IT-Related Barriers and Facilitators to the Implementation of a New European eHealth Solution, the Digital Survivorship Passport (SurPass Version 2.0): Semistructured Digital Survey.

BACKGROUND: To overcome knowledge gaps and optimize long-term follow-up (LTFU) care for childhood cancer survivors, the concept of the Survivorship Passport (SurPass) has been invented. Within the European PanCareSurPass project, the semiautomated and interoperable SurPass (version 2.0) will be optimized, implemented, and evaluated at 6 LTFU care centers representing 6 European countries and 3 distinct health system scenarios: (1) national electronic health information systems (EHISs) in Austria and Lithuania, (2) regional or local EHISs in Italy and Spain, and (3) cancer registries or hospital-based EHISs in Belgium and Germany. OBJECTIVE: We aimed to identify and describe barriers and facilitators for SurPass (version 2.0) implementation concerning semiautomation of data input, interoperability, data protection, privacy, and cybersecurity. METHODS: IT specialists from the 6 LTFU care centers participated in a semistructured digital survey focusing on IT-related barriers and facilitators to SurPass (version 2.0) implementation. We used the fit-viability model to assess the compatibility and feasibility of integrating SurPass into existing EHISs. RESULTS: In total, 13/20 (65%) invited IT specialists participated. The main barriers and facilitators in all 3 health system scenarios related to semiautomated data input and interoperability included unaligned EHIS infrastructure and the use of interoperability frameworks and international coding systems. The main barriers and facilitators related to data protection or privacy and cybersecurity included pseudonymization of personal health data and data retention. According to the fit-viability model, the first health system scenario provides the best fit for SurPass implementation, followed by the second and third scenarios. CONCLUSIONS: This study provides essential insights into the information and IT-related influencing factors that need to be considered when implementing the SurPass (version 2.0) in clinical practice. We recommend the adoption of Health Level Seven Fast Healthcare Interoperability Resources and data security measures such as encryption, pseudonymization, and multifactor authentication to protect personal health data where applicable. In sum, this study offers practical insights into integrating digital health solutions into existing EHISs.

Scaling up and implementing the digital Survivorship Passport tool in routine clinical care - The European multidisciplinary PanCareSurPass project.

BACKGROUND: Childhood cancer survivors (CCS), of whom there are about 500,000 living in Europe, are at an increased risk of developing health problems [1-6] and require lifelong Survivorship Care. There are information and knowledge gaps among CCS and healthcare providers (HCPs) about requirements for Survivorship Care [7-9] that can be addressed by the Survivorship Passport (SurPass), a digital tool providing CCS and HCPs with a comprehensive summary of past treatment and tailored recommendations for Survivorship Care. The potential of the SurPass to improve person-centred Survivorship Care has been demonstrated previously [10,11]. METHODS: The EU-funded PanCareSurPass project will develop an updated version (v2.0) of the SurPass allowing for semi-automated data entry and implement it in six European countries (Austria, Belgium, Germany, Italy, Lithuania and Spain), representative of three infrastructure healthcare scenarios typically found in Europe. The implementation study will investigate the impact on person-centred care, as well as costs and processes of scaling up the SurPass. Interoperability between electronic health record systems and SurPass v2.0 will be addressed using the Health Level Seven (HL7) International interoperability standards. RESULTS: PanCareSurPass will deliver an interoperable digital SurPass with comprehensive evidence on person-centred outcomes, technical feasibility and health economics impacts. An Implementation Toolkit will be developed and freely shared to promote and support the future implementation of SurPass across Europe. CONCLUSIONS: PanCareSurPass is a novel European collaboration that will improve person-centred Survivorship Care for CCS across Europe through a robust assessment of the implementation of SurPass v2.0 in different healthcare settings.

Perceived social support and characteristics of social networks of families with children with special healthcare needs following the COVID-19 pandemic.

