experienced increased symptom burden and complexity leading up to their death. These findings could serve as a flag to clinicians to closely monitor advanced cancer patients' symptoms, and provide appropriate support and interventions as needed.

Psychosocial risk, symptom burden, and concerns in families affected by childhood cancer.

PURPOSE: The revised Psychosocial Assessment Tool (PATrev) is a common family-level risk-based screening tool for pediatric oncology that has gained support for its ability to predict, at diagnosis, the degree of psychosocial support a family may require throughout the treatment trajectory. However, ongoing screening for symptoms and concerns (e.g., feeling alone, understanding treatment) remains underutilized. Resource limitations necessitate triaging and intervention based on need and risk. Given the widespread use of the PATrev, we sought to explore the association between family psychosocial risk, symptom burden (as measured by the revised Edmonton Symptom Assessment System (ESAS-r)), and concerns (as measured by the Canadian Problem Checklist (CPC)). METHODS: Families (n = 87) with children ≤ 18 years of age (M = 11.72, male: 62.1%) on or off treatment for cancer were recruited from the Alberta Children's Hospital. One parent from each family completed the PATrev and the CPC. Participants 8-18 years of age completed the ESAS-r. RESULTS: Risk category (universal/low risk = 67.8%, targeted/intermediate risk = 26.4%, clinical/high risk = 5.7%) predicted symptom burden (F[2, 63.07] = 4.57, p = .014) and concerns (F[2, 82.06] = 16.79, p < .001), such that universal risk was associated with significantly lower symptom burden and fewer concerns. CONCLUSION: Family psychosocial risk is associated with cross-sectionally identified concerns and symptom burden, suggesting that resources might be prioritized for families with the greatest predicted need. Future research should evaluate the predictive validity of the PATrev to identify longitudinal concerns and symptom burden throughout the cancer trajectory.

Social adjustment in survivors of acute lymphoblastic leukemia without cranial radiation therapy.

OBJECTIVE: To evaluate group differences in social adjustment in survivors of pediatric acute lymphoblastic leukemia (ALL) compared to survivor siblings and controls; identify disease-related predictors of social adjustment in survivors; and explore whether executive functioning explained differences in social adjustment across groups and between disease-related predictors. METHODS: Survivors of pediatric ALL (n = 38, average age at diagnosis = 4.27 years [SD = 1.97]; average time off treatment = 4.83 years [SD = 1.52]), one sibling (if available, n = 20), and one parent from each family were recruited from a long-term survivor clinic. Healthy age- and sex-matched controls (n = 38) and one parent from each family were recruited from the community. Parents completed the Behavioral Assessment System for Children, Parent Rating Scale (BASC-3) Social Withdrawal subscale as a measure of social adjustment, and the Behavior Rating Inventory of Executive Functions (BRIEF-2) as a measure of executive function for each of their children. Multilevel modeling and mediation analysis were used to achieve the study aims. RESULTS: Parents reported that survivors had significantly worse social adjustment compared to controls (b = 6.34, p = .004), but not survivor siblings. Among survivors, greater time off treatment (b = 2.06, p = .058) and poorer executive functioning (b = 0.42, p = .006) were associated with worse social adjustment. Executive function did not mediate differences in social withdrawal between survivors and controls or the relationship between time off treatment and social withdrawal among survivors. CONCLUSIONS: Survivors of pediatric ALL presenting to follow-up programs should be screened for difficulties with social adjustment. Future research should examine treatment- and nontreatment-related factors contributing to poorer social outcomes.

The pain of survival: Prevalence, patterns, and predictors of pain in survivors of childhood cancer.

