Survivors Of Childhood Brain Tumors Research Articles

Hormone replacement in survivors of childhood cancer and brain tumors: safety and controversies.

Childhood cancer survivors are at risk for developing endocrine disorders, including deficits in growth hormone, thyroid hormone and sex hormones. The influence these hormones have on cell growth and metabolism has raised concerns regarding the safety of their use as treatments in survivors of childhood cancer and brain tumors. This manuscript offers a summary of current knowledge, controversies and areas for future research pertaining to this area.

Long term quality of life outcomes following surgical resection alone for benign paediatric intracranial tumours

Survivors of paediatric intracranial tumours are at increased risk of psychosocial, neuro-developmental, and functional impairment. This study aimed to evaluate long-term health-related quality-of-life (HRQOL) outcomes in patients with benign paediatric brain tumours treated curatively with surgical resection alone. This was a cross-sectional study of patients with benign paediatric intracranial tumours managed with surgery alone between 2000 and 2015. Eligible patients with a minimum of 5-years follow-up after surgery were identified. Validated health-related quality of life (HRQOL) questionnaires were administered: SF-36, QLQ-BN20, QLQ-C30 and PedsQL™. Twenty-three patients participated (median age at surgery 13years; range 1-18; 12 male). The most common diagnosis was pilocytic astrocytoma (n = 15). Median time from surgery to participation was 11years(range 6-19). Fourteen patients achieved A-level qualifications and two obtained an undergraduate degree. Twelve patients were employed, eight were studying and three were unemployed or volunteering. HRQOL outcomes demonstrated significant limitation from social functioning (p = 0.03) and cognitive functioning (p = 0.023) compared to the general population. Patients also experienced higher rates of loss of appetite (p = 0.009) and nausea and vomiting (p = 0.031). Ten patients were under transitional teenager and young-adult (TYA) clinic follow-up. TYA patients achieved higher levels of education (p = 0.014), were more likely to hold a driver's license (p = 0.041) compared to patients not followed-up through these services. Childhood brain-tumour survivors have a greater risk of developing psychological, neuro-cognitive and physical impairment. Early comprehensive assessment, specialist healthcare and TYA services are vital to support these patients.

Methylphenidate improves cognitive function and health-related quality of life in survivors of childhood brain tumours

ABSTRACT Objectives: The growing population of survivors of childhood brain tumors present the challenge of long-term quality of survival. The domains most affected by tumor and treatment are those implicated in development of typical intellectual functions: attention, working memory, and processing speed, with consequent effects upon function and quality of life. In this paper we present service evaluation data on the 12-month effect upon processing speed, visual and auditory attentional domains in 29 patients receiving methylphenidate aged 5-16 years (Mean=10.6). Methods: Patients received immediate-release methylphenidate and were converted to modified-release as appropriate. Mean optimal dose of immediate-release methylphenidate was 0.34 mg/kg per dose (range 0.2-0.67). Results: Patients showed a significant positive impact of methylphenidate on attention in all tests of selective visual attention from the Test of Everyday Attention for Children 2. A significant improvement was also shown on response time. Significant change was not found on psychometric measures of sustained auditory or visual attention, or selective auditory attention. Ratings of Health-Related Quality of Life showed a positive benefit of methylphenidate at 12 months. Side effects were minimal and not statistically significant. Conclusions: Survivors of childhood brain tumor with attentional and processing speed deficit show clinical benefit from methylphenidate.

Neurocognitive function and health-related quality of life in a nationwide cohort of long-term childhood brain tumor survivors.

