Adolescents with Rett syndrome at critical care pathway junctures: Examining clinicians' decision to initiate invasive long-term ventilation.

BACKGROUND: The initiation of invasive long-term ventilation (I-LTV) for an adolescent with Rett Syndrome (RTT) involves many serious bioethical considerations. In moving towards a more inclusive model of patient participation, transparency surrounding the main influencing factors around this decision is important. OBJECTIVE: We aimed to identify the main drivers influencing a clinician's decision to support initiation of I-LTV for an adolescent with RTT. METHOD: We used an anonymous online vignette-based factorial survey. The survey was distributed internationally through eight professional multi-disciplinary organisations to reach clinicians working in paediatrics. RESULTS: We analysed 504 RTT vignettes completed by 246 clinicians using mixed effect regression modelling. The main three significant influencing factors identified were: parental agreement with the decision to support initiation, the family's support network, and proximity to a tertiary care centre. Additional comments from participants focused on family support, and the importance of on-going communication with the family. CONCLUSION: As the rights of those with disabilities improve and participation of adolescents in decision-making becomes more established, effective communications with the family around goals of care and particular sensitivity and reflective practice around methods of consensus building will likely contribute to a positive decision-making process at this difficult time.

The role of bioethics services in paediatric intensive care units: a qualitative descriptive study.

BACKGROUND: There is considerable variation in the functionality of bioethical services in different institutions and countries for children in hospital, despite new challenges due to increasing technology supports for children with serious illness and medical complexity. We aimed to understand how bioethics services address bioethical concerns that are increasingly encountered in paediatric intensive care. METHODS: A qualitative descriptive design was used to describe clinician's perspectives on the functionality of clinical bioethics services for paediatric intensive care units. Clinicians who were members of formal or informal clinical bioethics groups, or who were closely involved with the process of working through ethically challenging decisions, were interviewed. Interviews took place online. Resulting transcripts were analysed using thematic analysis. RESULTS: From 33 interviews, we identified four themes that described the functionality of bioethics services when a child requires technology to sustain life: striving for consensus; the importance of guidelines; a structure that facilitates a time-sensitive and relevant response; and strong leadership and teamwork. CONCLUSIONS: Clinical bioethics services have the potential to expand their role due to the challenges brought by advancing medical technology and the increasing options it brings for treatment. Further work is needed to identify where and how bioethics services can evolve and adapt to fully address the needs of the decision-makers in PICU.

The factorial survey as an approach to investigate clinical decision-making: examining influences on a clinician's decision to initiate life-sustaining clinical technology for a child with spinal muscular atrophy type 1.

Background: Spinal Muscular Atrophy (SMA) type 1 is a debilitating condition with a poor prognosis, though therapeutic advances are promising. Long-term ventilation is a common management strategy as respiratory function deteriorates. Without consensus on best practice respiratory management, the decision to initiate invasive LTV (I-LTV) for this group of young children involves many ethical considerations. Understanding the main influencing factors on a clinician's likelihood to initiative I-LTV for a child with chronic critical illness is important to maintain transparency and trust with the family during this challenging time. Methods: A factorial survey was used to identify the factors that influence a clinician to support initiation of I-LTV for children with SMA type 1. Factorial survey content was based on literature and evidence-based practice and the content was subject to extensive pretesting and pilot testing. An anonymous survey was disseminated (Oct 2021-Jan 2022), via eight international professional organisations, to clinicians with experience caring for children at the time of initiation of I-LTV. Results: 251 participants answered 514 vignettes on SMA type 1. The greatest influencing factor on clinician's likelihood to initiate I-LTV was parental agreement with the need to initiate I-LTV. Additional qualitative comments from participants support this finding. Clinicians also highlighted the important role of innovative therapies as well as the availability of supports for families when considering initiation however these findings were context based. Conclusions: The factorial survey approach provides a valuable way of identifying influencers on decision-making in sensitive situations. The findings demonstrate the acceptance of the centrality of parental influence in decisions on care delivery. Effective communication with the child's family is key to ensuring shared understanding and agreement of goals of care. More international research is needed on the long-term effects of novel treatments, as well as impact on quality of life and influence of geographical location, to inform decision-making.

Technology solutionism in paediatric intensive care: clinicians' perspectives of bioethical considerations.

