Trajectories of illness perceptions in paediatric cancer patients and their parents and associations with health-related quality of life: Results of a prospective-longitudinal study.

OBJECTIVE: In paediatric oncology, little is known about trajectories of illness perceptions and their longitudinal associations with health-related quality of life (HRQoL). Therefore, the aim of this study was to investigate changes in illness perceptions in children and parents over a one-year-period and to investigate predictive value of child's and parent's illness perceptions during acute treatment for child's HRQoL 1 year later. METHODS: N = 65 child-parent-dyads participated in a longitudinal study (retention rate: 80.2%). Children were 4-18 years of age and underwent acute cancer treatment at baseline. Children and parents reported on their own illness perceptions (Illness-Perception-Questionnaire-Revised), as well as on the child's HRQoL (KINDL-R) at baseline and one-year-follow-up. Paired-samples t-tests were calculated to investigate changes over time. A hierarchical multiple regression analysis was performed to investigate predictive value of child's and parent's illness perceptions for child's HRQoL. RESULTS: Child's HRQoL t(63) = -6.73, p < 0.001, their perceptions of coherence (i.e. understanding; t(54) = -2.36, p = 0.022) and consequences of their illness (t(54) = 2.86, p = 0.006), and parent's perception of cyclical trajectory (t(61) = 2.06, p = 0.044) improved from baseline to 1-year-follow-up. All other illness perceptions remained stable. Exploratory post-hoc analyses showed differences in the pattern of change in age-, gender-, and diagnosis-specific subgroups. After controlling for baseline levels of HRQoL, child's perceptions of symptoms and consequences were independent predictors of their HRQoL 1 year later (R2 = 0.396, F(2,52) = 10.782, p < 0.001), whereas no parent's illness perceptions added predictive value. CONCLUSION: In paediatrics, child's and parent's illness perceptions should be assessed. Our findings highlight the importance of illness perceptions as potential modifiable variables in interventions to improve child's HRQoL.

Bridging the gap: A quality improvement project to implement psychosocial care standards into clinical practice in pediatric oncology.

BACKGROUND: Psychosocial guidelines and standards systematically describe stressors and resources in particularly challenging situations and hence serve as a basis for interventions to achieve defined psychosocial goals. Despite fundamental principles and guidelines for psychosocial methods, the quality of provided care varies considerably depending on setting, provision, and profession. The purpose of the present protocol is to illustrate the development and evaluation of the standardized psychological intervention "My Logbook", a practical guide accompanying children through all stages of treatment by directly translating current quality standards of psychosocial care into practice. METHODS: In an evidence-based set-up, using face-to-face discussions and telephone conferences, a multi-professional team of local experts decide on critical disease-related issues, structure, content (information and intervention elements) and design of the quality improvement tool. Via delphi surveys an extended expert team is asked to rate the content, method, and design of all booklets which is concluded by a final agreement by the specialist group for quality assurance of the psychosocial working group in the Society for Pediatric Oncology (PSAPOH). The developed tools are piloted in an international multicenter study to evaluate the patient-reported outcome and feasibility and to integrate practical views of patients, as well as psychosocial and interdisciplinary professionals into the further development of the "My Logbook". DISCUSSION: The iterative development of the "My Logbook" including local and international experts as well as the patient and practical perspective allow for the design of a process-oriented, consensus - and evidence-based tool directly translating the S3-Guideline into clinical practice. Feasibility and applicability are fostered through an iterative process of constant evaluation and adaptation of the tool by international experts and through the clinical experience gathered in the multi-centered pilot study. Furthermore, the systematic evaluation of the tool by patients, psychosocial, and interdisciplinary professionals enables the identification of persisting gaps between evidence-based standards and clinical practice, discrepancies between the various stakeholders' perspectives as well as regional differences in feasibility, thereby directly linking practice and research. The preliminary results emphasize that psychological support can be standardized, enabling an evaluation and optimization of psychosocial care which future studies need to assess in multicenter clinical randomized controlled trials.

Initial Evidence for Positive Effects of a Psychological Preparation Program for MRI "iMReady" in Children with Neurofibromatosis Type I and Brain Tumors-How to Meet the Patients' Needs Best.