Childhood brain tumor survivors are at high risk of late effects, especially neurocognitive impairment. Limited data are available examining neurocognitive function and associations with quality of life (QoL) in childhood brain tumor survivors. Our aim was to examine neurocognitive function in childhood brain tumor survivors, and associations with QoL and symptom burden. Five-year survivors of brain tumors over the age of 15 were identified in the Danish Childhood Cancer Registry (n = 423). Eligible and consenting participants completed neuropsychological tests and questionnaires assessing QoL, insomnia, fatigue, anxiety, and depression. Survivors treated with radiation (n = 59) were statistically compared with survivors not treated with radiation (n = 102). In total, 170 survivors participated (40.2% participation rate). Sixty-six percent of the survivors who completed neurocognitive tests (n = 161) exhibited overall neurocognitive impairment. Survivors treated with radiation, especially whole-brain irradiation, exhibited poorer neurocognitive outcomes than survivors not treated with radiation. Neurocognitive outcomes for survivors treated with surgery were below normative expectations. Furthermore, a number of survivors experienced significant fatigue (40%), anxiety (23%), insomnia (13%), and/or depression (6%). Survivors treated with radiation reported lower quality of life (QoL) and higher symptom burden scores than survivors not treated with radiation; particularly in physical functioning, and social functioning with symptoms of fatigue. Neurocognitive impairment was not associated with QoL or symptom burden. In this study, a majority of the childhood brain tumor survivors experienced neurocognitive impairment, reduced QoL, and high symptom burden. Although not associated with each other, it is apparent that childhood brain tumor survivors experience not only neurocognitive dysfunction but may also experience QoL impairments and significant symptom burden.

Working Memory and Processing Speed Predict Math Skills in Pediatric Brain Tumor Survivors.

Childhood brain tumor (BT) survivors are at risk for working memory (WM) and processing speed (PS) deficits, which impact other cognitive domains. This study aimed to characterize WM, PS, and untimed mathematics calculation performance in pediatric BT survivors at least 2 years post-diagnosis, identify medical factors associated with deficits in mathematics, and examine whether WM and/or PS predict mathematics performance in this clinical sample. Retrospective data were gathered from 72 BT survivors between 7 and 21 years of age ( M =13.64y, SD =4.01y) for a clinical neuropsychologic evaluation. All participants completed Wechsler measures of WM and PS and a measure of untimed mathematics calculation. WM, PS, and the mathematics calculation were significantly lower than the normative mean. Math scores were not correlated with any of the examined medical factors. PS was negatively correlated with the Neurological Predictor Scale and positively correlated with age at diagnosis. Both WM and PS were associated with math outcomes and accounted for 30.4% and 19.2% of the variance, respectively. The findings indicate that WM and PS contribute to mathematics performance in pediatric BT survivors. Examining mathematics performance should be a part of clinical neuropsychological evaluations. Interventions to improve mathematics performance in this population should also focus on WM and compensatory strategies for slowed PS.

Pre- and postdiagnosis growth failure, adult short stature, and untreated growth hormone deficiency in radiotherapy-treated long-term survivors of childhood brain tumor.

PurposeGrowth failure is common in radiotherapy-treated long-term survivors of pediatric brain tumors, but studies on longitudinal growth in this patient group are lacking. Here, the aim was to assess the changes in growth patterns before and after brain tumor diagnosis, the adult height, and the risk factors for compromised growth. The incidence and treatment practices of growth hormone deficiency were analyzed.MethodsA cohort of 73 survivors of childhood brain tumor (median age 27.2 years, range 16.2 to 43.8 years) was studied after a median follow-up period of 20.4 years from diagnosis (IQR 14.9 to 22.9 years). Patients were treated in five university hospitals in Finland between 1970 and 2008. Growth curves, final height, and patient- and disease-related risk factors for compromised growth during different growth periods were analyzed. Laboratory analyses for IGF-1 and IGFBP-3 were performed at the follow-up.ResultsGrowth failure was evident at diagnosis, with a mean height decline of -0.6 SDS (standard deviation score) from birth (95% CI -1.15 to -0.05). Mean height SDS decline after the diagnosis was -1.09 SDS (95%CI -1.51 to -0.66). At follow-up, 37% of the study subjects (27/73) had true short stature (height < -2 SDS). The mean height deficit corrected for target height was -1.9 SDS (95% CI -1.45 to -2.40). Growth failure was associated with the age at diagnosis, corticosteroid dose, radiotherapy modality and mean dose of irradiation in the thalamic area. Low IGF-1 level (below -2.0 SDS) was found in 32% (23/72), and untreated growth hormone deficiency in 40% (29/72) of the subjects.ConclusionLongitudinal growth impairment was common in radiotherapy-treated survivors of childhood brain tumor, resulting in compromised adult height. Loss of growth potential was evident already at diagnosis and further accelerated by the treatments. At young adulthood, unrecognized growth hormone deficiency was common.

Bone mineral density and nutrition in long-term survivors of childhood brain tumors.

