Survivors Of Childhood Brain Tumors Research Articles

Bio-behavioural research in a rare disease population: a doctoral student's experience.

Novice researchers who aspire to contribute to the body of knowledge concerning rare diseases face unique challenges in developing and conducting studies. These include unknown effect sizes in previous research, limitations in recruitment and enrolment, and managing data from a multi-site sample. To describe the challenges in researching rare diseases and possible solutions using a doctoral student exemplar from a cross-sectional correlational study of fatigue. The authors discuss the lessons learned from the study, including the challenges in recruitment, communication, collecting biological data and managing data in general. They posit possible solutions, including improving multi-site coordination, feasible methods for exploring sleep and stress, and measures to prevent equipment and data loss. Rare populations, such as young survivors of childhood brain tumours, deserve a voice in building the body of knowledge needed for more precise, personalised healthcare. It is possible with foresight for the novice researcher to make such a contribution. Improved knowledge and assessment of symptoms during childhood will improve the detection of health risk factors and enable earlier intervention.

Considerations for Reliable Digit Span as a performance validity test for long-term survivors of childhood brain tumors

The Reliable Digit Span (RDS) is a performance validity test (PVT) used widely within non-clinical samples, but its utility is in question in clinical groups with cognitive impairment. To investigate, RDS scores were calculated and correlated with the Neurological Predictor Scale, an informant-reported Activities of Daily Living score, and a proxy measure of intelligence (Vocabulary) for 83 adult survivors of childhood brain tumors and 105 healthy controls. Analyses were covaried for age at examination. Participants were divided into passing and failing groups at each RDS cutoff, and ANCOVAs for each of the three variables of interest covaried for age at the examination were run. RDS was correlated with all three variables of interest in survivors but only Vocabulary in controls. At the ≤7 cutoff, passing and failing survivors demonstrated significant differences across all variables of interest, while passing and failing controls differed only on Vocabulary. Differences were also found between passing and failing survivors at lower cutoffs. RDS is related to and likely impacted by various neurological and cognitive challenges faced by brain tumor survivors. Using the standard RDS cutoff of ≤7 may result in inaccurate interpretation of valid performance in this population; therefore, the use of other PVTs is recommended.

Methylphenidate improves weight control in childhood brain tumor survivors with hypothalamic obesity.

Hypothalamic obesity causes unrelenting weight gain for childhood brain tumor survivors. No single therapy has proven effective for treatment. We aimed to evaluate effectiveness of long-term methylphenidate therapy on body mass index (BMI) change in children with hypothalamic obesity. A retrospective analysis included children with a history of brain tumor and hypothalamic obesity receiving methylphenidate (10-60mg/day) for hypothalamic obesity. Subjects were evaluated for BMI trajectory before and after methylphenidate start. Given that z-scores can be skewed in severely obese children, we calculated BMI as a percent of the BMI at the 95th percentile for the child's age and gender (BMI% 95th). Twelve patients with hypothalamic obesity completed methylphenidate therapy for at least 6months (median 3.1years, range 1.0-5.8years). All subjects had a suprasellar tumor (nine [75%] with craniopharyngioma) and pituitary dysfunction. Pretreatment median BMI percent of the 95th percentile was 125.6% (interquartile range [IQR] 25-75: 115.3-138.3%) with BMI z-score of 2.4 (IQR 25-75: 2.1-2.6). Following methylphenidate treatment, there was a 69.9% reduction in the median slope of BMI change. Eleven of 12 patients (92%) had a reduction in the slope of their BMI change on methylphenidate treatment. Postmethylphenidate median BMI percent of the 95th percentile decrease to 115.2% (IQR 25-75: 103.6-121.2%) with median BMI z-score of 2.1 (IQR 25-75: 1.8-2.2). Mild side effects were noted in six patients. Methylphenidate use reduced and sustained BMI change in children with hypothalamic obesity. Stimulant therapy is an effective first-line agent for treatment of hypothalamic obesity.

Symptom burden trajectories experienced by patients with brain tumors.

