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 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.

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.

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.

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.

Barriers to effective, safe communication and workflow between nurses and non-consultant hospital doctors during out-of-hours.

OBJECTIVE: This study aimed to evaluate the nature and type of communication and workflow arrangements between nurses and doctors out-of-hours (OOH). Effective communication and workflow arrangements between nurses and doctors are essential to minimize risk in hospital settings, particularly in the out-of-hour's period. Timely patient flow is a priority for all healthcare organizations and the quality of communication and workflow arrangements influences patient safety. DESIGN: Qualitative descriptive design and data collection methods included focus groups and individual interviews. SETTING: A 500 bed tertiary referral acute hospital in Ireland. PARTICIPANTS: Junior and senior Non-Consultant Hospital Doctors, staff nurses and nurse managers. RESULTS: Both nurses and doctors acknowledged the importance of good interdisciplinary communication and collaborative working, in sustaining effective workflow and enabling a supportive working environment and patient safety. Indeed, issues of safety and missed care OOH were found to be primarily due to difficulties of communication and workflow. Medical workflow OOH is often dependent on cues and communication to/from nursing. However, communication systems and, in particular the bleep system, considered central to the process of communication between doctors and nurses OOH, can contribute to workflow challenges and increased staff stress. It was reported as commonplace for routine work, that should be completed during normal hours, to fall into OOH when resources were most limited, further compounding risk to patient safety. CONCLUSION: Enhancement of communication strategies between nurses and doctors has the potential to remove barriers to effective decision-making and patient flow.

The relationship between body mass index and health-related quality of life in urban disadvantaged children.

INTRODUCTION: The study's aim was to establish, for children living in urban disadvantage, the nature and extent of the relationship between body mass index (BMI) and health-related quality of life (HRQoL), including the role of individual and family factors in influencing this relationship. METHODS: Within the context of a longitudinal design, 255 children aged 7-12 years (50 % male) self-reported their HRQoL (Kidscreen-27) and had their height and weight measured at year one and two. One parent/guardian for each child was also assessed at year one with the OSLO Social Support Scale and Hospital Anxiety and Depression Scale. Regression analysis was also conducted. RESULTS: BMI was weakly inversely associated with 'total HRQoL' (r = -.15, p < .05), 'physical well-being' and 'autonomy and parent relations'. Significant differences were found between normal weight and obese children on all but the latter dimension. Neither weight group, however, fell below the average European HRQoL range. BMI predicted physical well-being a year later and vice versa, whilst autonomy and parent relations also predicted BMI a year later. In terms of 'overweight' children (38 %), those approaching adolescence had poorer physical and school well-being than younger children, and those whose parents had moderate-to-severe levels of depression fared worse on school well-being than children whose parents were not depressed. CONCLUSION: The findings suggest that obesity programmes could aim to prevent/reduce obesity and optimise HRQoL in urban disadvantaged preadolescent children whilst also targeting parental mental health difficulties. Future research should examine mediators of the effect of BMI on HRQoL.