Childhood cancer survivors are at risk of unwanted late effects. The primary aim of this study was to assess bone mineral density Z-scores (BMDz) in long-term survivors of childhood medulloblastoma (MB) or central nervous system supratentorial primitive neuroectodermal tumor (CNS-PNET). Secondary aims were to describe nutrient intake, vitamin D status, physical activity and explore potential risk factors for decreased BMDz. All MB and CNS-PNET survivors treated at Oslo University Hospital from 1974 to 2013 were invited to participate in a cross-sectional study. Dual-energy x-ray absorptiometry (Lunar Prodigy) assessed BMDz lumbar spine, BMDz total body, and lean body mass. Decreased BMDz was defined as a combination of low BMDz-1 to-1.99 and very low BMDz ≤-2. Lean body mass index (LMI) was calculated by dividing lean body mass by the squared height. Nutrient intake was assessed by a 3-day food record. Serum 25(OH)D was analyzed. Physical activity was reported by a questionnaire. Descriptive statistics and multivariable Cox regression analyses were applied. Fifty survivors with a median age of 25.5 years (5.5-51.9) and a median follow-up time of 19.5 years (3.2-40.5) were included. Mean BMDz lumbar spine was-0.8 (SD 1.1, 95% CI:-1.1 to-0.4), and BMDz total body was-0.6 (SD 1.1, 95% CI:-0.9 to-0.3). Decreased BMDz was detected in 48% of the lumbar spine and 34% of the total body measurements. In all, 62% had low calcium, and 69% had low vitamin D intake. 26% of participants had serum 25(OH)D<50nmol/L, and 62% reported an inactive lifestyle. Male sex, higher age at diagnosis, and lower LMI were potential risk factors for decreased BMDz. Long-term survivors of childhood MB and CNS-PNET had decreased BMDz, and risk factors were male sex, higher age at diagnosis, and lower LMI. Inadequate calcium and vitamin D intake, an inactive lifestyle, and a high prevalence of 25(OH)D≤50nmol/L were detected.

Effectiveness of internet-based behavioral activation on quality of life among young adult survivors of childhood brain tumor: a randomized controlled trial

Purpose Diagnosis and treatment of childhood brain tumor have detrimental effects on physical, neurocognitive, psychological, and social functioning that lasts into adulthood and effects quality of life (QOL). To address diminished QOL, an Internet-based behavioral activation (BA) intervention was developed. Behavioral activation aims to increase activities and behaviors likely to improve thoughts, mood, and QOL. Methods Participants included 127 young adult survivors of childhood brain tumor (SCBT) randomized into the experimental group (n= 64) or the waitlist control group (n= 63). The dependent variables included: life satisfaction, stress, and activation and were assessed with a two-way mixed analysis of variance (ANOVA). Results Results revealed a significant interaction between the intervention and time on life satisfaction, F(1, 125)=4.793, p = 0.03. There were no significant main effects over time for perceived stress and activation. Conclusions Findings offer initial evidence that BA can be delivered over the internet and that Internet-delivered BA can have a positive effect on the QOL of young adult SCBT. Internet-based BA interventions can serve as a resource for young adult SCBT who desire to boost their mood and QOL. Implications for rehabilitation Behavioral activation (BA) is aimed at increasing positively reinforcing overt behaviors that are likely to promote improved thoughts, mood, and quality of life (QOL). Results indicated study participants in the experimental group demonstrated a significant gain in life satisfaction compared to the control group after receiving the Internet-based BA intervention; and provides support that the intervention was associated with positive changes across time. Findings offer initial evidence that BA can be delivered over the internet and that Internet-delivered BA can have a positive effect on the QOL of young adult survivors of childhood brain tumor (SCBT). Internet-based BA interventions can serve as a resource for young adult SCBT who desire to boost their mood and QOL.

