Susie Aldiss

BACKGROUND: Families and professionals caring for a child without a definitive diagnosis face unique challenges, particularly in relation to: managing uncertainty; access to healthcare; obtaining relevant information and support; and trying to navigate a healthcare system that is often fragmented. We used co-design to inform the establishment of the first UK specialist nursing post dedicated to working with children with undiagnosed genetic conditions and their families. OBJECTIVES: 1) To understand what families and hospital staff want from the service; 2) To understand how the post should be operationalised in practice; 3) To develop the job description and person specification for the post-holder. METHODSA range of approaches were used to collect data: interviews (nine parents and 10 hospital staff); a focus group (three parents); a creative workshop (six patients and siblings); and an online forum (81 parents). Data were analysed using framework and thematic analysis. The strands of data were brought together and reviewed as a whole to formulate the post-holder's job description and person specification. RESULTS: Stakeholders identified nine key elements to the role which were incorporated into the job description: practical support; point of contact; community liaison; signposting to other services; care coordination; supporting families; advocacy; raising awareness; and emotional support. CONCLUSIONS: Highlighted in this paper are the practical aspects of engaging and involving all relevant stakeholders in the process of co-designing a new post and subsequent staff recruitment. The flexibility employed in the setting and methods of data collection were instrumental in ensuring the views of a diverse range of participants were ascertained. A major consideration is the resources required to undertake co-design, in terms of time and finances. We believe that the resources required for the co-design are offset by the advantages of having the right person in the right post, doing the right job.

OBJECTIVEWe conducted a UK-wide survey to identify the top 10 research questions for young people's cancer. We conducted secondary analysis of questions submitted, which were 'out-of-scope' of the original survey aim. We sought to disseminate these questions, to inform practice, policy and the development of potential interventions to support young people with cancer. DESIGNJames Lind Alliance Priority Setting Partnership. PARTICIPANTSYoung people aged 13-24 with a current/previous cancer diagnosis, their families/friends/partners and professionals who work with this population. METHODSEight hundred and fifty-five potential research questions were submitted, and 326 were classified as 'out-of-scope'. These questions, along with 49 'free-text' comments, were analysed using thematic analysis. RESULTSThe 375 out-of-scope questions and comments were submitted by: 68 young people, 81 family members/partners/friends and 42 professionals. Ten overarching themes were identified: diagnostic experience; communication; coordination of care; information needs and lack of information; service provision; long-term effects and aftercare support; family support; financial impact; end-of life care; and research methods and current research. CONCLUSIONSThe need to tailor services, information and communication is a striking thread evidenced across the 'out-of-scope' questions. Gaps in information highlight implications for practice in revisiting information needs throughout the cancer trajectory. We must advocate for specialist care for young people and promote the research priorities and these findings to funding bodies, charities, young people and health and social care policymakers, in order to generate an evidence base to inform effective interventions across the cancer trajectory and improve outcomes. PATIENT/PUBLIC CONTRIBUTIONSPatients and carers were equal stakeholders throughout.

Objective: The objective of this scoping review is to identify and map support interventions provided during palliative care to families with dependent children when a parent has a terminal illness. Introduction: For dependent children, losing a parent through terminal illness can be one of the most traumatic experiences they will face. Without age-appropriate information and support, parental death can lead to unresolved grief, post-traumatic stress, and longer-term negative social, emotional, educational, and behavioral consequences. Support from health care professionals is provided, but challenges within hospice and community palliative care mean the needs of dependent children may not be addressed. Greater emphasis is often placed on children's and families’ bereavement needs as opposed to their pre-bereavement needs. The status of pre-bereavement support in palliative care for these families has not been documented. Inclusion criteria: We will include studies of support interventions provided to families with dependent children when a parent has a terminal illness that are delivered during palliative care and in settings including, but not limited to, hospices, hospitals, and the community (including family and care homes). Empirical, peer-reviewed studies based on qualitative, quantitative, or mixed methods data, and gray literature, reporting any stage of intervention development will be included. Methods: This review will follow the JBI approach for scoping reviews. Multiple database searches will be conducted. Two authors will independently review full-text articles and extract data. Interventions will be mapped in terms of characteristics, evaluation methods, and implementation challenges. Data will be presented in tables, diagrammatic form, and narrative summary.