OBJECTIVE: Survivors of childhood cancer experience late effects as a result of their cancer treatment. Evidence for the prevalence of pain as a late effect has been equivocal. This study aimed to describe the prevalence and patterns of pain and biospsychosocial variables that may be related to pain in this population. METHOD: Survivors of childhood cancer (n = 299; 52.5% male; median age = 16.1[4.6-32.6] years; years off therapy = 9.1[2.0-23.7]) were included. Survivors completed a health assessment questionnaire as part of their long-term survivor clinic appointment (median = 3.0 appointments, range = 1.0-7.0) annually or biannually between 2014 and 2017 (Time 1-Time 4). Prevalence of pain was examined and latent class analysis (LCA) was used to identify patterns of pain based on longitudinal reports of pain. Binary logistic regression examined biopsychosocial variables at Time 1 (T1) associated with class membership. RESULTS: Forty-seven percent of survivors reported pain during at least one clinic visit. Headaches were the most prevalent type of pain (26.4%). Survivors of Wilms' Tumor and Ewing's Sarcoma reported the highest prevalence of pain (51.5% and 50.0%, respectively). LCA revealed two clinically relevant profiles: "infrequent or no pain" (74.3%) and "persistent pain" (25.7%). Logistic regression showed that female sex (odds ratio, OR = 2.69, 95% confidence interval, CI [.99, 7.31]), depressive symptomatology at T1 (OR = 2.27, 95% CI [1.31, 3.94]), and drinking to intoxication at T1 (OR = 3.07, 95% CI [1.03, 9.15]), were related to persistent pain. CONCLUSION: Pain is prevalent among survivors of childhood cancer. Future research should characterize the experience of pain in this population so interventions may be developed. Assessment of pain during regular long-term follow-up appointments is warranted. (PsycInfo Database Record (c) 2021 APA, all rights reserved).

Perceived Health among Adolescent and Young Adult Survivors of Childhood Cancer.

Survivors of childhood cancer (SCCs) are at increased risk of late effects, which are cancer- and treatment-related side-effects that are experienced months to years post-treatment and encapsulate a range of physical, cognitive and emotional problems including secondary malignancies. Perceived health can serve as an indicator of overall health. This study aims to (1) understand how a patient reported outcome (PRO) of perceived health of SCCs compares to controls who have not had a cancer diagnosis and (2) examine the relationships between perceived health and demographic and clinical variables, and health behavior. A total of 209 SCCs (n = 113 (54.10%) males; median age at diagnosis = 6.50 years; median time off treatment = 11.10 years; mean age at study = 19.00 years) were included. SCCs completed annual assessments as part of Long-Term Survivor Clinic appointments, including a question on perceived health answered on a five-point Likert scale. Data were collected retrospectively from medical charts. Perceived health of SCCs was compared to a control group (n = 836) using data from the 2014 Canadian Community Health Survey. Most SCCs (67%) reported excellent or very good health. The mean perceived health of SCCs (2.15 ± 0.91) was not statistically different from population controls (2.10 ± 0.87). Pain (B = 0.35; p < 0.001), physical activity (B = -0.39; p = 0.013) and concerns related to health resources (B = 0.59; p = 0.002) were significant predictors of perceived health. Factors shown to influence SCCs' perceived health may inform interventions. Exploration into how SCCs develop their conception of health may be warranted.

Pain in long-term survivors of childhood cancer: A systematic review of the current state of knowledge and a call to action from the Children's Oncology Group.

Survivors of childhood cancer may be at risk of experiencing pain, and a systematic review would advance our understanding of pain in this population. The objective of this study was to describe: 1) the prevalence of pain in survivors of childhood cancer, 2) methods of pain measurement, 3) associations between pain and biopsychosocial factors, and 4) recommendations for future research. Data sources for the study were articles published from January 1990 to August 2019 identified in the PubMed, PsycINFO, EMBASE, and Web of Science data bases. Eligible studies included: 1) original research, 2) quantitative assessments of pain, 3) articles published in English, 4) cancers diagnosed between birth and age 21 years, 5) survivors at 5 years from diagnosis and/or at 2 years after therapy completion, and 6) a sample size >20. Seventy-three articles were included in the final review. Risk of bias was considered using the Cochrane risk of bias tool. The quality of evidence was evaluated according to Grading of Recommendations Assessment Development and Evaluation (GRADE) criteria. Common measures of pain were items created by the authors for the purpose of the study (45.2%) or health-related quality-of-life/health status questionnaires (42.5%). Pain was present in from 4.3% to 75% of survivors across studies. Three studies investigated chronic pain according the definition in the International Classification of Diseases. The findings indicated that survivors of childhood cancer are at higher risk of experiencing pain compared with controls. Fatigue was consistently associated with pain, females reported more pain than males, and other factors related to pain will require stronger evidence. Theoretically grounded, multidimensional measurements of pain are absent from the literature.

