Stakeholder Perceptions of Dignity Therapy for Children and Young People with Life-Limiting and Life-Threatening Conditions in the UK

In palliative and end of life care settings supporting people to make sense of their lives is as important as managing disease symptoms. Dignity Therapy is a validated psychotherapeutic intervention designed to bring about a sense of meaning and purpose for individuals at the end of life. Dignity Therapy has primarily been implemented and evaluated in adult palliative care settings. Prior to designing studies that evaluate Dignity Therapy for children and young people with life-limiting and life-threatening conditions, it is essential to establish stakeholders’ perceptions of Dignity Therapy, and whether adaptations would be required OBM Integrative and Complementary Medicine 2020; 5(1), doi:10.21926/obm.icm.2001012 Page 2/19 for this client group. We aimed to explore the acceptability of Dignity Therapy for children and young people with life-limiting and life-threatening conditions and health professionals, and report findings from our stakeholder activities. Stakeholder consultation activities involved one Death Café and two workshops, with over 80 health professionals, hosted at two hospice settings, and a range of social media activities including feedback from five young people with life-limiting and life-threatening conditions via a closed Facebook group and a twitter chat with 22 contributors. Data included field notes, workshop outputs (‘post-it ideas storm’ and ‘diamond ranking’) and social media postings that were analysed using thematic analysis. Across stakeholders, there was overwhelming support for explicit interventions that support the psychosocial and spiritual needs of children and young people with life-limiting conditions. Dignity Therapy could be a supportive intervention to promote open conversations about life, meaning and purpose and dying, between the child or young person, family members and health professionals, but needs adapting to meet their needs. A tool kit of interventions, appropriate to a range of ages and cognitive abilities was suggested, for example a storybook for young children and a digitalised version of dignity therapy for older children/young people. Collaboration with stakeholders is the first stage in developing, or adapting an existing intervention in a different context, a complex intervention. Our stakeholder consultation highlighted that Dignity Therapy has potential to improve the psychosocial and spiritual wellbeing of children and young people with life-limiting and lifethreatening conditions, through recalling memories and thinking about the things that are important to them and what they want to be remember for. Research is now needed to develop and test a Dignity Therapy intervention for children and young people with lifeliming and life-threatening conditions in the UK.


Background
Dignity Therapy (DT) is an evidence-based psycho-therapeutic intervention that has been found to improve the psycho-social and spiritual wellbeing of adults with life-limiting and life-threatening conditions (LLTC), through a process of life-review and meaning-making [1,2]. DT involves supporting patients to discuss their valued memories, accomplishments, life lessons and hopes for significant others. The intervention aims to bring about a sense of meaning and purpose for patients at the end of life, and their families [3].
DT emerged from the Dignity Conserving Model of Care (DCMC); this model was derived from qualitative studies undertaken with adult cancer patients receiving palliative care [4]. During DT, trained professionals undertake one-to-one semi structured interviews with individual patients, usually lasting for about an hour. The interview is structured by drawing on nine questions that relate to facets of the DCMC. This set of questions is referred to as the Dignity Therapy Questionnaire Protocol (DTQP) [1,4].
The DT interview is digitally recorded, transcribed, edited and returned to the patient, who can then make any changes before the document, called a generativity document or written legacy, is finalised. Many patients share these documents with significant others prior to their death, providing opportunity for meaningful conversations that can assist bereavement processes [5]. This person-centred therapeutic approach can also help health professionals understand individuals in the wider context of their lives and tailor support to their needs [6].
The growing evidence-base of DT in adult palliative care settings suggests that DT is more likely to improve spiritual wellbeing, overall quality of life, hope and sadness/depression scores that standard care alone [7][8][9][10][11], and has also been positively received by family members [12]. Health professionals report DT is a brief and flexible intervention that can be integrated into established supportive palliative care practices [12].

