Cari Malcolm

Prioritisation of future research topics for children’s hospice care by its key stakeholders: a Delphi study.

The Delphi process, widely used in health research to seek consensus on key issues amongst large stakeholder groups, was adopted to allow families, hospice staff/volunteers and linked professionals to identify and prioritise future research priorities for children’s hospice care. In the qualitative Round 1, interviews with families (n = 5), linked professionals (n = 18) and focus groups with hospice staff and volunteers (n = 44) led to the generation of 56 research topics categorised within 14 broad themes. To give a larger number of stakeholders (n = 621) (including families n = 293; hospice staff/volunteers n = 216 and professionals n = 112) the opportunity to rate the importance of each research topic and seek group consensus on the future research priorities for children’s hospice care, subsequent Rounds 2 and 3 involved the use of postal questionnaires. Response rates to questionnaires were 44% in Round 2 (274/621) and 83% in Round 3 (204/247). Participants prioritised research topics relating to 1) hospice and respite care needs of young people (aged 16 +), 2) pain and symptom management and 3) bereavement and end-of-life care. There was wide acknowledgement by those took part in the process of the difficulty in rating the topics, and emphasis on the fact that all of the topics raised during the project are of high importance and merit further research. The current salient issues perceived by key stakeholders as being the research priorities for children’s hospice care were identified. Addressing these priority topics for research would further contribute to the development of a much needed evidence base in children’s hospice and palliative care research and optimise the delivery of children’s hospice services that are underpinned by valid and robust research.

Challenging symptoms in children with rare life-limiting conditions: findings from a prospective diary and interview study with families

Aim:  The aim was to describe the nature, frequency, severity and management challenges of symptoms in children with two rare life‐limiting conditions [Mucopolysaccharide (MPS) and Batten disease].

Challenging symptom profiles of life-limiting conditions in children: a survey of care professionals and families:

This paper reports on data from the initial phase of a UK-wide study exploring life-limiting conditions (LLCs) in children where, because of their rarity, little is understood regarding the significant symptom challenges of families and care professionals who support them. In this initial phase, care professionals (n = 43) and families (n = 16) completed a survey to identify which rare LLCs present symptom challenges and which individual symptoms prove difficult to manage. Findings led to the prioritization of mucopolysaccharidoses, Batten Disease and leukodystrophy. Care professionals’ rationales for selecting these conditions included the presence of symptoms that are very difficult to manage, a requirement for additional clinical evidence to support symptom management, and the progressive nature of these diseases where symptoms frequently change and are difficult to predict. Families identified symptoms such as behavioural difficulties, visual impairments and communication impairments as those being most difficult to manage and having most impact on quality of life for children and families.

Exploring the experiences and perspectives of families using a children's hospice and professionals providing hospice care to identify future research priorities for children's hospice care

The main objective of this study is to generate a list of priority topics for children’s hospice care research in Scotland from the perspective of its key stakeholders. The method consists of qualitative semi-structured interviews with families using hospice services (n = 5), four focus groups with hospice staff and volunteers (n = 44) and telephone interviews with professionals associated with the hospice (n = 18). Fourteen broad themes emerged following thematic content and interpretive analysis of the interview data. Some of the research themes were specific to certain stakeholder groups, whereas other themes were identified unanimously across all the stakeholder groups as being priority areas for future research. Increasing awareness of and improving access to children’s hospice care, hospice and respite care needs of young people, community/home care and issues related to supporting the wider family arose, independently, in all three stakeholder groups as being priority topics for future research. In conclusion, a greater evidence base is required in the field of children’s palliative care and the topics researched should be identified and led by those most closely involved in the hospices. Engaging families and care providers in the process of identifying research priorities resulted in the development of an extensive research agenda, which will contribute to quality hospice care for children and families.

The Economic Costs of Health Service Treatments for Asbestos-Related Mesothelioma Deaths

Abstract:  This article explores the complex and neglected picture of occupational and environmental disease healthcare costs specifically relating to asbestos. Diagnosed mesothelioma cases in Scotland in one calendar year were used to investigate the subject in greater depth. Data from UK sources on asbestos disease types recorded in 2000 and their disease treatment costs were obtained. Acute care economic costs of these diseases are estimated. One hundred and twenty diagnosed, recorded, and treated cases of asbestos‐related diseases occurred in 2000 in Scotland. Mesothelioma accounted for 100 cases and directly cost Scottish National Health Service hospitals an estimated £942,038. The estimated UK figure in 2000 was at least £16,014,646 because official figures for diagnosed and recorded deaths from mesothelioma are running at over 1700 a year with rises predicted for 2010 of 2000 deaths. By 2003, 50,000 people in the UK had died from diagnosed and recorded mesothelioma since records began. Earlier disease treatment costs would have been significantly lower than those in 2000 but, at 2000 prices, cost to the UK was roughly £471,019,000 in acute hospital expenditure. Figures for primary care costs, including caregiver costs, are incomplete or unknown. These disease costs are substantial and have some international generalizability. Treatment patterns and costs vary greatly. Many lung cancer cases due to asbestos exposure occur globally for each mesothelioma case. Hence figures provided in this article are certain to be gross underestimates of the total health service and personal economic costs of asbestos illness and treatment in Scotland.

A relational understanding of sibling experiences of children with rare life-limiting conditions: findings from a qualitative study

Mucopolysaccharidoses (MPS) and Batten disease are rare life-limiting conditions (LLCs) characterised by progressive and permanent physical and cognitive decline. The impact of such conditions on families, and notably on siblings, has not yet been described or documented. This paper presents data from a UK-wide study that sought to understand the family experience of supporting a child with the rare degenerative LLCs of MPS and Batten disease. The aim of this paper is to report sibling experiences related to these rare degenerative and progressive conditions, in order to inform the future development of supportive interventions. Eight siblings of children with MPS (n = 7) and Batten Disease (n = 1) participated in semi-structured qualitative interviews. A card sort technique was utilised to support and engage the children. Siblings are clearly impacted emotionally, pragmatically and relationally by the ill health of another child in the family. The data indicate four key themes which demonstrate impacts on siblings: perceptions of the condition and its symptoms, impact on daily life, emotional consequences and ways of coping. Siblings often had considerable knowledge of the condition and took on important roles in symptom management. However, these experiences were in the context of managing relationships within the family (often protecting parents from an awareness of how much they knew) and relationships at school (including distraction from learning and being bullied by peers). The data highlight how sibling experiences are generated through a combination of negative disability discourses and support through peers and family members. The data indicate how these features shift as a consequence of witnessing the advancement of their brother’s or sister’s condition and the emotional sequelae of disease progression. Exploration of siblings’ experiences of living with such rare progressive and degenerative LLCs suggest the focus of interventions to support this group should address their emotional health and ways to overcome isolation and build connections with other siblings who share their unique experiences. Critically, the data suggest that sibling support should be cognisant of the trajectory of the illness as well as the family, school and peer relational contexts that siblings inhabit.