BACKGROUND: The use of long-term life-sustaining technology for children improves survival rates in paediatric intensive care units (PICUs), but it may also increase long-term morbidity. One example of this is children who are dependent on invasive long-term ventilation. Clinicians caring for these children navigate an increasing array of ethical complexities. This study looks at the meaning clinicians give to the bioethical considerations associated with the availability of increasingly sophisticated technology. METHODS: A hermeneutic phenomenological exploration of the experiences of clinicians in deciding whether to initiate invasive long-term ventilation in children took place, via unstructured interviews. Data were analysed to gain insight into the lived experiences of clinicians. Participants were from PICUs, or closely allied to the care of children in PICUs, in four countries. RESULTS: Three themes developed from the data that portray the experiences of the clinicians: forming and managing relationships with parents and other clinicians considering, or using, life sustaining technology; the responsibility for moral and professional integrity in the use of technology; and keeping up with technological developments, and the resulting ethical and moral considerations. DISCUSSION: There are many benefits of the availability of long-term life-sustaining technology for a child, however, clinicians must also consider increasingly complex ethical dilemmas. Bioethical norms are adapting to aid clinicians, but challenges remain. CONCLUSION: During a time of technological solutionism, more needs to be understood about the influences on the initiation of invasive long-term ventilation for a child. Further research to better understand how clinicians, and bioethics services, support care delivery may positively impact this arena of health care.

Integrated care for children living with complex care needs: an evolutionary concept analysis.

Children with complex care needs (CCNs) are in need of improved access to healthcare services, communication, and support from healthcare professionals to ensure high-quality care is delivered to meet their needs. Integrated care is viewed as a key component of care delivery for children with CCNs, as it promotes the integration of healthcare systems to provide family and child-centred care across the entire health spectrum. There are many definitions and frameworks that support integrated care, but there is a lack of conceptual clarity around the term. Furthermore, it is often unclear how integrated care can be delivered to children with CCNs, therefore reinforcing the need for further clarification on how to define integrated care. An evolutionary concept analysis was conducted to clarify how integrated care for children with CCNs is defined within current literature. We found that integrated care for children with CCNs refers to highly specialised individualised care within or across services, that is co-produced by interdisciplinary teams, families, and children, supported by digital health technologies.  Conclusion: Given the variation in terms of study design, outcomes, and patient populations this paper highlights the need for further research into methods to measure integrated care. What is Known: • Children with complex care needs require long-term care, and are in need of improved services, communication, and information from healthcare professionals to provide them with the ongoing support they need to manage their condition. • Integrated care is a key component in healthcare delivery for children with complex care needs as it has the potential to improve access to family-centred care across the entire health spectrum. What is New: • Integrated care for children with CCNs refers to highly specialised individualised care within or across services, that is co-produced by interdisciplinary teams, families, and children, supported by digital health technologies. • There is a need for the development of measurement tools to effectively assess integrated care within practice.

Development of a factorial survey for use in an international study examining clinicians' likelihood to support the decision to initiate invasive long-term ventilation for a child (the TechChild study).

BACKGROUND: The decision to initiate invasive long-term ventilation for a child with complex medical needs can be extremely challenging. TechChild is a research programme that aims to explore the liminal space between initial consideration of such technology dependence and the final decision. This paper presents a best practice example of the development of a unique use of the factorial survey method to identify the main influencing factors in this critical juncture in a child's care. METHODS: We developed a within-subjects design factorial survey. In phase 1 (design) we defined the survey goal (dependent variable, mode and sample). We defined and constructed the factors and factor levels (independent variables) using previous qualitative research and existing scientific literature. We further refined these factors based on expert feedback from expert clinicians and a statistician. In phase two (pretesting), we subjected the survey tool to several iterations (cognitive interviewing, face validity testing, statistical review, usability testing). In phase three (piloting) testing focused on feasibility testing with members of the target population (n = 18). Ethical approval was obtained from the then host institution's Health Sciences Ethics Committee. RESULTS: Initial refinement of factors was guided by literature and interviews with clinicians and grouped into four broad categories: Clinical, Child and Family, Organisational, and Professional characteristics. Extensive iterative consultations with clinical and statistical experts, including analysis of cognitive interviews, identified best practice in terms of appropriate: inclusion and order of clinical content; cognitive load and number of factors; as well as language used to suit an international audience. The pilot study confirmed feasibility of the survey. The final survey comprised a 43-item online tool including two age-based sets of clinical vignettes, eight of which were randomly presented to each participant from a total vignette population of 480. CONCLUSIONS: This paper clearly explains the processes involved in the development of a factorial survey for the online environment that is internationally appropriate, relevant, and useful to research an increasingly important subject in modern healthcare. This paper provides a framework for researchers to apply a factorial survey approach in wider health research, making this underutilised approach more accessible to a wider audience.