Introduction
There are over 50,000 children and young people (CYP) living with a life-limiting condition or life-threatening (LLTC) in the UK, which is predicted to increase with technological and medical advancements [13]. While these advancements have resulted in better management of these conditions, there remains uncertainty around prognosis, with many CYP living shortened lives [13]. The impact of LLTC on CYP, parents, siblings and the wider family is significant in terms of their emotional wellbeing [14,15]. Improving wellbeing throughout their shortened life journey, especially towards the end of life, requires care that addresses their psycho-social and spiritual needs, but this can be challenging for health professionals [16].
Palliative care for CYP supports the social, psychological and spiritual needs of CYP with expected shortened lives and their families [8,15]. However, there remains a significant unmet need in relation to meeting the psycho-social and spiritual wellbeing of CYP with LLTC, acknowledging their meaning-making and generativity needs and heightened risk of mental health problems [17,18]. Supporting CYP with LLTC to cope with their condition and integrating tailored psycho-social and spiritual support into care pathways could improve mental health and wellbeing outcomes [13,17].
Our evidence synthesis of published research focusing on CYP (aged up to 24 years) with LLTC found only four published studies about DT and related meaning making interventions for this age group [17]; one focused on young people (7-17 years) [19], the other three included young people but mean ages were 50-70 years [2,3,20]. In summary, and while recognising the limited evidence-base, we identified: 1. DT has potential to improve psycho-social wellbeing, emotional functioning, dignity and hope for CYP with LLTC; 2. DT is unique, welcomed and perceived as helpful for CYP and the family; 3. DT has not been widely used or evaluated specifically for CYP with LLTC. The studies that have included CYP have largely focused on older children / young people with cancer; 4. No UK or European studies were identified that had evaluated the acceptability, feasibility and effectiveness of DT or related meaning making interventions for CYP with LLTC.
Our review also highlighted missed opportunities to support CYP with LLTC and their families to engage in meaningful discussion about dignity, meaning making and end of life care in a structured way.
Since the publication of our review, Juliao et al [21] have published their work on adapting the DTQP for adolescent's aged 10 to 18 years in Portugal. They have developed an amended DT set of questions which could be used in practice for sick Portuguese adolescents. Although a five-stage process of validation is detailed, the amended DTQP is not provided and so it is unclear as to whether the new question format is aimed at those with typical/atypical cognitive development and for what conditions. At this stage we therefore cannot be sure if the proposed DT is appropriate for CYP in the UK with LLTC.
We have brought together expertise in advocacy for CYP with LLTC, palliative care, psychotherapy, children's nursing and spiritual care, to consider how to adapt and develop DT to produce an intervention/s that would be appropriate for CYP with LLTC in the UK.
We have also determined that stake holder consultation needs to be central to the proposal of amending/developing DT for CYP with LLTC for a number of reasons; 1) It is a moral obligation that healthcare delivery and research is responsive and addresses patient or in this case CYP / family priorities; 2) The sensitivity required when delivering DT type interventions must consider the views of CYP, their families and health professionals delivering care; 3) Involvement must not be tokenistic to meet funding body requirement; 4) The design and methods of undertaking DT research are appropriate for CYP with LLTC; 5) Stakeholder involvement is vital to shape and ensure the patient voice remains the focus of the research, even in the early stages of developing a programme of research.
The lead author (LW) is a core member of the MAGICYL team, and a young adult with a lifelimiting condition who has been involved in this study from its inception, co-designing our activities including preparing participant information and collecting information from young people.
The broad aim of our stakeholder activities was to explore whether DT would be acceptable for CYP with LLTC and health professionals.
The specific objectives were to: 1. Explore CYP with LLTC views of DT; 2. Explore health professionals, who support CYP with LLTC, perceptions of DT for this patient group; 3. Identify what, if any, meaning making activities are used in clinical practice for CYP with LLTC; 4. Review the DTQP and consider its application for CYP with LLTC.