Survivors of childhood brain tumors experience persistent health concerns across their lifespan. In the current study, the authors evaluated changes in symptom burden over the course of 12months using pediatric Patient-Reported Outcomes Measurement Information System (PROMIS) measures. Data from 202 survivors aged 8 to 21years and 262 parents of survivors who were aged 5 to 21years were analyzed. All completed a PROMIS Cognition short form and computerized adaptive tests of pediatric Anxiety, Depressive Symptoms, Fatigue, Mobility, Upper Extremity Function, and Peer Relationships. Approximately one-half of participants (223 participants: 97 survivors of childhood brain tumors and 126 parents) completed the 12-month follow-up. Linear mixed-effects models evaluated group-level symptoms over time. Cox proportional hazard models explored whether symptoms predicted survival, and latent class growth analysis investigated patterns of individual-level symptom changes over time. Linear mixed-effects models demonstrated that patient-reported Cognition and parent-reported Anxiety worsened over time. Latent class growth analysis results indicated that patient and parent reports diverged, both with regard to the number of classes identified and in the trends of these classes. Parents and patients reported similar patterns of depression over time. For the other areas, parents either were more likely to observe different patterns (Peer Relationships and Mobility) or less likely to observe different patterns (Upper Extremity Function, Cognition, Anxiety, and Fatigue). Baseline patient-reported Mobility and Upper Extremity Function were found to be associated with survival. Survivors of childhood brain tumors demonstrated different trajectory patterns of symptom burden. Along with baseline functioning status and days since treatment, patient-reported Mobility and Upper Extremity Function were associated with survival, suggesting a possible role for patient-reported outcomes in clinical care, especially individualized, tailored assessments such as PROMIS.

Double Dissociation of Auditory Attention Span and Visual Attention in Long-Term Survivors of Childhood Cerebellar Tumor: A Deterministic Tractography Study of the Cerebellar-Frontal and the Superior Longitudinal Fasciculus Pathways.

Right cerebellar-left frontal (RC-LF) white matter integrity (WMI) has been associated with working memory. However, prior studies have employed measures of working memory that include processing speed and attention. We examined the relationships between the RC-LF WMI and processing speed, attention, and working memory to clarify the relationship of RC-LF WMI with a specific cognitive function. Right superior longitudinal fasciculus II (SLF II) WMI and visual attention were included as a negative control tract and task to demonstrate a double dissociation. Adult survivors of childhood brain tumors [n = 29, age: M = 22 years (SD = 5), 45% female] and demographically matched controls were recruited (n = 29). Tests of auditory attention span, working memory, and visual attention served as cognitive measures. Participants completed a 3-T MRI diffusion-weighted imaging scan. Fractional anisotropy (FA) and radial diffusivity (RD) served as WMI measures. Partial correlations between WMI and cognitive scores included controlling for type of treatment. A correlational double dissociation was found. RC-LF WMI was associated with auditory attention (FA: r = .42, p = .03; RD: r = -.50, p = .01) and was not associated with visual attention (FA: r = -.11, p = .59; RD: r = -.11, p = .57). SLF II FA WMI was associated with visual attention (FA: r = .44, p = .02; RD: r = -.17, p = .40) and was not associated with auditory attention (FA: r = .24, p = .22; RD: r = -.10, p = .62). The results show that RC-LF WMI is associated with auditory attention span rather than working memory per se and provides evidence for a specificity based on the correlational double dissociation.

Circulating leptin levels are associated with adiposity in survivors of childhood brain tumors

Survivors of Childhood Brain Tumors (SCBT) are at a higher risk of developing cardiovascular disease and type 2 diabetes compared to the general population. Adiposity is an important risk factor for the development of these outcomes, and identifying biomarkers of adiposity may help the stratification of survivors based on their cardiovascular risk or allow for early screening and interventions to improve cardiometabolic outcomes. Leptin is an adipokine that positively correlates with the adipose mass in the general population and is a predictor of adverse cardiometabolic outcomes, yet its association with adiposity in SCBT has not been studied. The aim of this study was to determine if leptin levels are associated with the adipose mass in SCBT, and to define its predictors. This cross-sectional study included 74 SCBT (n = 32 females) with 126 non-cancer controls (n = 59 females). Total adiposity was measured using Bioelectrical Impendence Analysis (BIA) and central adiposity was measured using waist-to-hip ratio (WHR) and waist-to-height ratio (WHtR). We used multivariable linear regression analysis to determine if leptin predicts adiposity in SCBT and adjusted for age, sex, puberty, and cancer status. Leptin correlated strongly with total (p < 0.001) and central (WHR p = 0.001; WHtR p < 0.001) adiposity in SCBT and non-cancer controls. In conclusion, leptin is a potential biomarker for adiposity in SCBT, and further investigation is needed to clarify if leptin is a predictor of future cardiometabolic risk in SCBT.