OTHR-22. Malignant mesothelioma (MM) as second cancer in childhood brain tumor survivors: the first child with neurofibromatosis type 2 and concurrent MM

Abstract We report two cases of malignant mesothelioma (MM) in childhood brain tumor survivors. A 40-year-old man, who was treated at the age of ten with chemotherapy and craniospinal radiotherapy (up to 54.5 Gy) for a pineal secreting non-germinomatous germ cell tumor, had persistent fever, fatigue, and weight loss. A total body CT scan revealed several pulmonary and abdominal wall nodularities. The histological examination on a biopsy diagnosed a biphasic MM. The karyotype and array-CGH of peripheral blood were normal; a FISH of tumor cells showed a breakage of locus EWSR1 (22q12.2). In this case, environmental exposure was identified (asbestos in public water tanks). His MM followed an aggressive course and resulted in death after three months. A twelve-year-old boy affected by severe neurofibromatosis type 2 (NF2) already treated with several neurosurgical exeresis and chemotherapy lines (hydroxycarbamide, bevacizumab, sirolimus), developed multiple abdominal lesions, associated with pleural effusion and weight loss. The CT scan showed the thickening of the left pleura and multiple peritoneal nodules. The cytological examination on thoracentesis diagnosed an epithelioid MM. The karyotype on paracentesis-obtained cells revealed monosomy of chromosomes 14 and 22, loss of Y, and trisomy of 6, 10, 11, 16, and 20. NGS is currently ongoing to exclude germline or somatic second hits. Importantly, no exposure to asbestos was found. Less than 2% of MMs occur below 45 years of age; it is a rare secondary tumor in cancer survivors and environmental or genetic contributing causes are often detectable on a careful investigation. Variants in NF2 are considered to be the second most common after those in BAP1. To our knowledge, this is the first child with NF2 and concurrent MM reported to date. Compared to the expected prognosis, his MM has followed a mild course, and one year after diagnosis he is still alive.

QOL-08. Visual memory and potential clinical risk factors in long-term survivors of a childhood brain tumor

A brain tumor treatment has previously been associated with long-term neurocognitive sequelae. However, clinical profiles differ between certain patient subgroups. We investigated the impact of tumor location, radiotherapy (RT), and age at diagnosis in childhood brain tumor survivors on long-term cognitive outcomes. Adult survivors (n=32) of pediatric brain tumors (n=11 infratentorial, n=21 supratentorial; 14 astrocytomas, 3 craniopharyngiomas, 2 ependymomas, 2 germinomas, 1 hemangioblastomas, 4 medulloblastomas, 6 nervus opticus gliomas) participated in this neuropsychological study (n=11 RT) (16.8-35.1 years old, >2years after treatment, mean age at diagnosis = 9.2 years, 50% male). An extensive neurocognitive test battery was used to assess intelligence scales (n=5), verbal and visual memory (n=2), and language (n=3). In order to investigate the effects of tumor location (infra- versus supratentorial), RT (yes vs. no), and age at diagnosis on the cognitive scores, a multivariate ANCOVA model was tested including the main effects and interaction between age and RT. Of all included scales, only visual memory was significantly associated with the risk factors. More specifically, patients who received RT (F=10.3, p=.004) and were younger at diagnosis (F=6.9, p=.014) scored worse on this task. Furthermore, the interaction effect between these factors was also significant (F=8.8, p=.006). These findings suggest that younger patients could be more vulnerable to the radiotoxic effects to visual memory outcomes. Tumor location (supra- vs. infratentorial) was not significantly associated with any outcome. In this study, only visual memory appeared to be associated with the risk factors of interest. Both radiotherapy and age at radiotherapy, as well as their interaction, could be risk factors for altered neurodevelopmental patterns of brain areas associated with visual memory.

RARE-07. Low resting energy expenditure is associated with clinical and radiological hypothalamic damage in children surviving a suprasellar brain tumor