Background: Children with special healthcare needs (CSHCN) require more support than the average of their peers. Support systems for CSHCN were particularly affected by pandemic control measures. Perceived social support is a resource for health and wellbeing for CSHCN and their families. Associations of social support, mental health and socioeconomic status (SES) have been described. This study aims to (1) assess perceived social support in families with and without CSHCN; (2) describe structure and types of social networks of families with and without CSHCN; and (3) explore associations between perceived social support, disease complexity, child and caregiver mental health, and SES. Methods: This is the third of a sequential series of cross-sectional online surveys conducted among caregivers of children ≤ 18 years in Germany since the beginning of the COVID-19 pandemic, administered between 1st December 2022 and 10 March 2023. The Brief Social Support Scale (BS6) assessed perceived social support. Child and parental mental health were assessed using the Strengths and Difficulties Questionnaire (SDQ) and WHO-5 Wellbeing index. The CSHCN-Screener identified CSHCN. Descriptive statistics and linear regression modeling assessed associations between perceived social support, parent-reported child mental health problems, disease complexity, caregiver mental wellbeing and SES. Results: The final sample included 381 participants, among them 76.6% (n = 292) CSHCN. 46.2% (n = 176) of caregivers reported moderate, i.e., at least occasional social support. Social support was largely provided by informal social networks consisting of partners, relatives and neighbors/friends. Linear regression modeling revealed associations of lower perceived social support with higher disease complexity of the child, lower caregiver mental wellbeing, lower SES and increasing caregiver age. Conclusion: The results of this study describe inequalities in perceived social support according to disease complexity of the child, caregiver mental health and socioeconomic status. They highlight the importance of social support and support networks as a resource for wellbeing of caregivers and CSHCN. Moving on from the COVID-19 pandemic, recovery strategies should focus on low-threshold interventions based in the community to improve social support for families with CSHCN and actively involve caregivers in identifying needs and co-creating new approaches.

VersKiK qualitative study design: actual follow-up needs of paediatric cancer survivors, their informal caregivers and follow-up stakeholder perceptions in Germany.

INTRODUCTION: This article presents the study design of the qualitative part of the VersKiK study (Long-term care, care needs and wellbeing of individuals after cancer in childhood or adolescence: study protocol of a large scale multi-methods non-interventional study) aiming to explore actual follow-up needs of childhood and adolescence cancer survivors and their informal caregivers, gaps in current follow-up care provision and trajectories of cancer survivors' transition from paediatric to adult healthcare. METHODS AND ANALYSIS: We will conduct up to 30 interviews with survivors of childhood and adolescence cancer and their informal caregivers with up to 20 participant observations of follow-up appointments. The results of these will be discussed in up to four focus groups with healthcare professionals and representatives of self-help groups. The study design aims to evaluate follow-up care after childhood cancer considering perspectives from survivors, their informal caregivers as well as healthcare providers. The combination of different data sources will allow us to get an in-depth understanding of the current state of follow-up care after paediatric cancer in Germany and to suggest recommendations for care improvement. ETHICS AND DISSEMINATION: The VersKiK study was approved by the Ethics Committee Otto von Guericke University on 2 July 2021 (103/21), by the Ethics Committee of Johannes Gutenberg University Mainz on 16 June 2021 (2021-16035), by the Ethics Committee University of Lübeck on 10 November 2021 (21-451), by the Ethics Committee University of Hospital Bonn on 28 February 2022 (05/22). For each part of the qualitative study, a separate written informed consent is prepared and approved accordingly by the ethics committees named above. TRIAL REGISTRATION NUMBER: Registered at German Clinical Trial Register, ID: DRKS00026092.

European recommendations for short-term surveillance of health problems in childhood, adolescent and young adult cancer survivors from the end of treatment to 5 years after diagnosis: a PanCare guideline.

PURPOSE: Childhood, adolescent and young adult (CAYA) cancer survivors require ongoing surveillance for health problems from the end of cancer treatment throughout their lives. There is a lack of evidence-based guidelines on optimal surveillance strategies for the period from the end of treatment to 5 years after diagnosis. We aimed to address this gap by developing recommendations for short-term surveillance of health problems based on existing long-term follow-up (LTFU) care guidelines. METHODS: The guideline working group, consisting of healthcare professionals, parents and survivor representatives from 10 countries, worked together to identify relevant health problems that may occur in survivors between the end of treatment and 5 years after diagnosis and to develop recommendations for short-term surveillance of health problems. The recommendations were drawn from existing LTFU guidelines and adapted where necessary based on clinical expertise. RESULTS: The working group developed 44 recommendations for short-term surveillance of health problems, which were divided into four categories based on the level of surveillance required: awareness only (n = 11), awareness, history and/or physical examination without surveillance test (n = 15), awareness, history and/or physical examination with potential surveillance test (n = 1) and awareness, history and/or physical examination with surveillance test (n = 17). CONCLUSION: The development of a guideline for short-term surveillance of health problems fills a critical gap in survivorship care for CAYA cancer survivors, providing much-needed support immediately after treatment up to 5 years after diagnosis. IMPLICATIONS FOR CANCER SURVIVORS: This guideline will support healthcare professionals to provide appropriate follow-up care and improve the quality of life of CAYA cancer survivors.