To provide an effective alternative to sedation during MRI examinations in pediatric cancer and NF1 patients, the aims of the present study were to (1) exploratively evaluate a behavioral MRI training program, to (2) investigate potential moderators, as well as to (3) assess the patients' well-being over the course of the intervention. A total of n = 87 patients of the neuro-oncology unit (mean age: 6.83 years) underwent a two-step MRI preparation program, including training inside the scanner, and were recorded using a process-oriented screening. In addition to the retrospective analysis of all data, a subset of 17 patients were also analyzed prospectively. Overall, 80% of the children receiving MRI preparation underwent the MRI scan without sedation, making the success rate almost five times higher than that of a group of 18 children that opted out of the training program. Memory, attentional difficulties, and hyperactivity were significant neuropsychological moderators for successful scanning. The training was associated with favorable psychological well-being. These findings suggest that our MRI preparation could present an alternative to sedation of young patients undergoing MRI examinations as well as a promising tool for improving patients' treatment-related well-being.

Illness perceptions, fear of progression and health-related quality of life during acute treatment and follow-up care in paediatric cancer patients and their parents: a cross-sectional study.

BACKGROUND: This study examines the role of illness perceptions and fear of progression (FoP) in paediatric cancer patients and their parents for patient's health-related quality of life (HRQoL), controlling for sociodemographic and medical variables. 4-18-year-old patients in acute treatment or follow-up care and one parent were examined. METHODS: N = 46 patient-parent dyads in acute treatment and n = 84 dyads in follow-up care completed measures on illness perceptions (Illness-Perceptions-Questionnaire for 12-18-year-old patients and parents or as age-adapted puppet interview for 4-11-year-old patients) and FoP (Fear-of-Progression-Questionnaire for 7-18-year-old patients and parents). Patients also completed the KINDL-R to measure HRQoL. Hierarchical multiple regression analyses were calculated. RESULTS: In acute treatment, patient's perceptions of symptoms and cyclicity of their illness explained variation in their HRQoL in addition to sociodemographic and medical variables. In follow-up care, patient's FoP and parent's perception of consequences explained additional variation in patient's HRQoL. Overall, sociodemographic and medical variables explained less variation in HRQoL in follow-up care than in acute treatment. CONCLUSIONS: Our results stress the importance of psychological factors for the well-being of paediatric cancer patients, particularly in follow-up care, where sociodemographic and medical variables play a lesser role. We recommend screening for illness perceptions and FoP during and after acute treatment to support patients and parents. Furthermore, standardized interventions focussed on changing maladaptive illness perceptions should be developed and evaluated. As parents' perceptions, thoughts, and feelings may also play an important role for the well-being of the patients, interventions should be family-focussed and include parents. Trial registration The study has been pre-registered at the German Clinical Trials Register (registered 30/06/2020; DRKS00022034) and at the Open Science Framework ( https://osf.io/3uwrx ).

A Psychosocial Support Program for Young Adult Childhood Cancer Survivors in Austria: a Qualitative Evaluation Study.

Many adolescents and young adult (AYAs) childhood cancer survivors face disease- or therapy-related late-effects, which limit their participation in various areas of daily life. AYAs are often left alone in our health care system, and many worry about their ability to cope with long-term sequelae, and some are even lost to follow-up. Therefore, in the present study, a targeted aftercare program was developed and evaluated with the goal of facilitating three important "life skills": (1) self-perception, (2) social interaction and conflict management, and (3) self-conscious communication of support needs. A total of n = 13 participants (19.2-30.2 years, mean age 22.8 years) completed a 3-day aftercare seminar, at the end of which each participant wrote a reflection letter ("letter to my future self"), elaborating on observed effects of the seminar, applicability of the given information in daily life, and the direct impact of the seminar on their individual circumstances. The reflection letters were analyzed using qualitative content analysis. All target life skills were mentioned in the reflection letters. The participants reported individual benefits from the program especially with respect to self-perception and self-confidence, giving and taking feedback, and acceptance of personal strengths and weaknesses. Moreover, the feeling of "not being alone" was associated with the survivors' experience of emotional and social support. This evaluation highlights the potential of a one weekend aftercare seminar to address important life skills that are known to positively influence health behavior in AYAs. The detailed description of the seminar can serve as a basis for making this kind of aftercare accessible for other people in similar circumstances.

Sociocultural variables have a major impact on participation in patients treated for paediatric posterior fossa tumours.