Post-Treatment Thyroid Diseases in Children with Brain Tumors: A Single-Center Experience at "Prof. Dr. Ion Chiricuță" Institute of Oncology, Cluj-Napoca.

Aim of study: The purpose of the study was to evaluate the association of thyroid dysfunction occurring in pediatric patients treated for brain tumors. Patients and methods: A total of 255 patients with brain tumors were treated between 2001 and 2018 at the “Prof. Dr. Ion Chiricuță” Institute of Oncology, Cluj-Napoca. Due to a minimum follow-up of 4 years, we studied 184 out of the 255 patients. The cohort included 69 girls (37.5%) and 109 boys (62.5%), with a median age of 8.4 years. The evaluated tumors included medulloblastomas (47 patients), astrocytomas (44 patients), ependymomas (22 patients), gliomas (20 patients), germ cell tumors (12 patients), primitive neuroectodermal tumors (4 patients), as well as other types of tumors (15 patients); in 20 of the cases, biopsy could not be performed. Results: There was a 60% overall survival rate; among the 120 surviving patients, 11 (9.1%) were diagnosed with iatrogenic thyroid disease. We observed an important number of iatrogenic thyroid disease cases in this group of patients, thus revealing the importance of long-term thyroid function evaluation in all children who finalized their treatment for brain tumors. Through this study, we aimed to provide an accurate image of the methodology of monitoring for thyroid dysfunction in childhood brain tumor survivors. Conclusion: Given the fact that the probability of developing thyroid dysfunction in the pediatric population treated for brain tumors is not rare, we recommend that childhood brain tumor survivors be monitored for iatrogenic thyroid disease, in order to provide early diagnosis and treatment.

Abnormal weight gain with fatigue and stress in early survivorship after childhood brain tumor diagnosis.

Diagnosis and treatment for individuals with brain tumors during childhood involves sequelae, including abnormal weight gain. This symptom is commonly clustered with fatigue and increased risk for cardiovascular disease. Children's Oncology Group recommendations include annual surveillance of body mass index (BMI) and cardiometabolic comorbidities; however, there has been little emphasis on individualized screening early in survivorship. The primary purpose of this paper is to describe the findings of abnormal weight gain and its correlates in a sample of young childhood brain tumor survivors during early survivorship. This cross-sectional multi-site study included brain tumor survivors of ages 8-12 years who were less than 6 years posttreatment. Convenience sampling from two pediatric cancer centers in the southern United States was utilized. Data collected included BMI, parent report of sleep, and child report of fatigue and stress. The sample (N = 21) consisted of children who had received chemotherapy, radiation treatment, and surgery for childhood brain tumor. BMI in overweight and obese categories exceeded normative samples with 38% at or above the 85th percentile. There were clinically significant relationships with fatigue, stress about weight, tumor location, cranial radiation, chemotherapy, and recurrence PRACTICE IMPLICATIONS: Screening for abnormal weight gain and related factors, such as fatigue should begin early in survivorship after childhood brain tumor treatment completion with the aim of health promotion and disease prevention. Adiposity measurement techniques should be utilized in future clinical and research settings to improve assessment of cardiometabolic risk.

Radiation-induced accelerated aging of the brain vasculature in young adult survivors of childhood brain tumors.