The Meaning Given to Bioethics as a Source of Support by Physicians Who Care for Children Who Require Long-Term Ventilation.

The role and potential of bioethics input when a child requires the initiation of technology dependence to sustain life is relatively unknown. In particular, little is understood about the meaning physicians give to bioethics as a source of support during the care of children in pediatric intensive care who require long-term ventilation (LTV). We used a hermeneutic phenomenological approach to underpin the collection and analysis of data. Unstructured interviews of 40 physicians in four countries took place during 2020. We found that elements of trust, communication and acceptance informed the physicians' perceptions of the relationship with bioethics. These ranged from satisfaction to disappointment with their input into critical decisions. Bioethics services have potential to help physicians gain clarity over distressing and complex care decisions, yet physicians perceive the service inconsistently as a means of support. This research provides a sound basis to guide more beneficial interactions between clinicians and bioethics services.

Initiating technology dependence to sustain a child's life: a systematic review of reasons.

BACKGROUND: Decision-making in initiating life-sustaining health technology is complex and often conducted at time-critical junctures in clinical care. Many of these decisions have profound, often irreversible, consequences for the child and family, as well as potential benefits for functioning, health and quality of life. Yet little is known about what influences these decisions. A systematic review of reasoning identified the range of reasons clinicians give in the literature when initiating technology dependence in a child, and as a result helps determine the range of influences on these decisions. METHODS: Medline, EMBASE, CINAHL, PsychINFO, Web of Science, ASSIA and Global Health Library databases were searched to identify all reasons given for the initiation of technology dependence in a child. Each reason was coded as a broad and narrow reason type, and whether it supported or rejected technology dependence. RESULTS: 53 relevant papers were retained from 1604 publications, containing 116 broad reason types and 383 narrow reason types. These were grouped into broad thematic categories: clinical factors, quality of life factors, moral imperatives and duty and personal values; and whether they supported, rejected or described the initiation of technology dependence. The majority were conceptual or discussion papers, less than a third were empirical studies. Most discussed neonates and focused on end-of-life care. CONCLUSIONS: There is a lack of empirical studies on this topic, scant knowledge about the experience of older children and their families in particular; and little written on choices made outside 'end-of-life' care. This review provides a sound basis for empirical research into the important influences on a child's potential technology dependence.

Factors influencing the uptake of evidence in child health policy-making: results of a survey among 23 European countries.

BACKGROUND: The ability to successfully transfer knowledge across international boundaries to improve health across the European Region is dependent on an in-depth understanding of the many factors involved in policy creation. Across countries we can observe various approaches to evidence usage in the policy-making process. This study, which was a part of the Models of Child Health Appraised (MOCHA) project assessing patterns of children's primary care in Europe, focused on how and what kind of evidence is used in child health policy-making processes in European countries and how it is applied to inform policy and practice. METHOD: In this study, a qualitative approach was used. The data were analysed in accordance with the thematic analysis protocol. The MOCHA project methodology relies on experienced country agents (CA) recruited for the project and paid to deliver child health data in each of 30 European countries. CAs are national experts in the child health field who defined the country-specific structured information and data. A questionnaire designed as a semi-structured survey instrument asked CAs to indicate the sources of evidence used in the policy-making process and what needed to be in place to support evidence uptake in policy and practice. RESULTS: In our data we observed two approaches to evidence usage in child health policy formulation. The scientific approach in our understanding refers to the so-called bottom-up initiatives of academia which identify and respond to the population's needs. Institutional approaches can be informed by scientific resources as well; however, the driving forces here are governmental institutions, whose decisions and choices are based not only on the population needs but also on political, economic and organizational factors. The evidence used in Europe can also be of an external or internal nature. Various factors can affect the use of evidence in child health policy-making. Facilitators are correlated with strong scientific culture development, whereas barriers are defined by a poor tradition of implementing changes based on reliable evidence. CONCLUSIONS: Focusing on the facilitators and actively working to reduce the barriers can perceivably lead to faster and more robust policy-making, including the development of a culture of scientific grounding in policy creation.