BACKGROUND: This study investigates the severeness of participation barriers in patients surviving paediatric posterior fossa tumours (PFT) many years after treatment. In the past, aetiological studies on adverse outcome have primarily focused on pathobiological risk factors. The current analysis aims to investigate the importance of environmental variables. METHODS: On average, 10 years after diagnosis, educational and social difficulties of 42 patients surviving paediatric PFT (mean age 17 years) were inquired using a self-constructed questionnaire following the Psychosocial Resource-Orientated Assessment (PREDI). Educational or social participation barriers were defined by self- and/or proxy-reported difficulties in school or in peer relationships. Accordingly, the children and adolescents were categorized into groups of adequate and limited participation. Subsequently, the study identified potential pathobiological (treatment type, hydrocephalus, tumour relapse, gender, age at diagnosis, seizures and cerebellar mutism) and environmental (parental and maternal education, siblings, main language, discrepancies between personal and environmental values, regular physical activity and private living space) risk factors and investigated whether patients with adequate and limited participation differed in the number of risk factors. RESULTS: Almost one decade after treatment, two thirds of patients experienced educational and/or social difficulties. Patients with limited participation were more frequently associated with environmental factors such as low maternal education degree, siblings, main language other than German, discrepancies between societal and personal values and irregular physical activity, as well as the pathobiological risk factors treatment type, hydrocephalus, tumour relapse, cerebellar mutism and seizures. These variables significantly discriminated between patients with and without limited participation. CONCLUSIONS: Limited participation in patients treated for paediatric PFT is common. Next to pathobiological, also environmental risk factors play a major role in educational and social participation barriers. This highlights the fact that solely considering pathobiology is not sufficient when investigating risk factors for the emergence of late sequelae. Future aetiological studies must adopt a biopsychosocial perspective.

Evaluating the diagnostic validity of the cerebellar cognitive affective syndrome (CCAS) in pediatric posterior fossa tumor patients.

Background: The aim of this case-control study was to investigate the severity of the cerebellar cognitive affective syndrome (CCAS) in patients treated for pediatric posterior fossa tumors (PFT) and evaluate its diagnostic validity and predictive value for long-term effects. Methods: Using neuropsychological test data from 56 patients with PFT (average age: 14 years), the severity of deficits in the CCAS core areas (executive functions, verbal functions, visuospatial abilities and emotions/behaviour) was examined. Neuropsychological and academic long-term outcomes of patients with CCAS were compared to two control groups of PFT patients (treated with either surgery or surgery followed by radio-/chemotherapy) without the syndrome. Risk factors associated with various deficits were considered. Results: All but one PFT patient suffered from slight to severe impairments in at least one CCAS domain, while complete CCAS occurred in 35.7%. Seven years after tumor diagnosis CCAS patients performed worse in information processing, logical reasoning, verbal functions, visuospatial skills, and executive functioning and required more special educational support compared to the control groups. CCAS patients performed equally poor as patients treated with chemo-/radiotherapy in tasks measuring information processing speed. Risk factors were significantly associated with deficits in information processing speed but not CCAS emergence. Conclusions: Deficits in the core CCAS domains are commonly found in PFT patients, but varying in severity, which suggests the syndrome to be continuous rather than dichotomous. However, the validity of CCAS diagnosis was low and unspecific. The exclusion of relevant functions typically impaired in PFT patients (eg, information processing) resulted in difficulties being overlooked.

Impact of childhood cerebellar tumor surgery on cognition revealed by precuneus hyperconnectivity.

Background: Childhood cerebellar pilocytic astrocytomas harbor excellent overall survival rates after surgical resection, but the patients may exhibit specific cognitive and behavioral problems. Functional MRI has catalyzed insights into brain functional systems and has already been linked with the neuropsychological performance. We aimed to exploit the question of whether resting-state functional MRI can be used as a biomarker for the cognitive outcome assessment of these patients. Methods: We investigated 13 patients (median age 22.0 years; range 14.9-31.3) after a median interval between surgery and examination of 15.0 years (range 4.2-20.5) and 16 matched controls. All subjects underwent functional 3-Tesla MRI scans in a resting-state condition and battery neuropsychological tests. Results: Patients showed a significantly increased functional connectivity in the precuneus compared with controls (P < .05) and at the same time impairments in various domains of neuropsychological functioning such as a lower mean Wechsler Intelligenztest für Erwachsene (WIE) IQ percentile (mean [M] = 48.62, SD = 29.14), lower scores in the Trail Making Test (TMT) letter sequencing (M = 49.54, SD = 30.66), worse performance on the WIE subtest Digit Symbol Coding (M = 38.92, SD = 35.29), subtest Symbol Search (M = 40.75, SD = 35.28), and test battery for attentional performance (TAP) divided attention task (M = 783.92, SD = 73.20). Conclusion: Childhood cerebellar tumor treated by resection only strongly impacts the development of precuneus/posterior cingulate cortex functional connectivity. Functional MRI has the potential to help deciphering the pathophysiology of cerebellar-related cognitive impairments in these patients and could be an additional tool in their individual assessment and follow-up.

