Research prioritisation exercises related to the care of children and young people with life-limiting conditions, their parents and all those who care for them: A systematic scoping review

Abstract

Background:In planning high-quality research in any aspect of care for children and young people with life-limiting conditions, it is important to prioritise resources in the most appropriate areas.Aim:To map research priorities identified from existing research prioritisation exercises relevant to infants, children and young people with life-limiting conditions, in order to inform future research.Design:We undertook a systematic scoping review to identify existing research prioritisation exercises; the protocol is publicly available on the project website.Data sources:The bibliographic databases ASSIA, CINAHL, MEDLINE/MEDLINE In Process and Embase were searched from 2000. Relevant reference lists and websites were hand searched. Included were any consultations aimed at identifying research for the benefit of neonates, infants, children and/or young people (birth to age 25 years) with life-limiting, life-threatening or life-shortening conditions; their family, parents, carers; and/or the professional staff caring for them.Results:A total of 24 research prioritisation exercises met the inclusion criteria, from which 279 research questions or priority areas for health research were identified. The priorities were iteratively mapped onto an evolving framework, informed by World Health Organization classifications. This resulted in identification of 16 topic areas, 55 sub-topics and 12 sub-sub-topics.Conclusion:There are numerous similar and overlapping research prioritisation exercises related to children and young people with life-limiting conditions. By mapping existing research priorities in the context in which they were set, we highlight areas to focus research efforts on. Further priority setting is not required at this time unless devoted to ascertaining families’ perspectives.

What do we know about evidence-informed priority setting processes to set population-level health-research agendas: an overview of reviews

“…Of the 31 systematic reviews included in this overview, sixteen reviews (Alqahtani et al 2021 ; Bragge et al 2015 ; El-Harakeh et al 2019 ; El-Harakeh et al 2020 ; Fadallah et al 2020 ; Garcia et al 2017 ; Graham et al 2020 ; Roche et al 2021 ; Rudan et al 2017 ; Rylance et al 2010 ; Stewart et al 2011 ; Tong et al 2015 ; Tong et al 2017 ; Viergever et al 2010 ; Wade et al 2021 ; Yoshida 2016 ) had a global scope. Additionally, five reviews (Cadorin et al 2020 ; Hasson et al 2020 ; Hawwash et al 2018 ; Manafo et al 2018 ; Mörelius et al, 2020 ) specifically focused on HICs, two reviews (McGregor et al 2014 ; Tomlinson et al 2011 ) focused on LMICs, five reviews (Badakhshan et al 2018 ; Booth et al 2018 ; Bryant et al 2014 ; Garcia et al 2015 ; Reveiz et al 2013 ) focused on specific countries or regions of countries and three (Bourne et al 2018 ; Odgers et al 2018 ; Terry et al 2018 ) did not specify geographic settings.…”

“…Additionally, five reviews (Cadorin et al 2020 ; