A joint international consensus statement for measuring quality of survival for patients with childhood cancer.

The aim of treating childhood cancer remains to cure all. As survival rates improve, long-term health outcomes increasingly define quality of care. The International Childhood Cancer Outcome Project developed a set of core outcomes for most types of childhood cancers involving relevant international stakeholders (survivors; pediatric oncologists; other medical, nursing or paramedical care providers; and psychosocial or neurocognitive care providers) to allow outcome-based evaluation of childhood cancer care. A survey among healthcare providers (n = 87) and online focus groups of survivors (n = 22) resulted in unique candidate outcome lists for 17 types of childhood cancer (five hematological malignancies, four central nervous system tumors and eight solid tumors). In a two-round Delphi survey, 435 healthcare providers from 68 institutions internationally (response rates for round 1, 70-97%; round 2, 65-92%) contributed to the selection of four to eight physical core outcomes (for example, heart failure, subfertility and subsequent neoplasms) and three aspects of quality of life (physical, psychosocial and neurocognitive) per pediatric cancer subtype. Measurement instruments for the core outcomes consist of medical record abstraction, questionnaires and linkage with existing registries. This International Childhood Cancer Core Outcome Set represents outcomes of value to patients, survivors and healthcare providers and can be used to measure institutional progress and benchmark against peers.

Evaluating the feasibility, effectiveness and costs of implementing person-centred follow-up care for childhood cancer survivors in four European countries: the PanCareFollowUp Care prospective cohort study protocol.

INTRODUCTION: Long-term survival after childhood cancer often comes at the expense of late, adverse health conditions. However, survivorship care is frequently not available for adult survivors in Europe. The PanCareFollowUp Consortium therefore developed the PanCareFollowUp Care Intervention, an innovative person-centred survivorship care model based on experiences in the Netherlands. This paper describes the protocol of the prospective cohort study (Care Study) to evaluate the feasibility and the health economic, clinical and patient-reported outcomes of implementing PanCareFollowUp Care as usual care in four European countries. METHODS AND ANALYSIS: In this prospective, longitudinal cohort study with at least 6 months of follow-up, 800 childhood cancer survivors will receive the PanCareFollowUp Care Intervention across four study sites in Belgium, Czech Republic, Italy and Sweden, representing different healthcare systems. The PanCareFollowUp Care Intervention will be evaluated according to the Reach, Effectiveness, Adoption, Implementation and Maintenance framework. Clinical and research data are collected through questionnaires, a clinic visit for multiple medical assessments and a follow-up call. The primary outcome is empowerment, assessed with the Health Education Impact Questionnaire. A central data centre will perform quality checks, data cleaning and data validation, and provide support in data analysis. Multilevel models will be used for repeated outcome measures, with subgroup analysis, for example, by study site, attained age, sex or diagnosis. ETHICS AND DISSEMINATION: This study will be conducted in accordance with the guidelines of Good Clinical Practice and the Declaration of Helsinki. The study protocol has been reviewed and approved by all relevant ethics committees. The evidence and insights gained by this study will be summarised in a Replication Manual, also including the tools required to implement the PanCareFollowUp Care Intervention in other countries. This Replication Manual will become freely available through PanCare and will be disseminated through policy and press releases. TRIAL REGISTRATION NUMBER: Netherlands Trial Register (NL8918; https://www.trialregister.nl/trial/8918).

The PanCareFollowUp Care Intervention: A European harmonised approach to person-centred guideline-based survivorship care after childhood, adolescent and young adult cancer.

BACKGROUND: Long-term follow-up (LTFU) care, although endorsed, is not available for the majority of adult survivors of childhood, adolescence and young adult (CAYA) cancer. Barriers to implementation include lack of time, knowledge, personnel and funding. Sustainable solutions are urgently needed to address the needs of CAYA cancer survivors to improve the quality of life and reduce the burden of late effects on survivors, health care systems and society. The European Union-funded PanCareFollowUp project, initiated by the Pan-European Network for Care of Survivors after Childhood and Adolescent Cancer, was established to facilitate the implementation of person-centred survivorship care across Europe. PATIENTS AND METHODS: The PanCareFollowUp Care Intervention was co-developed with survivors as part of the PanCareFollowUp project. It is a person-centred approach to survivorship care, supported by guidelines and with flexibility to adapt to local health care settings. The Care Intervention consists of three steps: (1) previsit completion of a Survivor Questionnaire (by the survivor) and Treatment Summary (by the health care provider [HCP]), (2) a clinic visit including shared decision-making, and (3) a follow-up call to finalise the individualised Survivorship Care Plan. RESULTS: We developed the key components of the PanCareFollowUp Care Intervention: a PanCareFollowUp Survivor Questionnaire, Treatment Summary template, Survivorship Care Plan template, and educational materials for HCPs and survivors. Wide implementation of the PanCareFollowUp Care Intervention will be supported with a freely distributed Replication Manual on completion of the PanCareFollowUp project. CONCLUSIONS: The PanCareFollowUp Care Intervention will support the implementation of person-centred, guideline-based LTFU care in different health care settings across Europe to improve survivors' health and well-being.

The Concept of Cancer Survivorship and Models for Long-Term Follow-Up.

With improved survival of childhood, adolescent, and young adult (CAYA) cancer, the European survivor population of 300,000-500,000 continues to expand. Most survivors will experience at least one and often multiple cancer- and treatment-related late effects throughout their lives, including endocrine toxicities. Besides affecting their physical and psychosocial health status, these might reduce life expectancy and quality of life. Prevalent endocrine complications include hypothalamic-pituitary dysfunction, central precocious puberty, primary thyroid, male or female gonadal dysfunction, metabolic syndrome, and low bone mineral density. Long-term follow-up (LTFU) care, including education, risk-based prevention, and surveillance strategies, is essential to reduce the burden of endocrine complications and to allow for timely interventions. To integrate scientific expert knowledge, evidence-based clinical practice guidelines have been developed by the International Late Effects of Childhood Cancer Guideline Harmonization Group (IGHG) and PanCare. These guide LTFU care by describing risk populations and preferred surveillance modalities. Moreover, consensus-based recommendations have been developed by PanCareFollowUp where evidence-based guidance is still awaited. The PanCareSurFup models of care guidelines recommend multidisciplinary team care at or under guidance of a cancer survivorship expert center, so CAYA cancer survivors receive appropriate care and support to optimize health.

Early and midterm outcomes of bare metal stenting in small children with recurrent aortic coarctation.

AIMS: The aim of this study was to report our experience with the Cook Formula stent in the treatment of (recurrent) coarctation of the aorta in children below 12 kg. METHODS AND RESULTS: In vitro study of the Cook Formula 418 (8 mm) and 535 (8 and 10 mm) stents demonstrated successful down-crimping on smaller balloons and predictable fracturing patterns. Between November 2012 and January 2019, one patient with native, one patient with post-interventional and thirteen patients with post-surgical coarctation of the aorta underwent implantation of a Cook Formula stent. Patient and procedural characteristics were obtained as well as procedural success, complications, and follow-up. Median age was 4.3 months and median weight 5.5 kg. Arterial sheath size ranged from 5 to 7 Fr. In-stent diameters of 3.7 to 8.8 mm were obtained with a median residual gradient of 0 mmHg. Major complications consisted of periprocedural haemodynamic instability (n=1), dissection of the iliac artery (n=1) and non-deployment with surgical removal (n=1). Re-dilations were performed after a median interval of 24.3 months. Median follow-up was 31.7 months. CONCLUSIONS: The bare metal Cook Formula stent provides a durable and effective alternative to reoperation and balloon dilatation for native as well as post-surgical aortic coarctation in children below 12 kg.

European PanCareFollowUp Recommendations for surveillance of late effects of childhood, adolescent, and young adult cancer.

BACKGROUND: Long-term follow-up (LTFU) care for childhood, adolescent, and young adult (CAYA) cancer survivors is essential to preserve health and quality of life (QoL). Evidence-based guidelines are needed to inform optimal surveillance strategies, but many topics are yet to be addressed by the International Late Effects of Childhood Cancer Guideline Harmonization Group (IGHG). Therefore, the PanCareFollowUp Recommendations Working Group collaborated with stakeholders to develop European harmonised recommendations in anticipation of evidence-based IGHG guidelines. METHODS: The PanCareFollowUp Recommendations Working Group, consisting of 23 late effects specialists, researchers, and survivor representatives from nine countries, collaborated in the first Europe-wide effort to provide unified recommendations in anticipation of evidence-based guidelines. A pragmatic methodology was used to define recommendations for topics where no evidence-based IGHG recommendations exist. The objective was to describe the surveillance requirements for high-quality care while balancing the different infrastructures and resources across European health care systems. The process included two face-to-face meetings and an external consultation round involving 18 experts from 14 countries. RESULTS: Twenty-five harmonised recommendations for LTFU care were developed collaboratively and address topics requiring awareness only (n = 6), awareness, history and/or physical examination (n = 9), or additional surveillance tests (n = 10). CONCLUSIONS: The PanCareFollowUp Recommendations, representing a unique agreement across European stakeholders, emphasise awareness among survivors and health care providers in addition to tailored clinical evaluation and/or surveillance tests. They include existing IGHG guidelines and additional recommendations developed by a pragmatic methodology and will be used in the Horizon 2020-funded PanCareFollowUp project to improve health and QoL of CAYA cancer survivors.

The European multistakeholder PanCareFollowUp project: novel, person-centred survivorship care to improve care quality, effectiveness, cost-effectiveness and accessibility for cancer survivors and caregivers.

BACKGROUND: The majority of childhood cancer survivors are at risk of treatment-related adverse health outcomes. Survivorship care to mitigate these late effects is endorsed, but it is not available for many adult survivors of childhood cancer in Europe. The PanCareFollowUp project was initiated to improve their health and quality of life (QoL) by facilitating person-centred survivorship care. METHODS: The PanCareFollowUp consortium was established in 2018, consisting of 14 project partners from ten European countries, including survivor representatives. The consortium will develop two PanCareFollowUp Interventions, including a person-centred guideline-based model of care (Care Intervention) and eHealth lifestyle coaching (Lifestyle Intervention). Their development will be informed by several qualitative studies and systematic reviews on barriers and facilitators for implementation and needs and preferences of healthcare providers (HCPs) and survivors. Implementation of the PanCareFollowUp Care Intervention as usual care will be evaluated prospectively among 800 survivors from Belgium, Czech Republic, Italy and Sweden for survivor empowerment, detection of adverse health conditions, satisfaction among survivors and HCPs, cost-effectiveness and feasibility. The feasibility of the PanCareFollowUp Lifestyle Intervention will be evaluated in the Netherlands among 60 survivors. RESULTS: Replication manuals, allowing for replication of the PanCareFollowUp Care and Lifestyle Intervention, will be published and made freely available after the project. Moreover, results of the corresponding studies are expected within the next five years. CONCLUSIONS: The PanCareFollowUp project is a novel European collaboration aiming to improve the health and QoL of all survivors across Europe by developing and prospectively evaluating the person-centred PanCareFollowUp Care and Lifestyle Interventions.

The Critical Role of Clinical Practice Guidelines and Indicators in High-Quality Survivorship After Childhood Cancer.

Childhood cancer survivors are at significant risk for late cancer treatment-related morbidity and mortality. Physicians involved in the care of childhood cancer survivors should be aware of these specific health problems and provide high-quality, long-term follow-up care to preserve and improve survivors' health. The steps required to achieve high-quality care include synthesizing evidence (systematic reviews are helpful in this regard), developing clinical policy from evidence into evidence-based clinical practice guidelines, disseminating and implementing clinical practice guidelines, and evaluating their impact on quality of care and survivor health outcomes with quality indicators. This article describes these cornerstones of evidence-based medicine.