Illness perceptions in patients and parents in paediatric oncology during acute treatment and follow-up care.

OBJECTIVE: Recent evidence suggests that illness perceptions in paediatric patients and their parents may differ, with parents holding more negative views compared to their children. Little is known about illness perceptions of very young patients and their parents. This study investigates illness perceptions in paediatric cancer patients aged 4-18 years and their parents in acute treatment or follow-up care, distinguishing patients by age (4-11, 12-18) and stage of medical treatment. METHODS: N = 45 patient-parent dyads in acute treatment and n = 95 dyads in follow-up care were examined. Parents and older children aged 12-18 years completed the Illness Perception Questionnaire-Revised (IPQ-R) and younger children aged 4-11 years were examined using an age-adapted hand puppet interview containing the IPQ-R questions. Difference scores of illness perceptions (symptoms, timeline-acute/chronic, timeline-cyclical, personal control, illness coherence, consequences, emotional representations) between children and parents were tested for significance using Wilcoxon signed-rank tests. RESULTS: Overall, parents perceived more symptoms associated with their child's illness/treatment than the children themselves. In acute treatment, younger children indicated more negative and older children more positive views regarding chronicity than parents. Younger children held less negative views on consequences, and all children reported less negative emotional representations than parents. In follow-up care, all children held less negative views on consequences and emotional representations. Older children reported less negative views on chronicity, cyclicity and illness coherence. CONCLUSION: Differences in illness perceptions of paediatric patients and their parents should be considered during and after treatment/medication and psychosocial care to support illness coping in person- and family-centred interventions.

Specific Cognitive Changes due to Hippocalcin Alterations? A Novel Familial Homozygous Hippocalcin Variant Associated with Inherited Dystonia and Altered Cognition.

BACKGROUND: Recent research suggested an hippocalcin (HPCA)-related form of DYT2-like autosomal recessive dystonia. Two reports highlight a broad spectrum of the clinical phenotype. Here, we describe a novel HPCA gene variant in a pediatric patient and two affected relatives. METHODS: Whole exome sequencing was applied after a thorough clinical and neurological examination of the index patient and her family members. Results of neuropsychological testing were analyzed. RESULTS: Whole exome sequencing revealed a novel homozygous missense variant in the HPCA gene [c.182C>T p.(Ala61Val)] in our pediatric patient and the two affected family members. Clinically, the cases presented with dystonia, dysarthria, and jerky movements. We observed a particular cognitive profile with executive dysfunctions in our patient, which corresponds to the cognitive deficits that have been observed in the patients previously described. CONCLUSION: We present a novel genetic variant of the HPCA gene associated with autosomal recessive dystonia in a child with childhood-onset dystonia supporting its clinical features. Furthermore, we propose specific HPCA-related cognitive changes in homozygous carriers, underlining the importance of undertaking a systematic assessment of cognition in HPCA-related dystonia.

Potential Importance of Early Focal Radiotherapy Following Gross Total Resection for Long-Term Survival in Children With Embryonal Tumors With Multilayered Rosettes.

Embryonal tumor with multilayered rosettes (ETMR) is a rare, aggressive embryonal central nervous system tumor characterized by LIN28A expression and alterations in the C19MC locus. ETMRs predominantly occur in young children, have a dismal prognosis, and no definitive treatment guidelines have been established. We report on nine consecutive patients and review the role of initiation/timing of radiotherapy on survival. Between 2006 and 2018, nine patients were diagnosed with ETMR. Diagnosis was confirmed histopathologically, immunohistochemically and molecularly. Median age was 25 months (5-38). Location was supratentorial in five, pineal in three, and brainstem in one. Seven patients had a gross total resection, one a partial resection and one a biopsy at initial diagnosis. Chemotherapy augmented with intrathecal therapy started a median of 10 days (7-20) after surgery. Only two patients who after gross total resection received radiotherapy very early on (six weeks after diagnosis) are alive and in complete remission 56 and 50 months after diagnosis. All remaining patients for whom radiotherapy was deferred until the end of chemotherapy recurred, albeit none with leptomeningeal disease. A literature research identified 228 patients with ETMR. Including our patients only 26 (11%) of 237 patients survived >36 months with no evidence of disease at last follow-up. All but two long-term (>36 months) survivors received radiotherapy, ten of whom early on following gross total resection (GTR). GTR followed by early focal radiotherapy and intrathecal therapy to prevent leptomeningeal disease are potentially important to improve survival of ETMR in the absence of effective targeted therapies.