Childhood Cancer Survivorship: Daily Challenges.

Survivors of childhood cancer are at risk of experiencing fatigue, pain, lower levels of physical activity, increased engagement in risky health behavior, and poor social adjustment, after finishing treatment. Risks are more pronounced for survivors of specific diagnoses or receiving specific treatment protocols. Interventions to address these outcomes are in their infancy. Future research should focus on exploring the antecedents and consequences of these outcomes. In the meantime, researchers and cancer centers should attempt to provide high-quality and accessible health information to survivors through various media outlets to encourage healthy behaviors.

Sleep Disturbances in Survivors of Pediatric Acute Lymphoblastic Leukemia and Their Siblings.

OBJECTIVE: Sleep disturbances have been identified by patients with cancer as common and distressing; however, conflicting evidence about the prevalence of these outcomes exists for survivors of childhood cancers. Additionally, little is known about how the experience of cancer might impact survivor siblings' sleep. The current study compared the sleep of survivors of acute lymphoblastic leukemia who were 2-7 years off therapy and their siblings to healthy control/sibling dyads. METHODS: Participants (survivors, n = 45; survivor siblings, n = 27; controls, n = 45; control siblings, n = 41; 58% male) aged 8-18 (m = 11.64) completed a 7-day sleep diary and seven consecutive days of actigraphy. Parents (n = 90) completed the Children's Sleep Habits Questionnaire for each of their children. RESULTS: No between-group differences were found on measures of sleep diaries or actigraphy. Parents reported that survivor siblings had significantly poorer sleep habits than survivors or controls. For survivors, greater time off treatment and younger age at diagnosis were associated with less total sleep time, more wake after sleep onset, and decreased sleep efficiency via actigraphy. CONCLUSION: Sleep across all groups was consistent and below national guidelines. Although the survivor group did not have poorer sleep compared to their siblings or matched controls, within the survivor group, those who were diagnosed at an earlier age and those who were further off treatment had more disrupted sleep. Parent reports suggested that survivor siblings may be at risk for sleep problems.

Fear of cancer recurrence among survivors of childhood cancer.

OBJECTIVES: Fear of cancer recurrence (FCR) has not been widely explored in survivors of childhood cancer. Yet, childhood survivors are at risk of experiencing late effects and may be especially vulnerable. The aims of the current study were to conduct a retrospective chart review to determine the prevalence and persistence of FCR among survivors of childhood cancer and to examine factors that may be related to FCR. METHODS: Survivors of childhood cancer (n = 228, mean attained age = 14.5 years [range = 4.7-21 years]; mean diagnosis age = 4.4 years [range = 0-16.5 years]; mean time off treatment = 8.7 years [range = 2.8-19.3 years]) seen in a Long-Term Survivor Clinic (LTSC) completed questionnaires at each clinic visit detailing their current health. FCR was measured with a single item. Data from questionnaires from 2011 to 2018 were analyzed retrospectively. Descriptive statistics and a random effects model were used to address study aims. RESULTS: FCR was reported in 43% (n = 98) of survivors at least once across all clinic visits. Among survivors reporting FCR at least once, 66% were diagnosed with cancer under the age of 5, and 64% were 13 years or older at their most recent follow-up. Twenty-one percent of survivors (n = 48/224) reported FCR during at least 50% of their visits. Survivors with a higher number of depressive symptoms were more likely to report FCR (OR = 1.66, P = .03). CONCLUSIONS: FCR is prevalent among survivors of childhood cancer and is related to other health concerns. Research is needed to understand who is at risk and how to.