INTRODUCTION: Children with suprasellar brain damage are at risk for hypothalamic dysfunction (HD). HD may lead to decreased resting energy expenditure (REE) contributing to the development of hypothalamic obesity. Decreased REE, however, is not present in all children with hypothalamic damage. Our aim was to assess which children suspect for HD have low REE, and if REE outcome can be associated with clinical severity of HD or with radiological posterior hypothalamic damage. METHODS: A retrospective cohort study was performed evaluating all children diagnosed with brain injury at risk for HD in whom REE measurement was performed. Measured REE (mREE) was compared to predicted REE (pREE) using amongst others the Schofield equation. Low REE was defined as mREE<90% compared to pREE. Radiologic hypothalamic damage was scored using Muller grading score. The mREE/pREE quotient was associated to a clinical score for HD symptoms and to radiological hypothalamic damage. RESULTS: Sixty-seven children suspected for HD (94% brain tumor diagnosis) with a mean BMI SDS of +2.3 ± 1.0 were included. Of these, 45 (67.2%) had mREE <90% compared to the pREE. Children with severe HD symptoms had a significant lower mean mREE/pREE quotient compared to children with no, mild, or moderate HD symptoms. Mean mREE/pREE quotient of children with posterior hypothalamic damage was significantly lower compared to children with no damage or with anterior damage. Tumor progression or tumor recurrence, severe clinical HD, and panhypopituitarism with DI were significant risk factors for reduced REE. CONCLUSION: Not all children suspect for HD have a low REE. Low REE is associated with clinical and radiological scores for hypothalamic damage. REE measurements in childhood brain tumor survivors may be useful to distinguish between those who may benefit from obesity treatment that increases REE from those who would be better helped using other obesity interventions.

QOL-19. Post-Intensive-Care Syndrome – pediatric (PICS-p) among patients with brain tumors – therapeutic strategies and preventive recommendations

Abstract BACKGROUND: Patients with brain tumors needed in intensive care unit (ICU) stay due to tumors symptoms or neurosurgery, experience a complex of physical, psychological, and cognitive symptoms. This constellation of signs and symptoms has been named Post-Intensive-Care Syndrome – pediatric (PICS-p). An interesting problem emerging from this research and requires a deeper exploration are the differences between PICS-p symptoms and primary tumors effects or side effects of treatment e.g. irradiation. METHOD: Psychological repeated testing and long-term observation was performed in 350 childhood brain tumor survivors (various types and localizations of tumor) to determine PICS-p symptoms. Age at psychological diagnosis ranged from 6 to 20 years. Full psychological outcome included assessment of psychophysical, cognitive and affective problems. The patients were examined using battery of neuropsychological methods and psychological interview. Analysis of medical history was also performed. The next level of the study was distinguished between PICS-p symptoms and the effects of tumor localization and oncology treatment, including radiotherapy. RESULTS: In our sample patients presents low scores in the all PICS-p domains: physical, cognitive and mental. Low level of motor skills, impairing daily living activities are characteristic. In this group lower IQ scores, poorer visual motor integration, learning disabilities, memory and language problems persisted. Typical problems in mental area are depressive symptoms, anxiety with intrusive memories, emotional and behavioural disruptions, socialization problems with significantly low level of social contacts. CONCLUSIONS: The results of this study suggest that PICS-p well describes and explains the psychosocial problems of these patients. It allows prepare an individual psychological interventions and rehabilitative program for particular patient as well as therapeutic strategies for whole group of patients. Draft recommendations for diagnosis of PICS-p specific for patients with brain tumors, risk factors and preventive procedures will be presented and discussed.

QOL-20. Post-Intensive-Care Syndrome – pediatric (PICS-p) among patients with brain tumors – therapeutic strategies and preventive recommendations

Abstract BACKGROUND: Patients with brain tumors needed in intensive care unit (ICU) stay due to tumors symptoms or neurosurgery, experience a complex of physical, psychological, and cognitive symptoms. This constellation of signs and symptoms has been named Post-Intensive-Care Syndrome – pediatric (PICS-p). An interesting problem emerging from this research and requires a deeper exploration are the differences between PICS-p symptoms and primary tumors effects or side effects of treatment e.g. irradiation. METHOD: Psychological repeated testing and long-term observation was performed in 350 childhood brain tumor survivors (various types and localizations of tumor) to determine PICS-p symptoms. Age at psychological diagnosis ranged from 6 to 20 years. Full psychological outcome included assessment of psychophysical, cognitive and affective problems. The patients were examined using battery of neuropsychological methods and psychological interview. Analysis of medical history was also performed. The next level of the study was distinguished between PICS-p symptoms and the effects of tumor localization and oncology treatment, including radiotherapy. RESULTS: In our sample patients presents low scores in the all PICS-p domains: physical, cognitive and mental. Low level of motor skills, impairing daily living activities are characteristic. In this group lower IQ scores, poorer visual motor integration, learning disabilities, memory and language problems persisted. Typical problems in mental area are depressive symptoms, anxiety with intrusive memories, emotional and behavioural disruptions, socialization problems with significantly low level of social contacts. CONCLUSIONS: The results of this study suggest that PICS-p well describes and explains the psychosocial problems of these patients. It allows prepare an individual psychological interventions and rehabilitative program for particular patient as well as therapeutic strategies for whole group of patients. Draft recommendations for diagnosis of PICS-p specific for patients with brain tumors, risk factors and preventive procedures will be presented and discussed.