Psychometric properties of the Fear of Progression Questionnaire for parents of children with cancer (FoP-Q-SF/PR).

OBJECTIVE: Psychometric properties of the Fear of Progression Questionnaire - Short Form (FoP-Q-SF) were shown to be good in samples of adult cancer patients and their partners but have so far not been investigated in parents of children with cancer. This study therefore aimed to examine psychometric properties of the previously adapted parent version of the Fear of Progression Questionnaire (FoP-Q-SF/PR) in pediatric oncology. METHODS: N=181 parents (119 mothers, 62 fathers) of n=128 children with diverse cancer entities, up to ten years after diagnosis were recruited at six hospitals and six registered parent associations in Germany and Austria between 06/2015 and 05/2016 (cross-sectional design). Parents provided medical information about their child and completed standardized questionnaires (Hospital Anxiety and Depression Scale, HADS; State-Trait Anxiety Inventory, STAI; Impact of Event Scale-Revised, IES-R; Ulm Quality of Life Inventory for Parents, ULQIE; Giessen Physical Complaints Inventory for children and adolescents, GBB-KJ). RESULTS: Exploratory factor analysis yielded two factors (50.2% explained variance) and internal consistency was good (Cronbach's α=0.89). Significant medium to large correlations of the FoP-Q-SF/PR were observed with anxiety (HADS: r=0.68; STAI: r=0.60-0.61), depression (HADS: r=0.58), posttraumatic stress (IES-R: r=0.42-0.64) and quality of life (ULQIE: r=-0.59). The FoP-Q-SF/PR discriminated between sub-groups, e.g. parents with and without clinical anxiety levels (Cohen's d=1.26). CONCLUSION: The FoP-Q-SF/PR demonstrated good reliability and validity for parents of children with cancer. The FoP-Q-SF/PR is a feasible screening instrument, which is suitable for the assessment of parental FoP in pediatric oncology.

Cerebellar pilocytic astrocytoma in childhood: Investigating the long-term impact of surgery on cognitive performance and functional outcome.

OBJECTIVE: Previous studies differ regarding the long-term effects of surgically removed pediatric cerebellar pilocytic astrocytomas (CPA). Thus, the aim of this study was to investigate the long-term impact on neurocognitive and functional outcome and to analyze age as an influencing factor. METHODS: Fourteen CPA patients were compared to the age norm and to a group of 14 high-achieving peers regarding cognitive functioning, health-related quality of life (HRQoL), and stress regulation. Mean follow-up time after diagnosis was 13.29 years (range: 3-21 years). RESULTS: Patients showed satisfactory academic achievement and did not differ from the norm except for the bodily dimension of HRQoL. However, there were marked differences in specific neurocognitive functions between patients and high achievers. Age at diagnosis did not influence neurocognitive outcome. CONCLUSION: CPA patients treated with surgery only seem to have a favorable long-term outcome, yet, in comparison with high achievers specific cognitive impairments become apparent.

Do we still need IQ-scores? Misleading interpretations of neurocognitive outcome in pediatric patients with medulloblastoma: a retrospective study.