Barriers and facilitators to the implementation of a new European eHealth solution (SurPass v2.0): the PanCareSurPass Open Space study.

PURPOSE: To identify barriers and facilitators for implementing the Survivorship Passport (SurPass) v2.0 in six long-term follow-up (LTFU) care centres in Europe. METHODS: Stakeholders including childhood cancer survivors (CCSs), healthcare providers (HCPs), managers, information and technology (IT) specialists, and others, participated in six online Open Space meetings. Topics related to Care, Ethical, Legal, Social, Economic, and Information & IT-related aspects of implementing SurPass were evaluated. RESULTS: The study identified 115 barriers and 159 facilitators. The main barriers included the lack of standardised LTFU care in centres and network cooperation, uncertainty about SurPass accessibility, and uncertainty about how to integrate SurPass into electronic health information systems. The main facilitators included standardised and coordinated LTFU care in centres, allowing CCSs to conceal sensitive information in SurPass and (semi)automatic data transfer and filing. CONCLUSIONS: Key barriers to SurPass implementation were identified in the areas of care, ethical considerations, and information & IT. To address these barriers and facilitate the implementation on SurPass, we have formulated 27 recommendations. Key recommendations include using the internationally developed protocols and guidelines to implement LTFU care, making clear decisions about which parties have access to SurPass data in accordance with CCSs, and facilitating (semi)automated data transfer and filing using Health Level 7 (HL7) Fast Healthcare Interoperability Resources (FHIR). IMPLICATIONS FOR CANCER SURVIVORS: The findings of this study can help to implement SurPass and to ensure that cancer survivors receive high-quality LTFU care with access to the necessary information to manage their health effectively.

Proposal of a diagnostic algorithm for radiation-induced dropped head syndrome in long-term childhood cancer survivors based on a prospective study in a specialized clinical setting and a review of the literature.

PURPOSE: To prospectively assess the incidence of Dropped Head Syndrome (DHS) in childhood cancer survivors (CCS) and to develop and evaluate a diagnostic algorithm for DHS. METHODS: A systematic literature search for DHS in combination with neck radiotherapy (RT) exposure was performed. Analyses and a combination of the most common examination methods were integrated into a diagnostic algorithm. Almost all CCSs visiting the local late effects clinic between May 2020 and April 2022 were included in the study. CCS exposed to neck RT with doses ≥ 19 Gy received standardized clinical and neurological assessment and, in case of abnormal results, an MRI scan to confirm muscle atrophy. RESULTS: Two hundred and five CCS were included of whom 41 received RT to the neck with ≥ 19 Gy. In the entire cohort and in the subgroup receiving RT, 2.4% and 12% of CCS were affected by DHS, respectively. Results of clinical and neurological assessment correlated well with MRI results. Neck circumference and neck/thigh ratio were lower after neck RT. Over 50% of CCS experienced neck disability and pain. CONCLUSIONS: A relevant proportion of CCS exposed to neck RT is affected by DHS. High concordance of MRI results with the neurological examination supports the clinical value of the diagnostic algorithm. Measurement of neck circumference might be an easy tool for assessment of neck muscle atrophy in survivors at risk. IMPLICATIONS FOR CANCER SURVIVORS: Integration of a diagnostic algorithm for DHS in standard long-term follow-up care facilitates diagnosis as well as initiation of early treatment and obviates the need for invasive examinations.

Evaluating case management for caregivers of children with spinal muscular atrophy type I and II-an exploratory, controlled, mixed-methods trial.

Introduction: Spinal muscular atrophy (SMA) is a rare neuromuscular disease requiring various clinical specialists and therapists to provide care. Due to the disease's dynamic nature and the long distances between specialized centers and local providers, integrating care between disciplines can be challenging. Care that is inadequately integrated can compromise the quality of care and become a burden for patients and families. This trial aimed to improve the care of patients through a case management (CM) intervention. Methods: We conducted an exploratory, controlled, two-arm trial with pre-, post-, and follow-up measures (process and outcome evaluation). Proof of efficacy based on statistical significance was not our primary study objective since we were investigating a rare disease. Primary outcomes were caregivers' HRQoL and caregiver-rated quality of care integration. Our secondary outcome was the children's HRQoL. Results: Questionnaires and semi-structured interviews yielded heterogeneous results depending on caregivers' level of experience and desire (or possibility) to delegate care tasks. Discussion: Despite differing perceptions, all participants supported the establishment of a care coordination model. We recommend CM immediately after diagnosis to provide the greatest benefit to families. We hope that our trial will support the further development of CM interventions that can be customized for specific diseases.

Auditory complications among childhood cancer survivors and health-related quality of life: a PanCareLIFE study.

PURPOSE: Auditory complications are potential side effects from childhood cancer treatment. Yet, limited evidence exists about the impact of auditory complications-particularly tinnitus-on health-related quality of life (HRQoL) among childhood cancer survivors (CCS). We determined the prevalence of hearing loss and tinnitus in the European PanCareLIFE cohort of CCS and examined its effect on HRQoL. METHODS: We included CCS from four European countries who were diagnosed at age ≤ 18 years; survived ≥ 5 years; and aged 25-44 years at study. We assessed HRQoL (Short Form 36), hearing loss, and tinnitus using questionnaires. We used multivariable linear regression to examine associations between these two auditory complications and HRQoL adjusting for socio-demographic and clinical factors. RESULTS: Our study population consisted of 6,318 CCS (53% female; median age at cancer diagnosis 9 years interquartile range [IQR] 5-13 years) with median age at survey of 31 years (IQR 28-35 years). Prevalence was 7.5% (476/6,318; confidence interval [CI]: 6.9-8.2) for hearing loss and 7.6% (127/1,668; CI: 6.4-9.0) for tinnitus. CCS with hearing loss had impaired physical (coefficient [coef.] -4.3, CI: -7.0 to -1.6) and mental (coef. -3.2, CI: -5.5 to -0.8) HRQoL when compared with CCS with normal hearing. Tinnitus was associated with impaired physical (coef. -8.2, CI: -11.8 to -4.7) and mental (coef. -5.9, CI: -8.8 to -3.1) HRQoL. CONCLUSION: We observed reduced HRQoL among CCS with hearing loss and tinnitus. Our findings indicate timely treatment of hearing loss and tinnitus may contribute to quality of life of survivors. IMPLICATIONS FOR CANCER SURVIVORS: CCS who experience auditory complications should be counseled about possible therapeutic and supportive measures during follow-up care.

Implementation of a clinical long-term follow-up database for adult childhood cancer survivors in Germany: a feasibility study at two specialised late effects clinics.

PURPOSE: Childhood cancer survivors (CCS) are at risk for increased morbidity and reduced quality of life associated with treatment-related late effects. In Germany, however, only a few of the more than 40,000 CCS registered in the German Childhood Cancer Registry (GCCR) currently benefit from adequate clinical long-term follow-up (LTFU) structures. To establish a comprehensive knowledge base on CCS' long-term health in Germany, a database was developed in cooperation with the GCCR. Following a first evaluation phase at two German university centres, this database will be implemented more widely within Germany allowing longitudinal documentation of clinical LTFU data. METHODS: The feasibility study cohort comprised 208 CCS aged 18 or older whose medical, mental and psychosocial health data were collected during routine LTFU or first clinic visits in adult care. CCS were enrolled from 04/2021 to 12/2022, and data entry was completed by 03/2023. Descriptive data analysis was conducted. All CCS were stratified into three risk groups (RG) based on their individual risk for developing late effects resulting from their respective diagnoses and treatments. RESULTS: Chronic health conditions of various organ systems associated with late and long-term effects of cancer therapy affected CCS in all RG supporting the clinical relevance of risk-adapted LTFU. Enrolment into the database was feasible and broadly accepted amongst CCS. CONCLUSION: Implementation of a clinical follow-up care infrastructure and database in Germany will pave the way to collect clinically evaluated and regularly updated health data of potentially over 40,000 German CCS and facilitate future national and international cooperation.

Updated International Guidelines for Survivorship Care after Pediatric Cancer: Practice Implications in a German and Austrian Comprehensive Care Network.

BACKGROUND: Timely diagnosis of treatment-related chronic health conditions in childhood cancer survivors (CCS) may result in reduced long-term morbidity and mortality. Evidence-based guidelines serve as a tool to implement risk-adapted screening examinations in long-term follow-up (LTFU) of CCS. SUMMARY: New international LTFU guidelines from the last 3 years have been reviewed and included into a practical LTFU tool in order to provide an updated summary of LTFU recommendations. The inclusion of 13 new LTFU guidelines as well as 25 pragmatic recommendations resulted in an updated LTFU plan for implantation in daily practice. Special consideration of psychosocial and mental health aspects as well as recommendations for pregnant CCS complement holistic LTFU care. KEY MESSAGES: Risk-adapted LTFU in CCS offers the possibility for early detection and treatment of late effects. As these LTFU recommendations aim at asymptomatic individuals, benefits and potential risks of regular screening examinations have to be carefully balanced. Implementation of current evidence-based guidelines in clinical practice as well as the development of new application tools such as the Survivorship Passport can contribute to an individualized LTFU approach in order to ensure long-term health and quality of life in CCS.

Barriers and facilitating factors of care coordination for children with spinal muscular atrophy type I and II from the caregivers' perspective: an interview study.

BACKGROUND: Children with medical complexity (CMC) require long-term care accompanied by different health- and social care professionals. Depending on the severity of the chronic condition, caregivers spend a lot of time coordinating appointments, communicating between providers, clarifying social legal issues, and more. Effective care coordination is seen as key to addressing the fragmented care that CMC and their families often face. Spinal muscular atrophy (SMA) is a rare genetic, neuromuscular disease which care involves drug therapy and supportive treatment. We examined the care coordination experiences through a qualitative interview analysis of n = 21 interviews with caregivers of children with SMA I or SMA II. RESULTS: The code system consists of 7 codes and 12 sub-codes. "Disease and coordination management of the caregivers" describes the management of coordination-related illness demands. "General conditions of care" include enduring organizational aspects of the care network. "Expertise and skills" refers to both parent and professional expertise. "Coordination structure" describes the assessment of existing coordination mechanisms as well as the need for new ones. "Information exchange" defines the information exchange between professionals and parents as well as the exchange of parents among themselves and the perceived exchange between professionals. "Role distribution in care coordination" summarizes parents' "distribution" of coordinative roles among care network actors (including their own). "Quality of relationship" describes the perceived quality of the relationship between professionals and family. CONCLUSION: Care coordination is influenced peripherally (e.g., by general conditions of care) and directly (e.g., by coordination mechanisms, interaction in the care network). Access to care coordination appears to be dependent on family circumstances, geographic location, and institutional affiliation. Previous coordination mechanisms were often unstructured and informal. Care coordination is frequently in the hands of caregivers mainly as the care network's interface. Coordination is necessary and must be addressed on an individual basis of existing resources and family barriers. Existing coordination mechanisms in the context of other chronic conditions could also work for SMA. Regular assessments, centralized shared care pathways, and staff training and empowerment of families for self-management should be central components of all coordination models. TRIAL REGISTRATION: German Clinical Trials Register (DRKS): DRKS00018778; Trial registration date 05. December 2019-Retrospectively registered; https://apps.who.int/trialsearch/Trial2.aspx?TrialID=DRKS00018778 .

Equivalent doses for anticancer agents used in pediatric oncology: A literature review and evaluation of a novel approach for conversion factors.

BACKGROUND: Epidemiological research on late effects of therapy shows the necessity to aggregate chemotherapy agents to substance classes. This requires using conversion factors by substance classes. AIMS: The aim of this study was to identify previously used conversion factors from the literature, to present a novel approach for additional factors, and to compare these approaches. METHODS AND RESULTS: A literature review was performed, which identified two main principles of deriving conversion factors: effect-equivalence and equimolar. Thirty-five articles presenting effect equivalence-based factors in the widest sense were found in the literature. Ten articles presented the equimolar approach which can be applied to almost all chemotherapy substances. Based on a comprehensive list of treatment protocols used in German pediatric oncology, we derived alternative conversion factors from typical doses. We compared the conversion factors using Pearson correlation coefficients and linear regression. At least two types of conversion factor were available for each of the 49 substances included. The equivalent effect-based and the typical dose-based factors were highly correlated with a regression coefficient close to 1. The equimolar factors are independent. CONCLUSIONS: For substances for which no conversion factor based on some type of effect equivalence has been published so far, a factor based on a typical doses-approach may be used in epidemiological late effects research. Doses aggregated based on the equimolar approach may not be compatible with doses aggregated based on equivalent effects.

Establishment, Maintenance, and Performance of the Cooperative Osteosarcoma Study Group (COSS).

INTRODUCTION: Osteosarcoma treatment has benefitted greatly from collaborative research. This paper describes the history and accomplishments of the Cooperative Osteosarcoma Study Group (COSS), mainly dedicated to clinical questions, as well as remaining challenges. MATERIALS AND METHODS: Narrative review of over four decades of uninterrupted collaboration within the multi-national German-Austrian-Swiss COSS group. RESULTS: Since its very first prospective osteosarcoma trial starting in 1977, COSS has continuously been able to provide high-level evidence on various tumor- and treatment-related questions. This includes both the cohort of patients enrolled into prospective trials as well as those patients excluded from them for various reasons, followed in a prospective registry. Well over one hundred disease-related publications attest to the group's impact on the field. Despite these accomplishments, challenging problems remain. DISCUSSION: Collaborative research within a multi-national study group resulted in better definitions of important aspects of the most common bone tumor, osteosarcoma, and its treatments. Important challenges continue to persist.

Barriers and facilitators to implementation of the interoperable Survivorship Passport (SurPass) v2.0 in 6 European countries: a PanCareSurPass online survey study.

PURPOSE: Long-term follow-up (LTFU) care for childhood cancer survivors (CCSs) is essential to improve and maintain their quality of life. The Survivorship Passport (SurPass) is a digital tool which can aid in the delivery of adequate LTFU care. During the European PanCareSurPass (PCSP) project, the SurPass v2.0 will be implemented and evaluated at six LTFU care clinics in Austria, Belgium, Germany, Italy, Lithuania and Spain. We aimed to identify barriers and facilitators to the implementation of the SurPass v2.0 with regard to the care process as well as ethical, legal, social and economical aspects. METHODS: An online, semi-structured survey was distributed to 75 stakeholders (LTFU care providers, LTFU care program managers and CCSs) affiliated with one of the six centres. Barriers and facilitators identified in four centres or more were defined as main contextual factors influencing implementation of SurPass v2.0. RESULTS: Fifty-four barriers and 50 facilitators were identified. Among the main barriers were a lack of time and (financial) resources, gaps in knowledge concerning ethical and legal issues and a potential increase in health-related anxiety in CCSs upon receiving a SurPass. Main facilitators included institutions' access to electronic medical records, as well as previous experience with SurPass or similar tools. CONCLUSIONS: We provided an overview of contextual factors that may influence SurPass implementation. Solutions should be found to overcome barriers and ensure effective implementation of SurPass v2.0 into routine clinical care. IMPLICATIONS FOR CANCER SURVIVORS: These findings will be used to inform on an implementation strategy tailored for the six centres.

Secondary Malignancies after Ewing Sarcoma-Epidemiological and Clinical Analysis of an International Trial Registry.

Ewing sarcoma (EwS) represents highly aggressive bone and soft tissue tumors that require intensive treatment by multi-chemotherapy, surgery and/or radiotherapy. While therapeutic regimens have increased survival rates, EwS survivors face long-term sequelae that include secondary malignant neoplasms (SMNs). Consequently, more knowledge about EwS patients who develop SMNs is needed to identify high-risk patients and adjust follow-up strategies. We retrospectively analyzed data from 4518 EwS patients treated in five consecutive EwS trials from the Cooperative Ewing Sarcoma Study (CESS) group. Ninety-six patients developed SMNs after primary EwS, including 53 (55.2%) with solid tumors. The latency period between EwS and the first SMN was significantly longer for the development of solid SMNs (median: 8.4 years) than for hematologic SMNs (median: 2.4 years) (p < 0.001). The cumulative incidence (CI) of SMNs in general increased over time from 0.04 at 10 years to 0.14 at 30 years; notably, the specific CI for hematologic SMNs remained stable over the different decades, whereas for solid SMNs it gradually increased over time and was higher for metastatic patients than in localized EwS patients (20 years: 0.14 vs. 0.06; p < 0.01). The clinical characteristics of primary EwS did not differ between patients with or without SMNs. All EwS patients received multi-chemotherapy with adjuvant radiotherapy in 77 of 96 (80.2%) patients, and the use of radiation doses ≥ 60 Gy correlated with the occurrence of SMNs. The survival rate after SMNs was 0.49, with a significantly better outcome for solid SMNs compared with hematologic SMNs (3 years: 0.70 vs. 0.24, respectively; p < 0.001). The occurrence of SMNs after EwS remains a rare event but requires a structured follow-up system because it is associated with high morbidity and mortality.