A systematic concept analysis of 'technology dependent': challenging the terminology.

There are an increasing number of children who are dependent on medical technology to sustain their lives. Although significant research on this issue is taking place, the terminology used is variable and the concept of technology dependence is ill-defined. A systematic concept analysis was conducted examining the attributes, antecedents, and consequences of the concept of technology dependent, as portrayed in the literature. We found that this concept refers to a wide range of clinical technology to support biological functioning across a dependency continuum, for a range of clinical conditions. It is commonly initiated within a complex biopsychosocial context and has wide ranging sequelae for the child and family, and health and social care delivery.Conclusion: The term technology dependent is increasingly redundant. It objectifies a heterogenous group of children who are assisted by a myriad of technology and who adapt to, and function with, this assistance in numerous ways. What is Known: • There are an increasing number of children who require medical technology to sustain their life, commonly referred to as technology dependent. This concept analysis critically analyses the relevance of the term technology dependent which is in use for over 30 years. What is New: • Technology dependency refers to a wide range of clinical technology to support biological functioning across a dependency continuum, for a range of clinical conditions. It is commonly initiated within a complex biopsychosocial context and has wide-ranging sequelae for the child and family, and health and social care delivery. • The paper shows that the term technology dependent is generally portrayed in the literature in a problem-focused manner. • This term is increasingly redundant and does not serve the heterogenous group of children who are assisted by a myriad of technology and who adapt to, and function with, this assistance in numerous ways. More appropriate child-centred terminology will be determined within the TechChild project.

Approaches to the initiation of life-sustaining technology in children: A scoping review of changes over time.

Little is understood about the dynamic circumstances within which the initiation of technology dependence takes place in children. The aim of this scoping review was to identify the influences on the initiation of technology dependence and the issues that require further exploration and consideration. Scientific literature that directly or indirectly discussed the initiation of technology dependence in children was identified. A three-stage screening process of title and abstract scrutiny, full-text scanning and in-depth full-text reading resulted in 63 relevant articles from 1133 initially reviewed. These were then subjected to descriptive and thematic analysis. Articles ranged from the 1970s to the present, reflecting the evolution of ethical debates around the approaches to clinical practice and changes in cultural and societal attitudes. Three themes emerged: how technology alters the meaning of futile care, dissonance in the perspectives of decision makers and increasing support for joint decision-making. Only articles in English and predominantly from the clinician's rather than the patient's perspective were included. Societal and cultural factors as well as the structural, financial and cultural environment influence the initiation of technology dependence in children. However, to what extent these overt and implicit influences guide decision-makers in this field remains largely unknown.

Tailored communication methods as key to implementation of evidence-based solutions in primary child health care.

BACKGROUND: Evidence-based policies should underpin successful implementation of innovations within child health care. The EU-funded Models of Child Health Appraised project enabled research into effective methods to communicate research evidence. The objective of this study was to identify and categorize methods to communicate evidence-based research recommendations and means to tailor this to stakeholder audiences. METHODS: We conducted an online survey among national stakeholders in child health. Analysis of the most effective strategies to communicate research evidence and reach the target audience was carried out in order to ensure implementation of optimal child health care models at a national level. RESULTS: Representatives of stakeholders from 21 of the then 30 EU MS and EEA countries responded to the questionnaire. Three main approaches in defining the strategies for effective communication of research recommendations were observed, namely: dissemination of information, involvement of stakeholders and active attitude towards change expressed in actions. The target audience for communicating recommendations was divided into two layers: proximal, which includes those who are remaining in close contact with the child, and distal, which contains those who are institutionally responsible for high quality of child health services. They should be recipients of evidence-based results communicated by different formats, such as scientific, administrative, popular and personal. CONCLUSIONS: Influential stakeholders impact the process of effective research dissemination and guide necessary actions to strengthen the process of effective communication of recommendations. Communication of evidence-based results should be targeted to each audience's profile, both professional and non-professionals, by adjusting appropriate communication formats.

Do European Union countries adequately address the healthcare needs of adolescents in the area of sexual reproductive health and rights?

BACKGROUND AND OBJECTIVES: Adolescent sexual and reproductive health and rights (SRHR) are of particular relevance given their potential short-term or long-term health consequences. This study evaluates recommendations and policies regarding access to care in this area in 31 European countries (European Union (EU) plus Iceland, Norway and Switzerland). METHODS: As part of the EU funded Models of Child Health Appraised project, data were gathered using a 43-item questionnaire sent to experts responsible for collecting information in each country. RESULTS: Ten countries have not developed any formal policy or recommendation that guarantee the respect of confidentiality and the possibility of consulting a physician without parents knowing. Nearly half of the countries do not have centres specialised in adolescent healthcare, tackling comprehensive health issues or focusing specifically on SRH. Access to emergency contraception and information regarding pregnancy, including testing, is easy in most countries. However, oral contraception is delivered free of charge in only 10 countries. Twenty-three countries do not meet current standards in terms of providing policy-based pregnancy care, and only 13 have set up special programmes for pregnant adolescents. In only seven countries can adolescents definitely have their pregnancy terminated without their parents knowing (and in another seven countries in selected situations). CONCLUSION: The provision and availability of adolescent-friendly SRHR care are far from optimal in around half of the surveyed countries. These results call for the review and implementation of policies, specialised healthcare centres and training initiatives for primary care providers.

Contextual determinants of CHILDREN'S health care and policy in Europe.

BACKGROUND: The main objective of this study was to explore the contextual determinants of child health policies. METHODS: The Horizon 2020 Models of Child Health Appraised (MOCHA) project has one Country Agent (CA) in all 30 EU and EEA countries. A questionnaire designed by MOCHA researchers as a semi-structured survey instrument asked CAs to identify and report the predominating public and professional discussions related to child health services within the last 5 years in their country and the various factors which may have influenced these. The survey was issued to CAs following validation by an independent Expert Advisory Board. The data were collected between July and December 2016. The data was qualitatively analysed using software Nvivo11 for data coding and categorization and constructing the scheme for identified processes or elements. RESULTS: Contextual determinants of children's health care and policy were grouped into four categories. 1) Socio-cultural determinants: societal activation, awareness, communication, trust, freedom, contextual change, lifestyle, tolerance and religion, and history. 2) Structural determinants which were divided into: a) external determinants related to elements indirectly correlated with health care and b) internal determinants comprising interdependent health care and policy processes. 3) International determinants such as cross-nationality of child health policy issues. 4) The specific situational determinants: events which contributed to intensification of debates which were reflected by behavioural, procedural, institutional and global factors. CONCLUSIONS: The influence of context across European countries, in the process of children's health policy development is clearly evident from our research. A number of key categories of determinants which influence child health policy have been identified and can be used to describe this context. Child health policy is often initiated in reaction to public discontentment. The multiple voices of society resulted, amongst others, in the introduction of new procedures, action plans and guidelines; raising levels of awareness, intensifying public scrutiny, increasing access and availability of services and provoking introduction of structural changes or withdrawing unfavourable changes.

Human papillomavirus vaccination and respect for children's developing autonomy: Results from a European Union wide study.

Children's rights to autonomy of choice are differently expressed throughout Europe. We explored differences regarding expressions of respect for children's autonomy throughout Europe, using the procedure of human papillomavirus (HPV) vaccination offer as indicator. We used a mixed methods approach, utilizing an expert survey within the frame of "Models of Child Health Appraised" (MOCHA), among all 30 European Union (EU) and European Economic Area states. A questionnaire was designed using vignettes regarding the vaccine provision. Thirty MOCHA country agents were invited to respond from June 2017 to April 2018. In total, 28 country agents responded. We studied the following themes: (i) provision of informed consent, (ii) parental and medical paternalism, (iii) relevance of the child's chronological age or maturity, and (iv) vaccination programs targeting boys. These are being handled differently across the region. We explored associations of these implemented practices with the national vaccine coverage rate across Europe. We used the processes of HPV vaccination to study child's autonomy, the paradigm change toward libertarian paternalism and issues of sex-equity. Interestingly, greater respect for children's autonomy tends to be associated with medium or high vaccination coverage rates and lower respect with lower rates. Respect and empowerment seem to have practical as well as moral benefits. Identifying and transferring the most suitable ethical approaches is crucial and should be strengthened.