Purpose: The overall aim was to evaluate the key worker role across principal treatment centres for children with cancer in England, Wales and Scotland.Methods: Mixed-methods case study gathering data from multiple perspectives using questionnaires, interviews, focus groups and reports/performance documents over a two-year period. Framework approach was adopted to analyse transcripts and documentary data. Results: Participants included: 22 nurse specialist key workers, 103 parents, 85 professionals and 10 children/young people. Qualitative and quantitative data were weaved together, to best illuminate key worker services. Four main models of care were described as well as the context of care and process of care.  Key working effectiveness centred around three pillars: care coordination; expert knowledge, experience and expertise; relationship. These were essential to improved family experience, emotional wellbeing, and delivery of individualised care closer to home. Conclusions: The role is complex and diverse, responding to local needs. Certain conditions, (e.g. high caseload) placed limits on enacting the three pillars, diminishing the positive experience of families. When they worked well, key workers reduced the fragmented nature of services and families placed great value on keeping the same key worker from diagnosis into long-term care. Retaining these roles, where already in place, or including, if not, we would recommend, factoring into budgets to sustain and expand such roles.

Families and healthcare professionals caring for a sick or disabled child without a definitive diagnosis face unique challenges, particularly in relation to managing uncertainty, access to healthcare and coordination of care. There has been little research exploring the impact this has on families, their support needs or their experience of health services. This qualitative interview study included interviews with 14 mothers of children with undiagnosed genetic conditions. Transcripts were analysed using thematic analysis. Four themes emerged, uncovering overlapping patterns in the data: (1) living with complexity amidst uncertainty-'We don't know what tomorrow will bring'; (2) parental role-'I do everything I can'; (3) parental role-'Not coping is not an option'; and (4) support needs-'There's lots of help that just isn't out there'. The results clearly demonstrate the stresses faced when caring for a child with an undiagnosed genetic condition. Some themes are shared with the experience of other families caring for children with complex needs. However, parents were doing all they could for their child in the context of a life of uncertainty, with the absence of a clear diagnosis clearly causing additional stress that impacted on the whole family. Impact on their emotional and physical well-being was evident; they described times of feeling stressed, worried and anxious. They were confused due to being overloaded with information and frustrated by a lack of care coordination. Parents did not appear to prioritize their own well-being and held back their emotions to protect themselves and others. As a result, they had many unmet needs, particularly relating to emotional support.

Background Both under- and overnutrition may have adverse impact on outcome of cancer in children and teenagers/young adults (CTYA). Previous studies confirm that screening for nutritional risk and detection of nutritional abnormality is inconsistently undertaken in practice. Methods We undertook a survey of dietetic resource and nutritional assessment in CTYA principal treatment centres (PTC) in the United Kingdom. Responses were received from 95% children's and 69% TYA PTC. Results Only 13/18 (72%) children's PTC, and one of 11 (9%) TYA PTC, met national standards for dietetic resource; one of 18 (6%) paediatric and six of 11(54%) TYA PTC had no such resource. While resources were greater in larger paediatric PTC, who were also most likely to undertake stem cell transplantation, resources in TYA PTC were too low to assess relationship to centre size. Most centres focused resources on inpatient care and