Better detection of reduced motor functioning in brain tumor survivors based on objective motor assessments: an incentive for improved standardized follow-up

Long-term sequelae are well-known in childhood brain tumor survivors, but motor functioning remains poorly described. This cross-sectional study aimed to assess objective motor functioning, patient-specific risk factors, and parental perceptions. Fifty-two childhood brain tumor patients (pilocytic astrocytoma, medulloblastoma, and other types) who were at least 6 months out of treatment were evaluated. Mean age at testing was 11.7 years. Objective motor functioning was assessed with the Movement Assessment Battery for Children (MABC-2-NL) and/or Bruininks-Oseretsky test of motor proficiency (BOT-2). Functional walking capacity was assessed with the 6-min walk test (6MWT). Parent-reported motor functioning was addressed using the ABILHAND-Kids, ABILOCO-Kids questionnaires, and a standardized anamnesis. Patients showed impaired motor functioning in all domains (p < 0.001). Regarding risk factors, younger age at diagnosis (< 5 year) was significantly associated with lower scores on body coordination (p = 0.006). Adjuvant treatment resulted in lower scores for fine manual control of the BOT-2 (p = 0.024) and balance of MABC-2-NL (p = 0.036). Finally, questionnaires revealed an underestimation of motor problems as perceived by the parents. In conclusion, many children who are in follow-up for a brain tumor show impaired motor functioning on multiple aspects, with younger age at diagnosis and adjuvant treatment as specific risk factors. Based on the questionnaires and anamnesis, motor problems appear to be underestimated by the parents. Conclusion: These findings point to the need for timely prospective screening of motor functioning. Based on a screening assessment, adequate rehabilitation programs can be applied in childhood brain tumor survivors, aiming to reduce the adverse impact on their daily lives, both for functional activities and cardiovascular fitness.What is Known:• A pediatric brain tumor and its treatment are associated with potential long-term motor sequelae.• Test assessments could enable us to objectify motor functioning of these patients.What is New:• Pediatric brain tumors survivors show lower motor performance compared to the norm, which is often underestimated by parents.• Younger age at diagnosis and adjuvant treatment could be specific risk factors.

Health-related quality of life in long-term survivors of childhood brain tumors: a population-based cohort study

PurposeSurvivors of childhood brain tumors (BT) are at high risk for long-term physical and psychological sequelae. Still, knowledge about health-related quality of life (HRQL) and associated factors in this population is sparse. This study investigated HRQL and its predictors in long-term survivors of childhood BT.MethodsSurvivors of childhood BT (mean age = 28.1 years, SD = 6.8, n = 60) underwent clinical examination and neurocognitive examination, and completed self-rating questionnaires assessing HRQL (RAND-36) and depressive symptoms (Beck Depression Inventory-II). Socio-demographic information was gathered via a questionnaire. Tumor- and treatment-related information was collected from medical records. Control group data were collected from age-matched controls (n = 146) without a history of cancer, randomly selected from the local population registry. Multiple linear regression models were used to investigate predictors of HRQL; separate models were fitted for each domain of the RAND-36.ResultsMale survivors (mean age = 27.0, SD = 6.0, n = 39) reported significantly lower HRQL than male controls in the domains of physical functioning, general health, vitality, social functioning, and role limitations-emotional. Female survivors (mean age = 30.2 years, SD = 7.6, n = 21) reported comparable levels as female controls in all domains except physical functioning. A higher burden of late effects, not working/studying, being diagnosed with BT during adolescence, and reporting current depressive symptoms were significant predictors of lower HRQL.ConclusionOur results highlight that male survivors of childhood BT are at particular risk of impaired HRQL. Also, results point to the close relation between symptoms of depression and impaired HRQL in survivors of childhood BT which should be acknowledged by long-term follow-up care.