Materials and Methods
We followed the National Research Ethics Service (NRES) and INVOLVE guidance [22], which states that ethical approval is not needed where people are acting as specialist advisers, providing valuable knowledge and expertise based on their experiences. However, in keeping with good practice, we obtained written consent from participants of the death café and closed Facebook group and verbal consent from workshop participants and have only included data if permission was given. For the twitter chat, participation was assumed if people contributed. All participants were above 16 years of age. AR, PI of the research team has completed the Dignity Therapy training course, undertaken in Canada. In addition, permissions were sought and received from the founder of DT, Prof Harvey Max Chochinov to explore amending and developing DT for CYP with LLTC in the UK.
Our health professional consultation experts were drawn from two children's hospices in the North of England, that provide family led care for CYP with LLTC. Our social media activities were not geographically restricted and included anyone with an interest in improving the psycho-social and spiritual wellbeing for people with LLTC and included young people. We undertook a range of consultation activities, in summary: 1. A Death Café was hosted by one of the hospices, facilitated by AR and JS, with a total 9 participants; 1 art therapist, 1 psychologist, 5 children's nurses, 1 psychologist, 1 adult nurse.
A Death Café is a directed discussion about death rather than a support group, and while public facing cafés have no agenda or objectives, Death Café type groups are increasingly being offered as a support mechanism for health professionals (Nelson, 2018). The first hour of the Death Café was unstructured giving participants opportunity to talk if they wished, or just listen to the conversations about death and dying. In the second hour DT was introduced, and participants were guided to discuss their views of the concept of DT and its possible application to CYP with LLTC.
2. Our social media activities involved: 1) Asking young people via a closed Facebook account, that LW established, whether DT could be relevant to them, they shared their views in a short-written narrative; 5 individuals with LLTC participated, aged 16-24 years.
2) We hosted a Twitter chat, for one hour, that involved tweeting a series of questions and responding to tweets to generate an online debate and capture broader views about DT and its relevance to CYP with LLTC. We summarised the chat in an online story that enabled us to include postings in subsequent data analysis [23]. Twenty-two individuals who engaged in the chat identified themselves; 8 children's nurses, 2 adult nurses, 6 Psychologists, 3 young people with LLTC and 2 parents of CYP with LLTC.
Two tasks were undertaken: 1) The first included 'post-it ideas storm', 'dot voting' and 'diamond ranking' from 'Participation -spice it up: a practical tool' [24] to identify what activities, whether labelled as DT or not, health professionals who support CYP with LLTC undertake that focus on end of life issues, and to maximise everyone's contributions. The process involved everyone writing down the activities they used, or thought would be useful on a separate post-it and placed them on a wall ('post-it ideas storm'). 'Dot voting' was used to gain a consensus about which activities would be most useful or could be delivered in practice; participants were given 3 sticker dots each and asked to place their dots next to those activities they considered most important or relevant to DT. 'Diamond Ranking' involved placing the post-its on a large diamond, grouping similar activities topics together and placing those identified as most relevant (through dot voting) at the top of the diamond and those least relevant at the bottom. Through discussion and negotiation, a consensus of possible activities that might support DT type interventions were identified. We photographed the 'diamond ranking' collage from both groups so that the data could be incorporated into our analysis.
2) Each participant was provided with the DTQP and either individually or if they preferred working in pairs were asked to consider each question and document their thoughts in relation to whether the question was suitable for CYP with LLTC, and if not how could be adapted.
The principles of thematic analysis were used to bring the data sets together, describe and summarise our activities [25]. First, AR typed the field notes and activities into a usable format; a word document that include both narratives and tables. AR and JS read the data sets and began the process of grouping data together, initially using the consultation objectives as a guide. Gradually as the different data were analysed categories emerged. Categories were modified as new insights became apparent until a coherent account developed that represented all data sets. Categories were grouped into two overarching themes. Bias was reduced by ongoing refinement of categories and discussions between all authors at three team meetings.
Participants in the Death Café and closed Facebook group were given a shopping voucher as a thank you for their time, with participants at the workshop given a thank you gift. This did not influence participants' decisions to contribute as they were unaware of the gift in advance, but we wanted to recognise their willingness to share their insights, experiences, and being involved in open discussions about a sensitive topic and area of care.

Results
One hundred and twenty-one people participated in our consultation events. Analysis of the data highlighted that both CYP and health professionals' perceived DT was highly relevant for CYP with LLTC but that discussing death and dying often lacked focus and could be particularly challenging when working with young children.
Our stakeholder participants overwhelmingly supported the relevance of a DT type intervention/s for CYP with LLTC: 'it is asking the questions that need to be asked' (Workshop participant). 'I love the idea that it's something to remember me by and something that would hold everything I think that's happened to make my life amazing' (Facebook participant). However, a DT for CYP with LLTC needs to flexible to meet a range of ages, cognitive and physical disabilities to be embedded into practice. The two themes and associated categories that emerged from the analysis are presented in Table 1.  (Figure 1).

Figure 1
Diamond rank of meaning making activities.
Stakeholder participants perceived that for some CYP with LLTC there may have been limited opportunities to fulfil their wishes and hopes and suggested that through DT a range of memory making activities could be identified, planned and captured, for example days out / experiences. Participant across data sets highlighted that the illness trajectory for CYP with LLTC is more uncertain than in adults populations, and therefore DT type interventions must be adaptable across the LLTC trajectories of individuals, be flexible and revisited at different transition stages, rather than an intervention that is delivered as a time bound task. Examples of participants accounts of DT for CYP with LLTC included shared sentiments: ' Workshop stakeholder participants highlighted that DTQP could be more meaningful for CYP, and there needed to be greater flexibility for health professionals when working with the younger age group and for CYP with communication difficulties. A shared concern was the use of the 'past tense' in the original DTQP which was perceived as not useful / lacked sensitivity (  Table 2 summarises the second task undertaken by workshop participants which was to consider each question of DT. There was a consensus that some of the questions could be difficult to understand for younger children and for young people with communication difficulties and complex needs. For example, CYP are likely to struggle to appreciate the difference between questions three and four. However, questions five to nine were highlighted as highly relevant and could be discussed through creative methods/activities. There was also further consideration that the intervention could be digitised for CYP: '  Language is very adult orientated, but it was agreed that this is an important question. Change items in brackets to just friends and family, and the question could be clearer. Question 7 and 8 could be combined, as they seem to be asked similar things.

Suggestions included:
Is there anything you would like to pass on to your loved ones before you die? (Be honest, you can be direct without being insensitive). What advice or words of guidance would you wish to pass along to your friends and family? 6. What advice or words of guidance would you wish to pass to your friends and family? Prompts: Is there anything you would like to pass on to family and / or friends before you die? 8. Are there words or perhaps even instructions you would like to offer your family to help prepare them for the future?
There was great support for this question, but it was perceived that it could be a burden for some CYP. More suitable for older children. Question 7 and 8 could be combined, seem to be asked similar things, make clear purpose of question Participants perceived this question, with adaptations, was highly relevant Remove 'permanent' -it is highly likely that CYP would want to change and edit their document over time.
Art ideas and memory boxes could fulfill this question. A good opportunity to make a digital record, which could be multimodal using arts and media such as music!

Suggestions included:
Is there anything that needs to be included that we have not talked about? Can we create a special time for you? A special day with your family?

Is there anything that needs to be included that we have not talked about?
There could be scope with CYP with LLTC to build on responses to question 9 and engage them in focused DT influenced creative arts, activities, related discussions, to contribute to what could be a DT project for the individual.

Can we create a special time/activity for you?
This question is an addition to the original DT protocol. For example, and within local limits -A special day with your family. Creating music work, with you and your family. Creating artwork, with you and your family. Followed by memories captured of the day

Embedding DT into Practice
Some of the DT type interventions already described are delivered in practice but are not aligned to a DT based or meaning making model/intervention. For example, there may be reflection or memory making activities undertaken in hospice settings, but these are usually undertaken just as creative and enjoyable events. Often activities are undertaken without consideration of broader meaning making issues or discussion over how the activity may contribute to legacy, directly with the CYP. In consideration of how DT could become a tool for CYP palliative and psychotherapeutic care, our stakeholder participants offered comments that can allow us to consider how we could integrate DT into current practice and what support health professionals would need themselves to engage with DT. . DT is an evidenced based psychotherapeutic intervention that requires training to appreciate its underpinnings and to consider ways to best support individuals in life review, meaning making and legacy leaving. The generativity documents/legacies created need to support the needs of the CYP with LLTC but also be supportive to recipients.