How does societal reaction to children's health issues contribute to health policy in Europe? Results of a survey.

BACKGROUND: In the European context the awareness of societal responsibility for children's health has increased with greater attention to children's rights and child empowerment processes. Child health issues are considered particularly sensitive; thus, they often provoke strong societal reactions, which, as a consequence, influence national health policies across Europe. Effectiveness of societal influences increases with the involvement of various actors in the context. METHODS: A qualitative approach was used to identify the level of societal involvement in health decision-making. A questionnaire was sent to the Country Agents (CAs) of the Models of Child Health Appraised (MOCHA) project. CAs are contact points in each of the 30 participating in the project countries and were asked to identify strong public and professional discussions related to child health services in their countries. Data collection was undertaken between July and December 2016. RESULTS: Based on 71 case studies, we identified eight thematic patterns, which characterize societal reactions to the currently worrisome child health issues across Europe. We devoted our attention to the three most controversial: child vaccination, child poverty and child abuse. The cases described by the CAs show the broad perspective in the perception of child health problems. Child health issues involve the public and raise nationwide debates. Public concerns were directly or indirectly related to child health and depicted the national overtone. CONCLUSIONS: Concerns in Europe about child health care are twofold: they are devoted to systemic issues (indirect patient orientation) and to child health and well-being (direct patient orientation). The phenomenon of societal responsibility for children's health is important for the support of public acceptance of child health policy.

A Heuristic Governance Framework for the Implementation of Child Primary Health Care Interventions in Different Contexts in the European Union.

To adopt and implement innovative good practices across the European Union requires developing policies for different political and constitutional contexts. Health policies are mostly decided by national political processes at different levels. To attain effective advice for policy making and good practice exchange, one has to take different models of governance for health into account. We aimed to explore which concepts of governance research are relevant for implementing child health policies in a European Union context. We argue that taking into account the insights of good intersectoral and multilevel governance in research and practice is essential and promising for future analyses. These governance concepts help to understand what actors and institutions are potentially of relevance for developing and implementing child-centric health care approaches not only within health care but also outside health care. The framework we developed has the potential to advise on and thus support effectively the spreading and implementation of good practices of child-centric health policy approaches across the European Union. With this heuristic framework, the variety of relevant stakeholders and institutions can better be mapped and taken into account in implementation processes. Also, the normative side-particularly stressing values that make governance "good governance"-is to be taken into account.

Management and integration of care for children living with complex care needs at the acute-community interface in Europe.

With improvements in neonatal and paediatric care, more children living with complex care needs are surviving beyond infancy into late childhood and adulthood than in the past. We examined the current approach to the management and integration of care of children living with complex care needs in 30 European countries, as well as the implications for primary care service delivery. This descriptive study, with an embedded qualitative aspect, consisted of questions adapted from the Standards for Systems of Care for Children and Youth with Special Health Care Needs, and included questions on a complex care European survey of change, adapted from the Eurobarometer survey. The analysis indicates that few systems are in place in countries across Europe to identify all the health-care providers who deliver care to a child living with complex care needs, and that less than half of all countries surveyed have policies in place to support care coordination for these children. Primary care physicians have little involvement in care planning for children before their discharge to the community setting, and there is little parental participation in policy development. Access to, and governance of, care for these children varies widely. Particular issues identified pertain to parents as catalysts of optimum integration of care, pathways to specialist care, and the need for comprehensive national integrated care programmes.

Principles for provision of integrated complex care for children across the acute-community interface in Europe.

This Viewpoint presents and discusses the development of the first core principles and standards for effective, personalised care of children living with complex care needs in Europe. These principles and standards emerged from an analysis of data gathered on several areas, including the integration of care for the child at the acute-community interface, the referral-discharge interface, the social care interface, nursing preparedness for practice, and experiences of the child and family. The three main principles, underpinned by a child-centric approach, are access to care, co-creation of care, and effective integrated governance. Collectively, the principles and standards offer a means to benchmark existing services for children living with complex care needs, to influence policy in relation to service delivery for these children, and to provide a suite of indicators with which to assess future service developments in this area.