BackgroundCranial radiotherapy may damage the cerebral vasculature. The aim of this study was to understand the prevalence and risk factors of cerebrovascular disease (CVD) and white matter hyperintensities (WMHs) in childhood brain tumors (CBT) survivors treated with radiotherapy.MethodsSeventy CBT survivors who received radiotherapy were enrolled in a cross-sectional study at a median 20 years after radiotherapy cessation. The prevalence of and risk factors for CVD were investigated using MRI, MRA, and laboratory testing. Tumors, their treatment, and stroke-related data were retrieved from patients’ files.ResultsForty-four individuals (63%) had CVD at a median age of 27 years (range, 16-43 years). The prevalence rates at 20 years for CVD, small-vessel disease, and large-vessel disease were 52%, 38%, and 16%, respectively. Ischemic infarcts were diagnosed in 6 survivors, and cerebral hemorrhage in 2. Lacunar infarcts were present in 7, periventricular or deep WMHs in 34 (49%), and mineralizing microangiopathy in 21 (30%) survivors. Multiple pathologies were detected in 44% of the participants, and most lesions were located in a high-dose radiation area. Higher blood pressure was associated with CVD and a presence of WMHs. Higher cholesterol levels increased the risk of ischemic infarcts and WMHs, and lower levels of high-density lipoprotein and higher waist circumference increased the risk of lacunar infarcts.ConclusionsTreating CBTs with radiotherapy increases the risk of early CVD and WMHs in young adult survivors. These results suggest an urgent need for investigating CVD prevention in CBT patients.

Behavioral outcome among survivors of childhood brain tumor: a case control study

BackgroundAdvances in the treatment of childhood brain tumors have significantly improved survival rates. With improved survival rates, long-term treatment-related toxicities have become important, and the resulting complications can affect patients’ emotion and behavior. This study aimed to 1) evaluate behavioral outcomes among survivors of childhood brain tumors, 2) compare behavioral outcomes among survivors of childhood brain tumors with survivors of childhood leukemia and healthy children, and 3) determine any demographic, disease, and/or treatment-related factors that could affect the behavioral outcomes of survivors of childhood brain tumors.MethodsA comparative cross-sectional study was conducted over a period of 1 year (June 1st, 2018–May 31st, 2019) in two tertiary referral centers in Kuala Lumpur, Malaysia. Thirty-eight survivors of childhood brain tumors aged 6 to 18 years old who had been off-treatment for at least 1 year and were in remission, 38 age- and gender-matched survivors of childhood leukemia who had been off-treatment for at least 1 year and were in remission, and 38 age- and gender-matched unrelated healthy children were recruited. The Child Behaviour Checklist (CBCL) parent report and Youth Self-Report (YSR) questionnaires were used to assess behavioral outcomes.ResultsSurvivors of childhood brain tumors showed statistically significantly worse behavioral outcomes than healthy children for social problems and attention problems (p < 0.05, respectively). A significantly worse outcome was found for “social problems” (p < 0.05) in survivors of childhood brain tumors compared to survivors of childhood leukemia. Significant associations were also found between physical disability, visual impairment, education level of survivors, and father’s occupation and behavioral outcomes among survivors of childhood brain tumors.ConclusionsSurvivors of childhood brain tumors in our center showed poor behavioral outcomes for social problems and attention problems. Thus, effective psychosocial support interventions tailored to individual patients as soon as treatment is completed are important to prevent potentially debilitating emotional problems.

What are the effects of exercise training in childhood cancer survivors? A systematic review.

This systematic review aimed to summarize evidence on the effects of physical exercise interventions in childhood cancer survivors (CCS) who had finished anticancer therapy ≥ 1year before the study. Relevant articles were identified in the electronic databases PubMed, Web of Science, and SPORTDiscus (from inception to June 27, 2019). The PEDro scale was used to assess methodological quality. Twelve studies including 109 CCS met all inclusion criteria and were included in the systematic review. The quality of the included studies was overall low. Physical exercise improved endothelial function, reduced waist circumference, and waist-to-hip ratio and increased physical activity levels. Preliminary evidence was found regarding benefits on brain volume and structure after exercise interventions in childhood brain tumor survivors. Only two studies reported exercise-related adverse events. Physical exercise seems to be safe and effective for improving several health markers in CCS, but further high-quality research and especially randomized controlled trials are needed to confirm these results.