Screening for psychosocial distress in pediatric cancer patients: An examination of feasibility in a single institution.

Despite the availability of tools to assess psychosocial screening in pediatric oncology, little is known about the feasibility and acceptability of systematic screening. We aimed to assess the feasibility of implementing a tool, or set of tools, capable of screening for psychosocial distress in pediatric cancer patients across the cancer continuum (on treatment, off treatment). Psychometric criteria were also evaluated. Patients 8-18 years were recruited from a pediatric oncology program. Patients completed self-report measures of the Distress Thermometer (DT) and Pediatric Quality of Life Inventory (PedsQL). One parent of each patient completed three screening tools: DT (proxy-report); PedsQL (proxy-report), and the Psychosocial Assessment Tool adapted for the Canadian context (PATrev), as well as a measure of patient psychological functioning (Behavioral Assessment System for Children-2), and an assessment of screening tool acceptability. Recruitment rates and acceptability informed feasibility of implementation. Ninety-five patients (58 men) with a mean age of 11.47 participated in the study (on treatment, n = 43; off treatment, n = 52). Recruitment rates were on treatment: 56.6% and off treatment: 47.3%. Mean acceptability scores of tools ranged from 3.41 to 4.97 out of 7. Screening tools were comparable with respect to their psychometric properties. The DT took the least amount of time to complete, while the PATrev offered the most robust data with respect to psychometrics. Feasibility of screening for psychosocial distress with our tool was moderate and may be enhanced when administered by a known health-care provider. Future research exploring how to further enhance feasibility of implementation for pediatric cancer patients is warranted.

Falling through the cracks. A thematic evaluation of unmet needs of adult survivors of childhood cancers.

OBJECTIVE: The population of adult survivors of childhood cancers (ASCCs) is growing, resulting in unique long-term challenges. This study explored experiences of perceived unmet ASCC survivorship needs. METHODS: We invited ASCCs to complete surveys sent through the cancer registry. Four open-ended questions allowed participants to write in comments. We analyzed responses to these open-ended questions thematically, employing a process of constant comparison. RESULTS: Our sample included 94 ASCCs who completed open-ended questions (61 female; aged 20-78 years, mean age = 34.47, SD = 11.84, mean = 23.27 years post diagnosis). Identified themes included (1) overlooked experiences of distress; (2) lack of counseling: system, patient, and family barriers; (3) difficulty negotiating future life milestones exacerbated by lack of knowledge; and (4) dissatisfaction with service provision: past and present. Prevalent issues identified by participants included lack of supportive care to address needs, distress due to missed developmental milestones as a result of cancer, lack of knowledge about late-term and long-term effects of cancer treatment, and concern over absence of organized long-term follow-up. CONCLUSIONS: Adult survivors of childhood cancers continue to experience unmet needs during their cancer diagnosis, treatment, and long into survivorship due to the treatment for cancer and ongoing side effects. Solutions could focus on addressing the needs of survivors to bridge system gaps and barriers. Specifically, there is a need to improve psychological interventions and transitions from pediatric to adult-care facilities.

Social adjustment and repressive adaptive style in survivors of pediatric cancer.

PURPOSE: The aim of the study was to explore the relationship between repressive adaptive style and self-reports of social adjustment in survivors of pediatric cancer compared to their siblings. We hypothesized that there would be a greater proportion of repressors among survivors of pediatric cancer compared to siblings, and that repressive adaptive style would be significantly associated with more positive self-reports of social adjustment. METHODS: We utilized a cross-sectional approach. Seventy-seven families participated. Survivors of pediatric cancer (n = 77, 48% male; 8-18 years of age) and one sibling (n = 50, 48% male; 8-18 years of age) completed measures assessing repressive adaptive style and social adjustment. As well, one parent from each family completed a socio-demographic questionnaire. Questionnaire packages were mailed to eligible families who agreed to participate, and were mailed back to investigators in a pre-addressed, pre-stamped envelope. RESULTS: Chi-square analyses revealed there was no significant difference in the proportion of repressors among survivors and siblings. Social adjustment scores were subjected to a two (group: survivor, sibling) by two (repressor, nonrepressor) ANCOVA with gender and age as covariates. There was a significant main effect of repressive adaptive style (F = 5.69, p < .05, η2 = 0.05) with a modest effect. Survivors and siblings with a repressive style reported significantly higher social adjustment scores (M = 106.91, SD = 11.69) compared to nonrepressors (M = 99.57, SD = 13.45). CONCLUSIONS: Repressive adaptive style explains some of the variance in survivors and siblings' self-reports of social adjustment. Future research should aim to better understand the role of the repressive adaptive style in survivors and siblings of children with cancer.