[Care of refugee children and adolescents with chronic diseases and disabilities]. / Versorgung von geflüchteten Kindern und Jugendlichen mit chronischen Erkrankungen und Behinderungen: Anspruch und Möglichkeiten in Deutschland.

Refugee children and adolescents with chronic diseases and disabilities are among the most vulnerable persons as their health and developmental chances are considerably at risk. This article describes the challenges and opportunities in the care of this group of patients from the perspective of different care sectors: initial reception center, public health service, pediatricians in private practice, social pediatric centers and patient organizations. The starting point is a presentation of the rights to optimal healthcare that can be derived from the United Nations (UN) Convention on the Rights of Persons with Disabilities and the UN Convention on the Rights of the Child. It becomes clear that for children and adolescents in the status of asylum seekers there are systematic gaps in the recognition and care of chronic diseases, disabilities and support needs. An expansion of the health examination after arrival, which has so far focused on the detection of communicable diseases, is important and necessary in order to identify individual needs and improve the data situation for this group. A strengthening of the school entry screening by the public health service, especially for older children entering school as lateral entrants, could also significantly improve the nationwide coverage. In contrast to these deficits, which require changes at the political level, there are innovative models of care, especially in local contexts, such as pediatric consultation clinics in initial reception centers, diverse examples of voluntary commitment or the use of social media in patient organizations, which are presented as examples.

Mental health of children with and without special healthcare needs and of their caregivers during COVID-19: a cross-sectional study.

OBJECTIVE: To describe mental health outcomes and measures of pandemic burden of children with and without special healthcare needs, and their caregivers following the second wave of the COVID-19 pandemic in Germany. DESIGN: This is the second of a sequential series of cross-sectional online surveys conducted among caregivers of children ≤18 years since the onset of the COVID-19 pandemic, administrated between 2 April 2021 and 31 July 2021. MAIN OUTCOME MEASURES: Child and parental mental health were assessed using the Strengths and Difficulties Questionnaire and WHO-5 Well-being index. Children with Special Healthcare Needs (CSHCN) were identified using the CSHCN-Screener. Descriptive statistics, linear and hierarchical logistic regression modelling assessed associations between parent-reported child mental health problems and measures of pandemic burden, disease complexity, caregiver mental well-being and socioeconomic status. RESULTS: 521 participants were included in the final sample. There was a high prevalence of parent-reported mental health problems in n=302 (66.7%) children, particularly in CSHCN. Among caregivers, n=372 (72.5%) screened positive for depression. Logistic regression modelling showed a strong association of child mental health problems and disease complexity, parental mental well-being, increase in family conflict and inadequate social support. CONCLUSIONS: Our study identifies CSHCN as a particularly vulnerable group in terms of mental health outcomes. Psychosocial factors were important predictors of parent-reported child mental health problems. Policy measures should consider the importance of social support systems for vulnerable children and their families, and aim to provide accessible mental health support for caregivers.

Patientenpartizipation in der pädiatrischen Versorgungsforschung am Universitätsklinikum Freiburg: von der Projektbeteiligung zum Patientenbeirat.

Participation of patients and relatives in research means that those affected are involved in the research process in a partnership role. Despite the growing importance of participatory approaches and the large number of available concepts, many researchers and patients are faced with the question of how participatory research can be realized and organized in concrete terms. Here we report on our experiences with two different forms of patient participation in research in the context of pediatric health care research at a university hospital: (1) In a project for the development and evaluation of a case management for patients with spinal muscular atrophy, patient representatives have an consultative role. (2) In the patient advisory board, which is to accompany the research activities of the research group at the site continuously and systematically, i.e. in all phases, the participation currently corresponds to a contributory role (involvement) which, in the future, could be moved onto the collaborative stage. In both forms of participation, the essential questions include the selection of the participating patients, the type and extent of participation, and the evaluation of the effect of participation on the research that is carried out. In our experience, both forms of participation add value to research from the perspective of all participants. At the same time, they bring different opportunities and challenges. While in project-based participation the sphere of influence is already delineated by researchers, the context of the patient advisory board provides more room and openness to develop, for example, a research agenda and thus identify new research topics. In our experience, however, sufficient resources (in terms of time and money) are required from all participants, as well as good, trusting cooperation with jointly developed processes to realize both forms of participation.