Reintegration into school of childhood brain tumor survivors: a qualitative study using the International Classification of Functioning, Disability and Health – Children and Youth framework

Purpose The purpose of this study was to describe experiences of childhood brain tumor survivors (CBTS) and key figures in their environment after returning to school, using the International Classification of Functioning, Disability and Health – Children and Youth framework. Methods We conducted semi-structured interviews with five children, nine parents, 28 teachers, and 14 health professionals at three predetermined times over a two-year period. A qualitative content analysis was performed by linking meaningful units from transcribed interviews to ICF codes. Results Children experienced diverse body dysfunctions that could impede school participation, mostly related to knowledge acquisition and application, executive functioning and social life. Support at school and professional aftercare were essential to alleviate the child’s difficulties. The teacher’s attitude, parental involvement, and practices of collaboration belonging to the child’s supportive network further influenced the reintegration process. Also, child-specific factors including emotional reactions to illness, age, and balance between school life and leisure time were considered to affect school readjustment. Conclusions The International Classification of Functioning, Disability and Health framework is a useful instrument to systematically describe experiences of CBTS and key figures regarding school life, providing a common language to communicate for stakeholders in education and healthcare following the child’s return. IMPLICATIONS FOR REHABILITATION The International Classification of Functioning, Disability and Health – Children and Youth framework is a useful instrument to describe experiences regarding school life of childhood brain tumor survivors (CBTS) and key figures in their environment. The ICF-CY model can be combined with a standardized assessment of neurocognition or performance to achieve a comprehensive view of the child’s participation at school, by exploring both personal and environmental factors. Childhood brain tumor survivors mostly show impairments in acquiring and applying knowledge, executive functioning and social life, which may require increased support at school and professional aftercare. A positive attitude of school staff, high parental involvement in school life and adequate collaboration between parents, education and healthcare are important to prioritize in the context of reintegration into school of CBTS.

Systematic review: measurement properties of patient-reported outcome measures evaluated with childhood brain tumor survivors or other acquired brain injury.

Survivors of childhood brain tumors or other acquired brain injury (ABI) are at risk of poor health-related quality of life (HRQoL); its valid and reliable assessment is essential to evaluate the effect of their illness on their lives. The aim of this review was to critically appraise psychometric properties of patient-reported outcome measures (PROMs) of HRQoL for these children, to be able to make informed decisions about the most suitable PROM for use in clinical practice. We searched MEDLINE, EMBASE, and PsycINFO for studies evaluating measurement properties of HRQoL PROMs in children treated for brain tumors or other ABI. Methodological quality of relevant studies was evaluated using the consensus-based standards for the selection of health status measurement instruments checklist. Eight papers reported measurement properties of 4 questionnaires: Health Utilities Index (HUI), PedsQL Core and Brain Tumor Modules, and Child and Family Follow-up Survey (CFFS). Only the CFFS had evidence of content and structural validity. It also demonstrated good internal consistency, whereas both PedsQL modules had conflicting evidence regarding this. Conflicting evidence regarding test-retest reliability was reported for the HUI and PedsQL Core Module only. Evidence of measurement error/precision was favorable for HUI and CFFS and absent for both PedsQL modules. All 4 PROMs had some evidence of construct validity/hypothesis testing but no evidence of responsiveness to change. Valid and reliable assessment is essential to evaluate impact of ABI on young lives. However, measurement properties of PROMs evaluating HRQoL appropriate for this population require further evaluation, specifically construct validity, internal consistency, and responsiveness to change.

Metabolic response to exercise in childhood brain tumor survivors: A pilot controlled study.

Survival rates in children diagnosed with malignant brain tumors exceed 70%. A higher risk of dyslipidemia, central obesity, and insulin resistance has been reported among these children. We investigated substrate utilization during submaximal exercise. Ten brain tumor survivors and 10 healthy children were matched by sex, age, and Tanner stage. Participants completed a submaximal incremental exercise test to determine their fat and carbohydrate oxidation rates. The relative oxygen volume (VO2 ) peak was significantly higher in the control group than in the survivors of childhood brain tumors (43.3±11.9 and 32.4±10.2mL/kg /min, P=.04). At the same relative exercise intensity, there was no difference in the carbohydrate or lipid oxidation rate between the two groups, or in the maximal fat oxidation (MFO) rate, or in the heart rate or percentage of VO2 peak to reach MFO. Healthy children achieved MFO at significantly higher muscular power than did brain tumor survivors (47.9±20.8 and 21.8±9.6 W, P=.003). Because child brain tumor survivors are less physically fit than healthy children, and substrate utilization during submaximal exercise is not different, physical activity should be promoted for child brain tumor survivors.