Unmet Needs of Adult Survivors of Childhood Cancers: Associations with Developmental Stage at Diagnosis, Cognitive Impairment, and Time from Diagnosis.

PURPOSE: Adult survivors of childhood cancers (ASCCs) are a unique and growing population. Because these individuals were diagnosed in childhood, their developmental stage at diagnosis may influence medical sequelae and perception of their cancer diagnosis and potentially result in long-term complications and challenges. Our aim was to determine how developmental stage, time since diagnosis, and cognitive impairment relate to Canadian ASCC distress and unmet needs. METHODS: Canadian ASCCs aged 19-77 years (N = 115) diagnosed between ages 0 and 5 (n = 25), 6 and 12 (n = 22), or 13 and 18 (n = 68) completed demographic, neurocognitive self-report, depression, and anxiety and unmet needs questionnaires. RESULTS: The developmental stage predicted distress, ß = -0.29, p = 0.01. Survivors diagnosed in middle childhood reported significantly more distress than those diagnosed in adolescence. Shorter time since diagnosis predicted greater psychosocial needs, ß = -0.24, p = 0.05, and greater distress, ß = -0.22, p = 0.05. Greater memory impairment predicted higher need across outcomes, ß = -0.36-0.61, p < 0.05. In adjusted analyses for unmet needs, endorsement of cancer affecting education and/or work importantly altered outcomes. CONCLUSION: Our results indicate that greater self-reported memory impairment increases childhood cancer survivors' care needs. We additionally suggest that supportive care interventions might best target those reporting work or education interruption due to cancer. Identification of ASCCs who report work/school interruptions may provide a quick screen for health providers to assess possible need for intervention. ASCCs still experience unmet needs long into survivorship.

Systematic review and meta-analysis of health-related quality of life in pediatric CNS tumor survivors.

BACKGROUND: Pediatric central nervous system (CNS) tumor survivors are at high risk for numerous late effects including decreased health-related quality of life (HRQOL). Our objective was to summarize studies describing HRQOL in pediatric CNS tumor survivors and compare HRQOL outcomes in studies that included a comparison group. PROCEDURE: EMBASE, MEDLINE, and PsychINFO were used to identify relevant articles published until August, 2016. Eligible studies reported outcomes for pediatric CNS tumor survivors diagnosed before age 21, at least 5 years from diagnosis and/or 2 years off therapy and used a standardized measure of HRQOL. All data were abstracted by two reviewers. Random-effects meta-analyses were performed using Review Manager 5.0. RESULTS: Of 1,912 unique articles identified, 74 were included in this review. Papers described 29 different HRQOL tools. Meta-analyses compared pediatric CNS tumor survivors to healthy comparisons and other pediatric cancer survivors separately. HRQOL was significantly lower for CNS (n = 797) than healthy comparisons (n = 1,397) (mean difference = -0.54, 95% confidence interval [CI] = -0.72 to -0.35, P < 0.001, I2 = 35%). HRQOL was also significantly lower for CNS (n = 244) than non-CNS survivors (n = 414) (mean difference = -0.56, 95% CI = -0.73 to -0.38, P < 0.00001, I2 = 0%). CONCLUSIONS: Pediatric CNS tumor survivors experience worse HRQOL than healthy comparisons and non-CNS cancer survivors. Future HRQOL work should be longitudinal, and/or multisite studies that examine HRQOL by diagnosis and treatment modalities.