Families and healthcare professionals caring for a sick or disabled child without a definitive diagnosis face unique challenges, particularly in relation to managing uncertainty, access to healthcare and coordination of care. There has been little research exploring the impact this has on families, their support needs or their experience of health services. This qualitative interview study included interviews with 14 mothers of children with undiagnosed genetic conditions. Transcripts were analysed using thematic analysis. Four themes emerged, uncovering overlapping patterns in the data: (1) living with complexity amidst uncertainty-'We don't know what tomorrow will bring'; (2) parental role-'I do everything I can'; (3) parental role-'Not coping is not an option'; and (4) support needs-'There's lots of help that just isn't out there'. The results clearly demonstrate the stresses faced when caring for a child with an undiagnosed genetic condition. Some themes are shared with the experience of other families caring for children with complex needs. However, parents were doing all they could for their child in the context of a life of uncertainty, with the absence of a clear diagnosis clearly causing additional stress that impacted on the whole family. Impact on their emotional and physical well-being was evident; they described times of feeling stressed, worried and anxious. They were confused due to being overloaded with information and frustrated by a lack of care coordination. Parents did not appear to prioritize their own well-being and held back their emotions to protect themselves and others. As a result, they had many unmet needs, particularly relating to emotional support.

The transition from child to adult services is a crucial time in the health of young people who may potentially fall into a poorly managed 'care gap'. A multi-site, multi-staged study was undertaken to identify the key aspects of a transitional programme of care for young people. Through a process of mapping, which involved drawing on primary and secondary data, a clinical practice-benchmark tool was developed. Benchmarks are a health care quality performance measurement 'tool'. They provide clinical teams with standards that services can measure themselves against to see how they are doing. They are used in a comparing and sharing activity, using a structured and systematic approach, to share best practice. They offer a mechanism to look at processes, and provide an opportunity to analyse skills and attitudes, which may be the hidden narrative in benchmarking. This paper describes steps in the development of benchmarks for transition to adult care, often associated with low patient and family satisfaction. Qualitative data were collected through focus groups, workshops and interviews from 13 young people with long-term health conditions, 11 parents, 36 professionals and 21 experts leading on transition within the United Kingdom. Transcripts were analysed using qualitative content analysis. For young people and their parents/carers to experience timely and effective transition, eight factors and their associated indicators of best practice were developed from the primary and secondary data and refined through an iterative process. We recommend their use to clinical teams to inform system level strategies as well as evaluation programmes.

Adolescents and young adults (AYA) face individual and social challenges different to other age groups that shape their experience of cancer and the associated psychological distress. This critical review examined the availability of AYA-specific psychosocial interventions in order to assess the impact they have and identify elements that make them successful. Five literature databases were searched for psychosocial intervention studies involving AYAs with cancer, on and off-treatment, aged 10-30 years, published between 1980 and 2016. Eleven out of 42 identified studies were included in the final review after quality assessment. All but two interventions showed positive results on at least one measure of psychosocial wellbeing. A distinctive difference between the supportive needs of those on and off-treatment was noted, and differences in intervention design and delivery formats were identified between the two sub-groups. Analysis of specific intervention characteristics that achieved the successful outcomes was more challenging due to the large variation in outcome measures used. This review demonstrates that psychosocial intervention research for AYA with cancer is increasing and the results show a potential benefit of intervention participation. Further work is needed to validate whether the benefits achieved by intervention participation remain over time.

Background: Technology is becoming an important medium for supporting patients in health care. However, successful application depends on user acceptability. The Advanced Symptom Management System (ASyMS©) involves patients reporting cancer chemotherapy-related symptoms using mobile phone technology. Objective: The aim of this article was to report a study of how young people were involved in the development of ASyMS© using the Medical Research Council framework for evaluating complex interventions. Methods: A convenience sample of young people aged 13 to 18 years undergoing cancer chemotherapy were recruited from 2 principal cancer treatment centers in London. Results: In phase 1, young people selected 5 symptoms from an adapted version of the Memorial Symptom Assessment Scale that were most important to them. In phase 2, young people completed the ASyMS©-YG PDA (personal digital assistant) questionnaire daily on days 1 to 14 of a cycle of chemotherapy and pre/post-use questionnaires. In phase 1, 5 young people chose diarrhea, nausea, vomiting, constipation, and weight loss as the most important symptoms. In phase 2, 25 young people reported positively to using PDA technology, found ASyMS©-YG simple and easy to complete, and liked that they were monitored at home. In addition to the 5 core symptoms, the ASyMS©-YG reports showed the number (n = 37) of other symptoms young people experienced. CONCLUSIONS:: This early development work indicates the acceptability of ASyMS©-YG and has informed an exploratory trial (phase 3) and randomized controlled trial (stage 4). Implications for practice: This study reaffirms the importance of promoting communication between young people and health professionals. Copyright © 2010 Lippincott Williams & Wilkins.