High occurrence of sleep problems in survivors of a childhood brain tumor with neurocognitive complaints: The association with psychosocial and behavioral executive functioning.

Survivors of childhood brain tumors are prone to sleep and neurocognitive problems. Effective interventions to improve neurocognitive functioning are largely lacking. In general, sleep problems are negatively related to neurocognitive functioning, but this relationship is unclear in survivors of childhood brain tumors. Therefore, the occurrence of sleep problems, potential risk factors, and the relation between sleep and executive functioning were evaluated. Baseline data of a randomized controlled trial on the effectiveness of neurofeedback were used. Childhood brain tumor survivors 8-18 years of age with parent-reported neurocognitive complaints ≥2 years after treatment were eligible. Parents completed the Sleep Disturbance Scale for Children. Executive functioning was assessed by parents and teachers (Behavior Rating Inventory of Executive Functioning). Multiple linear regression analyses were used to examine sociodemographic and medical characteristics and emotional difficulties and hyperactivity/inattention (Strength and Difficulties Questionnaire) as potential risk factors for sleep problems, and to assess the association between sleep and executive functioning. Forty-eight percent of survivors (n=82, 7.0±3.6 years post diagnosis, age 13.8±3.2years) had sleep problems and scored significantly worse than the norm on the subscales Initiating and Maintaining Sleep, Excessive Somnolence, and the total scale (effect sizes 0.58-0.92). Emotional problems and/or hyperactivity/inattention were independent potential risk factors. Sleep problems were associated with worse parent-reported executive functioning. Sleep problems occur among half of childhood brain tumor survivors with neurocognitive problems, and are associated with worse executive functioning. Future studies should focus on the development of sleep interventions for this population, to improve sleep as well as executive functioning.

Prevalence of endocrine disorders in childhood brain tumor survivors in South Korea: A nationwide population-based, longitudinal study.

e21517 Background: Brain tumors are the most common solid cancers in child patients with a current expected 5-year overall survival rate of 73%. Also, patients with brain cancer tend to have a high rate of neuroendocrine disorders. These endocrine problems may have a significant negative effect on quality of life and treatment compliance. Methods: The aim of this longitudinal study was to analyze the prevalence of endocrine disorders in childhood brain tumor survivors using claims data in South Korea. We confirmed endocrine disorders in a nationwide cohort of 1,058 patients who were diagnosed with brain tumor between January 1, 2009 and March 29, 2016 and who survived &gt; 2 years after diagnosis. Multivariable logistic regression was used to evaluate association between demographic and treatment related variables and the prevalence of endocrine disorders. Results: After a median follow-up of 60.0 months, a total of 393 (37.1%) patients were diagnosed with at least one endocrine disorder. The median follow-up time from primary brain tumor diagnosis to diagnosis of first endocrine disorders was 26.3 months (range, 0.06 to 96.7). Of those patients, 333 childhood brain tumor survivors were diagnosed with endocrine disorders during their first 5 years after diagnosis. The overall frequency of endocrine disorders peaked during 2 months after the cancer diagnosis. The most common endocrine disorders were pituitary dysfunction (21.9%), thyroidal disease (6.1%), precocious puberty (4.6%), and growth hormone deficiency (4.3%). Female patients were at a higher risk for endocrine disorders (odds ratio: 1.45, p = 0.005). The patients with radiotherapy were more likely to have endocrine disorders compared without radiotherapy (odds ratio: 1.79, p &lt; 0.001). Conclusions: Endocrine disorders in childhood brain tumor survivors was high and showed different patterns of prevalence depending on the nature of disease and time sequence. In childhood brain tumor survivors who are risk of endocrine disorders, regular assessment of endocrine function and timely intervention were needed. [Table: see text]

The Effects of Radiation and Sex Differences on Adaptive Functioning in Adult Survivors of Pediatric Posterior Fossa Brain Tumors.