Development of Training in Problem Solving for Caregivers of Childhood Brain Tumor Survivors.

Family management (FM) challenges of maternal caregivers of young adult survivors of childhood brain tumors are well documented, but there are no evidence-based caregiver interventions to improve FM. The aims of this study were to (1) generate the knowledge necessary for developing a caregiver intervention (stage 0) and (2) modify an existing, efficacious intervention by engaging stakeholders (stage 1). Stages 0 and 1 of the National Institutes of Health Stage Model for Behavioral Intervention Development and the FM Styles Framework were used in this study. In stage 0, families with condition-focused FM patterns were identified as at risk for poor problem solving. The 12-item Condition Management Ability scale of the FM Measures was selected as the screener to identify condition-focused maternal caregivers. Problem solving was identified as a potential mechanism for promoting behavior change. In stage 1, Bright IDEAS for Everyday Living was modified by integrating the FM Styles Framework creating Training in Problem Solving for Caregivers of Young Adult Survivors of Childhood Brain Tumors. Qualitative and quantitative assessments of feasibility and acceptability by maternal caregivers were excellent and used to improve selected areas of concern. Feedback from stakeholders indicates that Training in Problem Solving is a promising approach to shifting FM patterns and improving the functioning of caregivers, young adult survivors, and families. When developing interventions, the use of systemic methods can provide both clinically based and scientifically acceptable solutions. Those interventions based on both problem solving and FM are potentially promising but need further testing.

QOLP-30. LONG-TERM UNMET SUPPORTIVE CARE NEEDS OF TYA CHILDHOOD BRAIN TUMOUR SURVIVORS AND THEIR CAREGIVERS: A CROSS-SECTIONAL SURVEY

Abstract INTRODUCTION The supportive care needs of long-term childhood brain tumour survivors, now teenagers and young adults (TYAs), and their caregivers are largely unknown. TYAs are a unique patient cohort with specific challenges and vulnerabilities differing from children or older adults. We aimed to describe their supportive care needs and explore associations between needs and quality of life (QoL).This is the first study to collect quantitative data about needs in this survivorship group. METHODS Participants were recruited from long-term follow-up clinics (in three National Health Service Trusts in England) and online. Participants included childhood brain tumour survivors, ≥ 5 years from diagnosis, currently aged 13-30, and their primary caregivers. Survivors completed the Supportive Care Needs Survey (SCNS) Short-Form and Paediatric Functional Assessment of Cancer Therapy – Brain (Peds-FACT-Br). While caregivers completed the SCNS-Partners and Caregivers (SCNS-P&amp;C) and the Caregiver Quality of Life Index–Cancer (CQOLC). RESULTS In total, 112 individuals (69 survivors/43 caregivers) participated. Survivors reported on average 9.4 (±8.5) unmet needs. Needs were greatest in the psychological domain, with anxiety (60.3%), uncertainty about the future (50.7%) and feeling down and depressed (48.5%) most commonly reported. Caregivers reported on average 12.4 (±12.3) unmet needs. Again, the greatest number of unmet needs were observed in the psychological domain. Many caregivers also reported information needs around financial support/government benefits (42.9%) and possible survivor fertility problems (42.9%). Multivariable linear regression analysis showed that female survivors, unemployed survivors, survivors further away from diagnosis, and single caregivers were more likely to report unmet needs. More unmet needs were significantly associated with poorer QoL in survivors and caregivers. CONCLUSION This research provides leads to improving supportive care and long-term follow-up services. Psychological support appears to be the biggest gap in care. Understanding unmet needs and recognising what services are required is critical to improving quality of long-term survival.

Long-term unmet supportive care needs of teenage and young adult (TYA) childhood brain tumour survivors and their caregivers: a cross-sectional survey