Background There is evidence that the process of transition from paediatric (child) to adult health services is often associated with deterioration in the health of adolescents with chronic conditions.Transitional care is the term used to describe services that seek to bridge this care gap. It has been defined as ‘the purposeful, planned movement of adolescents and young adults with chronic physical and medical conditions from child-centred to adult-oriented health care systems’. In order to develop appropriate services for adolescents, evidence of what works and what factors act as barriers and facilitators of effective interventions is needed. Objectives To evaluate the effectiveness of interventions designed to improve the transition of care for adolescents from paediatric to adult health services. Search methods We searched The Cochrane Central Register of Controlled Trials 2015, Issue 1, (including the Cochrane Effective Practice and Organisation of Care Group Specialised Register), MEDLINE, EMBASE, PsycINFO, and Web of Knowledge to 19 June 2015. We also searched reference lists of included studies and relevant reviews, and contacted experts and study authors for additional studies. Selection criteria We considered randomised controlled trials (RCTs), controlled before- and after-studies (CBAs), and interrupted time-series studies (ITSs) that evaluated the effectiveness of any intervention (care model or clinical pathway), that aimed to improve the transition of care for adolescents from paediatric to adult health services. We considered adolescents with any chronic condition that required ongoing clinical care, who were leaving paediatric services and going on to receive services in adult healthcare units, and their families. Participating providers included all health professionals responsible for the care of young people.

Purpose: To describe the development and implementation of the specialist nurse key worker  role across 18 children’s cancer centres in the United Kingdom, and draw out significant factors  for success to inform future development of the role across a range of specialities.  Method: Data were obtained through 42 semi‐structured interviews and a focus group with 12  key workers. Framework analysis revealed two main themes: models of care and key workers’  perspectives of the role.  Results: Four models of care were identified and described, roles were organised along a  continuum of in reach and outreach with either the presence or absence of home visits and  direct delivery of clinical care. Key workers’ perspectives of the advantages of the role included:  coordination of care (being the main point of contact for families/professionals), experience  and expertise (communication/information) and the relationship with families. The main  challenges identified were: time, caseload size, geographical area covered, staffing numbers  and resources available in the hospital and community.  Conclusion: The label ‘key worker’ was disliked by many participants, as the loss of ‘specialist  nurse’ in the title failed to reflect professional group. Leaving aside terminology, key workers  shared core role elements within a continuum of in reach and outreach work and their  involvement in direct clinical care varied throughout the pathway. Irrespective of the model  they worked in, the key worker provided clinical, emotional, educational, and practical support  to families, through the coordination of care, experience and expertise and relationship with  families and professionals.