Over the past decades, many studies used global outcome measures like the IQ when reporting cognitive outcome of pediatric brain tumor patients, assuming that intelligence is a singular and homogeneous construct. In contrast, especially in clinical neuropsychology, the assessment and interpretation of distinct neurocognitive domains emerged as standard. By definition, the full scale IQ (FIQ) is a score attempting to measure intelligence. It is established by calculating the average performance of a number of subtests. Therefore, FIQ depends on the subtests that are used and the influence neurocognitive functions have on these performances. Consequently, the present study investigated the impact of neuropsychological domains on the singular "g-factor" concept and analysed the consequences for interpretation of clinical outcome. The sample consisted of 37 pediatric patients with medulloblastoma, assessed 0-3 years after diagnosis with the Wechsler Intelligence Scales. Information processing speed and visuomotor function were measured by the Trailmaking Test, Form A. Our findings indicate that FIQ was considerably impacted by processing speed and visuomotor coordination, which leaded to an underestimation of the general cognitive performance of many patients. One year after diagnosis, when patients showed the largest norm-deviation, this effect seemed to be at its peak. As already recommended in international guidelines, a comprehensive neuropsychological test battery is necessary to fully understand cognitive outcome. If IQ-tests are used, a detailed subtest analysis with respect to the impact of processing speed seems essential. Otherwise patients may be at risk for wrong decision making, especially in educational guidance.

Neuronal correlates of cognitive function in patients with childhood cerebellar tumor lesions.

While it has been shown that cerebellar tumor lesions have an impact on cognitive functions, the extent to which they shape distant neuronal pathways is still largely undescribed. Thus, the present neuroimaging study was designed to investigate different aspects of cognitive function and their neuronal correlates in patients after childhood cerebellar tumor surgery. An alertness task, a working memory task and an incompatibility task were performed by 11 patients after childhood cerebellar tumor surgery and 17 healthy controls. Neuronal correlates as reflected by alterations in functional networks during tasks were assessed using group independent component analysis. We were able to identify eight networks involved during task performance: default mode network, precuneus, anterior salience network, executive control network, visual network, auditory and sensorimotor network and a cerebellar network. For the most 'basic' cognitive tasks, a weaker task-modulation of default mode network, left executive control network and the cerebellar network was observed in patients compared to controls. Results for higher-order tasks are in line with a partial restoration of networks responsible for higher-order task execution. Our results provide tentative evidence that the synchronicity of brain activity in patients was at least partially restored in the course of neuroplastic reorganization, particularly for networks related to higher-order cognitive processes. The complex activation patterns underline the importance of testing several cognitive functions to assess the specificity of cognitive deficits and neuronal reorganization processes after brain lesions.

Applying the International Classification of Functioning-Children and Youth Version to Pediatric Neuro-oncology.

Given the increased survival rates in patients with pediatric central nervous system tumors, late effects such as treatment- and/or illness-related neurologic sequelae as well as neuropsychological deficits and social difficulties have moved into focus in follow-up care. In order to provide personalized treatment recommendations for pediatric brain tumor survivors, it is crucial not only to assess cognitive impairments but also to measure a patient's functional deficiencies, for example, restricted participation in everyday social activities. Thus, this article introduces the International Classification of Functioning-Children and Youth version (ICF-CY) as a conceptual framework for quantifying functional limitations and informing long-term care in pediatric neuro-oncology. A standardized self-report and proxy-report questionnaire for measuring participation is briefly discussed and specific recommendations based on so-called core sets for clinical practice in pediatric neuro-oncology are provided.

Strategies to improve the quality of survival for childhood brain tumour survivors.

BACKGROUND: Tumours of the central nervous system (CNS) are the most frequent solid tumours and the second most frequent type of cancer in children and adolescents. Overall survival has continuously improved in Germany, since an increasing number of patients have been treated according to standardised, multicentre, multimodal treatment recommendations, trials of the German Paediatric Brain Tumour Consortium (HIT-Network) or the International Society of Paediatric Oncology-Europe (SIOP-E) during the last decades. Today, two out of three patients survive. At least 8000 long-term childhood brain tumour survivors (CBTS) are currently living in Germany. They face lifelong disease- and treatment-related late effects (LE) and associated socioeconomic problems more than many other childhood cancer survivors (CCS). METHOD: We review the LE and resulting special needs of this particular group of CCS. RESULTS: Despite their increasing relevance for future treatment optimisation, neither the diversity of chronic and cumulative LE nor their pertinent risk factors and subsequent impact on quality of survival have yet been comprehensively addressed for CBTS treated according to HIT- or SIOP-E-protocols. Evidence-based information to empower survivors and stakeholders, as well as medical expertise to manage their individual health care, psychosocial and educational/vocational needs must still be generated and established. CONCLUSION: The establishment of a long-term research- and care network in Germany shall contribute to a European platform, that aims at optimising CBTSs' transition into adulthood as resilient individuals with high quality of survival including optimal levels of activity, participation and acceptance by society.