IntroductionThe supportive care needs of long-term childhood brain tumour survivors, now teenagers and young adults (TYAs), and their caregivers are largely unknown. We aimed to describe their supportive care needs and explore associations between needs and quality of life (QoL).MethodsParticipants were recruited from long-term follow-up clinics (in three NHS Trusts in England) and online. Participants included childhood brain tumour survivors, ≥ 5 years from diagnosis, currently aged 13–30, and their primary caregivers. Survivors completed the Supportive Care Needs Survey (SCNS) Short Form and caregivers the SCNS-Partners & Caregivers, alongside validated QoL questionnaires (Peds-FACT-Br and CQOLC).ResultsIn total, 112 individuals (69 survivors/43 caregivers) participated. Survivors reported on average 9.4 (± 8.5) unmet needs. Needs were greatest in the psychological domain, with anxiety (60.3%), uncertainty about the future (50.7%) and feeling down and depressed (48.5%) most commonly reported. Caregivers reported on average 12.4 (± 12.3) unmet needs. Again, the greatest number of unmet needs was observed in the psychological domain. Many caregivers also reported information needs around financial support/government benefits (42.9%) and possible survivor fertility problems (42.9%). Multivariable linear regression analysis showed that female survivors, unemployed survivors, survivors further away from diagnosis and single caregivers were more likely to report unmet needs. More unmet needs were significantly associated with poorer QoL in survivors and caregivers.ConclusionThis research provides leads to improving supportive care and long-term follow-up services. Psychological support represents the biggest gap in care. Understanding unmet needs and recognising what services are required are critical to improving quality of long-term survival.

Prevalence and risk factors of hypothalamic-pituitary dysfunction in infant and toddler childhood brain tumor survivors.

Childhood brain tumor survivors (CBTS) are at risk to develop hypothalamic-pituitary (HP) dysfunction (HPD). The risk for HPD may vary between different age groups due to maturation of the brain and differences in oncologic treatment protocols. Specific studies on HPD in infant brain tumor survivors (infant-BTS, 0-1 years at diagnosis) or toddler brain tumor survivors (toddler-BTS, ≥1-3 years) have not been performed. A retrospective nationwide cohort study in CBTS was performed. Prevalence and risk factors for HPD were compared between infant-, toddler-, and older-BTS. Subgroup analysis was performed for all non-irradiated CBTS (n = 460). In total, 718 CBTS were included, with a median follow-up time of 7.9 years. Overall, despite the less frequent use of radiotherapy (RT) in infants, no differences in the prevalence of HPD were found between the three groups. RT (OR: 16.44; 95% CI: 8.93-30.27), suprasellar tumor location (OR: 44.76; 95% CI: 19.00-105.49), and younger age (OR: 1.11; 95% CI: 1.05-1.18) were associated with HP dysfunction. Infant-BTS and toddler-BTS showed more weight gain (P < 0.0001) and smaller height SDS (P = 0.001) during follow-up. In non-irradiated CBTS, infant-BTS and toddler-BTS were significantly more frequently diagnosed with TSH-, ACTH-, and ADH deficiency, compared to older-BTS. Infant and toddler brain tumor survivors seem to be more vulnerable to develop HP dysfunction than older children. These results emphasize the importance of special infant and toddler brain tumor treatment protocols and the need for endocrine surveillance in children treated for a brain tumor at a young age.

Late effects care for childhood brain Tumor Survivors: A Quality-Improvement Initiative

Childhood and adolescent brain tumor survivors are at risk for long-term consequences of therapy. We reviewed adherence to long-term follow-up (LTFU) guidelines, assessed provider perspectives, and studied the needs, experience and quality of life (QOL) of pediatric malignant brain tumor survivors in the McMaster Children’s Hospital Neuro-Oncology clinic. LTFU areas for improvement were evaluated using an anonymous health provider needs assessment questionnaire. The Cancer Care Experience Questionnaire (CCEQ), Cancer Worry Scale (CWS), Self-Management Skills Scale (SMSS), and PedsQL measured parents/patients’ needs and QOL. Individual care plans were based on the Children’s Oncology Group (COG) LTFU guidelines. Based on 17 responses, staff perceived areas for improvement included: increased multi-disciplinary participation, improved patient education and increased surveillance for therapy-related late effects. Thirty-two families participated, most felt they received high-quality care. Mean cancer worry scores were low (71.8 (± 28.4)). Survivors reported limited self-management skills (58.5 (±18.2)), requiring support with medical needs and activities of daily living. Overall median QOL scores were ‘good’ (parental report 72.3 (±17.7), survivor 68.2 (±16.6)). Utilizing survivorship guidelines and assessments from patients, caregivers and health providers, we implemented improvements in our provision of neuro-oncology survivorship care. Lessons learned may assist other LTFU programs.