BACKGROUND: Little is known about whether children's cancer services actually meet children's needs, as the majority of previous research has sought the views of parents as proxies. OBJECTIVES: To explore children's and young peoples' views of cancer care and to present a conceptual model of communication and information sharing. SETTINGS: Three Principal Cancer Treatment centres in the United Kingdom. PARTICIPANTS: Thirty-eight participants at different stages of the cancer journey (e.g. on treatment, near end of treatment, up to 18 months following treatment) were grouped for data collection by age: young children (4-5 years), older children (6-12 years) and young people (13-19 years). METHODS: Data were collected concurrently over 6 months using age-appropriate, participatory-based techniques. Techniques included play and puppets, the draw and write method, interviews and an activities day. RESULTS: Some findings confirmed previously reported issues, such as, young children's inabilities to voice their preferences, and the importance of familiar environments and parental support for all ages. New findings suggested children worry about the permanence of symptoms, and older children are unhappy about their parents leading communications with health professionals. As communication and information sharing featured as an area in which children's and young people's preferences changed most dramatically, we propose a conceptual model of communication roles of patients, their parents, and health professionals to illuminate communication patterns. The model suggests children (aged 4-12 years) reside in the background of information sharing with health professionals until they gain autonomy as young people (around age 13). They then move into the foreground, and their parents transition into a supportive background role. Reviewing this model may help younger children realise their abilities to voice their preferences and older children to move into the foreground. Parents and professionals, in turn, can learn to develop in their supportive background roles. We encourage further testing of the model to define roles within relevant contexts. CONCLUSIONS: Overall, this study offers a perspective on the needs and preferences of children and young people receiving cancer care. Differences across ages were more striking with reference to preferences for communication. These differences are highly relevant to the way we communicate information to children and young people.

This study aimed to: 1) explore views of known experts leading on transition, 2) gather insights on the essential features of transitional care models, 3) highlight opportunities for and barriers to change: to inform core elements of benchmarks for transitional care. We held three workshops (n=20), and used a telephone interview (n=1) with health and social care professionals with expertise working with young people with a range of health conditions and disabilities. The workshops included individual brainstorming, and group discussion. Data were analysed using qualitative content analysis. The general consensus from stakeholders’ discussions about transition was that, ‘things have become stuck’. Themes included: professionals’ attitudes towards and knowledge about young people and transition, organisational barriers and ‘lack of joined up thinking’ between services. Our work offers further insight into experts’ perceptions of transition services within the United Kingdom. It is clear there is still much to be done to improve transition, to better meet the needs of young people and parents and begin to offer equitable access to transitional care programmes. The benchmarks offer a starting point for professionals seeking to improve transition through enabling the identification of gaps in services and providing a platform to share successful practice initiatives.

Background: Unintentional injuries are a leading cause of preventable death and a major cause of ill health and disability in children under five years of age. A health promotion mobile phone application, ‘Grow up Safely’, was developed to support parents and carers in reducing unintentional injuries in this population of children. Methods: A prototype of the mobile application was developed to deliver health education on unintentional injury prevention linked to stages of child development. In order to explore the usability of the app and refine its content, three focus groups were conducted with 15 mothers. Data were analysed using thematic analysis. Results: The majority of participants reported previous use of health apps, mainly related to pregnancy, and recommended by health professionals. The app was considered user‐friendly and easy to navigate. Participants in two focus groups found the app informative, offered new information and they would consider using it. Participants in the ‘young mum's’ group considered the advice to be ‘common sense’, but found the language too complex. All participants commented that further development of push‐out notifications and endorsement by a reputable source would increase their engagement with the app. Conclusion: The ‘Grow Up Safely’ mobile phone app, aimed at reducing unintentional injuries in children under five, was supported by mothers as a health promotion app. They would consider downloading it, particularly if recommended by a health professional or endorsed by a reputable organisation. Further development is planned with push‐out notifications and wider feasibility testing to engage targeted groups, such as young mothers, fathers and other carers.

Researchers are responsible for using techniques that allow children to contribute their perspectives in a way that is most suited to their strengths and preferences. The draw and write technique is a method that needs to be used carefully and sensitively if children are to become active participants in revealing their world as they see it. In this article, we explore the issues for the child and researcher of using the draw and write technique as a participatory research methodology. Reflections on the technique are drawn from our experience of using it with children aged 6 to 12 years. This was part of a larger study undertaken to enable children aged 4 to 18 years with a diagnosis of cancer to give their views about hospital care. The participating children's verbal and artistic contributions illustrate how children are able to convey their opinions when there is an enabling climate created.