Discussion
Our findings suggest DT has the potential, with adaptations, to support CYP with LLTC and improve their overall wellbeing and quality of life. DT type interventions need to be developed and evaluated specifically for CYP with LLTC in the UK. The DTQP and related activities need to be responsive to the uncertainty of conditions and be appropriate to a range of ages and cognitive abilities.
Meaningful discussion about dignity, legacy and end of life care are currently being missed in CYP palliative care because activities are not identified as interventions to support the psychosocial and spiritual needs of CYP with LLTC facing shortened lives [26]. The DCMC hypothesises that there is a reciprocal interaction between aspects of human experience in DT which alleviates psycho-social and spiritual burdens [4,6] which is highly appropriate for CYP with LLTC. Meeting the spiritual and existential needs of CYP with LLTC is likely to help them cope with their condition and develop resilience to meet life challenges [27,28].
Our stakeholder consultation work suggests that there is scope to create a toolbox of DT activities for CYP with LLTC. DT generativity documents are being discussed in the literature and are evolving; for example, DT with older adults has resulted in producing picture books, including cherished photographs alongside key commentaries [29] and digital storytelling has shown to be beneficial with CYP with cancer conditions [19]. The stakeholder consultation findings highlight that for many CYP with LLTC that the generativity output would need to be updated: a digitalized format would provide CYP with LLTC the scope to edit and change and to save content and rethink and add at different junctures in their illness trajectories.
Many CYP with LLTC reach a crisis in terms of physical deterioration in adolescence or early adulthood, yet their psycho-social and spiritual needs are not currently addressed adequately [13]. CYP with LLTC facing impending death also need opportunities to explore their feelings without fear of upsetting family members [15,30,31].
We recognise that CYP with LLTC have unique physical and psycho-social needs, many rely on parents as their main source of emotional support and to undertake tasks of daily living [13]. DT would support CYP with LLTC if they wished, to share experiences and wishes without reliance on their parents or significant others. DT would allow CYP with LLTC to bequeath material they feel is important to share but may be too emotionally difficult to discuss with loved ones directly.

Conclusions
We have explored the views of young people with LLTC, parents and health professionals about DT and its relevance as a supported intervention for CYP with LLTC. Our stakeholder participants support the amendment and development of DT for CYP with LLTC. The key conclusions achieved following these 18 months of preparatory work include: 1) DT could be a supportive and supported intervention for CYP with LLTC; 2) There is potential psycho-social and spiritual benefit to CYP with LLTC recalling memories, imparting advice and considering meaningfulness through DT; 3) DT could promote open conversations about dying between the CYP with LLTC, family members and HCP; 4) For CYP with LLTC whose prognosis may be more favourable or unknown, little can be offered by hospice care. Being able to offer DT could therefore be invaluable for this group; 5) CYP with LLTC have lived most, if not all their lives in the technological age, therefore they may be more willing or motivated to engage in a digitalised DT; 6) There is consensus that DCMC is suitable for CYP with LLTC, but the phrasing of some questions of the DTQP could be more appropriate for CYP with LLTC; 7) An amended DTQP for CYP with LLTC is suggested; the DTQP must be rigorously refined so that it retains the original purpose and is meaningful and relevant to CYP with LLTC.
8) A toolbox of DT based activities would be supportive for both HCP and CYP with LLTC in practice.
Our next step is to take forward the findings of these stakeholder consultation activities and begin to plan studies that will focus on the amendment and development of DT for CYP with LLTC and to test their feasibility.