Radiation therapy (RT) improves rates of survival of patients with childhood brain tumors but increases deficits in cognition and independent living skills. Previous literature has studied difficulties in basic cognitive processes, but few explore impairment in higher-order skills such as adaptive functioning. Some studies identify females as at risk for cognitive deficits due to RT, but few investigate sex differences in adaptive functioning. It was hypothesized that females would exhibit poorer long-term independent living skills and core cognitive skills relative to males following RT. Forty-five adult survivors of posterior fossa childhood brain tumors (24 females) completed the Wechsler Abbreviated Scale of Intelligence (WASI-II), Wechsler Memory Scale, Third Edition (WMS-III) Digit Span Forward (DSF) and Backward (DSB), and Oral Symbol Digit Modalities Test (OSDMT). Informants completed the Scales of Independent Behavior-Revised (SIB-R). DSF and OSDMT were positively correlated with all five SIB-R domains, full-scale IQ (FSIQ) was positively correlated with four SIB-R domains, and DSB was positively correlated with three SIB-R domains. There was an interaction between sex and RT for OSDMT and community living skills with trend level interactions for personal living skills and broad independent living skills, where females without RT had higher scores than females with RT. Female survivors were more affected by RT than males across the community living skills domain of adaptive functioning as well as processing speed. Processing speed deficits may have a cascading impact on daily living skills. Future studies should investigate how clinical and biological factors may contribute to personalized treatment plans between sexes. (JINS, 2019, 25, 729-739).

Implications of health status, health-related quality of life, and socioeconomic outcomes in childhood brain tumor survivors.

Implications of health status, health-related quality of life, and socioeconomic outcomes in childhood brain tumor survivors.

Radiation-Induced Meningiomas After Childhood Brain Tumor: A Magnetic Resonance Imaging Screening Study.

Purpose: Childhood brain tumors (CBTs) and their treatment increase the risk of secondary neoplasms (SNs). We studied the incidence of secondary craniospinal tumors with magnetic resonance imaging (MRI) screening in a national cohort of survivors of CBT treated with radiotherapy, and we analyzed the Finnish Cancer Registry (FCR) data on SNs in survivors of CBT with radiotherapy registered as a part of the primary tumor treatment. Methods: A total of 73 survivors of CBT participated in the MRI study (mean follow-up of 19 ± 6.2 years). The incidence of SNs in a cohort of CBT patients (N = 569) was retrieved from the FCR (mean follow-up of 11 ± 12.9 years). Brain tumors were diagnosed at age ≤16 years between the years 1970 and 2008 in the clinical study and the years 1963 and 2010 in the FCR population. Results: Secondary brain tumors, meningiomas in all and schwannoma in one, were found in 6 of the 73 (8.2%) survivors with a mean of 23 ± 4.3 years after the diagnosis of the primary tumor. The cumulative incidence was 10.2% (95% confidence interval [CI] 3.9-25.1) in 25 years of follow-up. In the FCR data, the 25-year cumulative incidence of SNs was 2.4% (95% CI 1.3-4.1); only two brain tumors, no meningiomas, were registered. Conclusion: Survivors of CBT treated with radiotherapy have a high incidence of meningiomas, which are rarely registered in the FCR.

Challenges to Family Management for Caregivers of Adolescent and Young Adult Survivors of Childhood Brain Tumors [Formula: see text

Due to the complexity of cancer late effects, the education required to provide anticipatory guidance and support to the caregivers of adolescent and young adult (AYA) survivors of childhood brain tumors can be difficult. Therefore, identifying challenges to family management (FM) could be helpful in anticipating complications with the integration of tumor and treatment late effects into family life. Building on previous research that described FM for children with chronic conditions, children who survived cancer, and the Family Management Styles Framework, the purpose of this study was to identify FM challenges for caregivers of AYA survivors of childhood brain tumors to guide clinical practice and research. Directed content analysis was used to identify FM challenges in data from semistructured interviews with 45 maternal caregivers for AYA survivors of childhood brain tumors living with them. Caregivers were largely White (89%) with an average age of 52 years, educated beyond the high school level (67%), and were partnered or married (53%). On average, caregivers had been caring for the AYA for 21 years since diagnosis, and 56% of their survivors had moderate functional restrictions. A primary and a secondary analyst were assigned to the data for each interview and completed a single summary matrix. A list of challenges was created by the research team based on Family Management Styles Framework, the literature, and clinical expertise. Seven core challenges to FM were identified: ensuring survivor well-being, supporting survivor independence, encouraging sibling well-being, planning family activities, sustaining parents as caregivers, attending to survivor late effects, and providing support and advocacy.