Background: The transition from child to adult services is a crucial time in the health of young people who may potentially fall into a poorly managed 'care gap'. Health service provision, which fails to meet the needs of young people and families at this time of significant change, may result in deterioration in health or disengagement with services, which can have negative long-term consequences. Developing transitional care packages has become a focus of activity in the United Kingdom and elsewhere. Indeed policy documents have been trying to guide practice for many years, with some variable success. There is much work still to be done, particularly around how guidance and the sharing of best practice, when combined can result in a change practice. Objective: This study aimed to explore the views of professionals involved in transitional care, the process of transition in their services, the barriers and facilitators to transition. Methods: This was a qualitative study using focus group methodology. Four focus groups were carried out, attended by 36 health professionals across child and adult services. They had expertise in working with young people with various health conditions and disabilities. Transcripts were analysed using qualitative content analysis. Results: Eight key factors that impact on transition emerged from the data. These included factors associated with the patient group (such as age, health condition, having complex needs) as well as factors associated with services (such as the availability of equivalent services within adult care and the links between child and adult team). Conclusion: It is imperative that health professionals consider the population they are working with when planning transitional care and take into account the factors which can lead to delayed transition so that this can be avoided if possible. Numerous examples of initiatives to facilitate more timely transition were shared: these have been reflected in our ‘Benchmarks for Transition from Child to Adult Health Services’. We offer these benchmarks to inform and guide the practice of others, and illustrate their potential for use in the context of the findings shared here.

ASyMS© is an Advanced Symptom Management System utilising mobile phone technology to monitor chemotherapy-related symptoms and promote self-care. It was first developed with an adult cancer population and is now being evaluated with young people through an iterative development process. ASyMS© involves patients recording and sending symptom reports to the hospital and receiving tailored self-care advice. Health professionals are alerted when severe symptoms are reported. Three phases of the ASyMS©-YG (young people) study are complete. Phase 1 involved young people identifying the symptoms to be assessed. Phase 2 involved young people testing the symptom report system, and ascertaining young people's, parents' and professionals' perceptions of ASyMS©-YG. This paper reports on Phase 3, in which the system was developed further in preparation for a randomised controlled trial (RCT). Health professionals devised an alert system based on risk modelling side-effect severity, and young people and professionals developed self-care advice. A pilot study was conducted to test the alert system and study design in readiness for a definitive RCT. The contribution young people and professionals have made to this project is invaluable in evaluating the practical effectiveness of ASyMS©-YG and ensuring the intervention is acceptable and works in everyday practice. © The Author(s) 2010.

Objectives To conduct a UK-wide survey of young people who have experienced cancer, carers and professionals, to identify and prioritise research questions to inform decisions of research funders and support the case for research with this unique cancer population. Design James Lind Alliance Priority Setting Partnership. Setting UK health service and community. Methods A steering group oversaw the initiative and partner organisations were recruited. Unanswered questions were collected in an online survey. Evidence searching verified uncertainties. An interim survey was used to rank questions prior to a final prioritisation workshop. Participants Young people aged 13–24 years with a current or previous cancer diagnosis, their families, friends, partners and professionals who work with this population. Results Two hundred and ninety-two respondents submitted 855 potential questions. Following a refining process and removal of ‘out of scope’ questions, 208 unique questions remained. Systematic evidence checking identified seven answered questions and 16 were the subject of ongoing studies. The interim survey was completed by 174 participants. The top 30 questions were prioritised at a workshop attended by 25 young people, parents and multidisciplinary professionals. The top three priorities are: (1) What psychological support package improves psychological well-being, social functioning and mental health during and after treatment? (2) What interventions, including self-care, can reduce or reverse adverse short-term and long-term effects of cancer treatment? (3) What are the best strategies to improve access to clinical trials? The remaining questions reflect the complete cancer pathway: new therapies, life after cancer, support, education/employment, relapse and end-of-life care. Conclusions We have identified shared research priorities for young people with cancer using a rigorous, person-centred approach involving stakeholders typically not involved in setting the research agenda. The breadth of priorities suggest future research should focus on holistic and psychosocial care delivery as well as traditional drug/biology research.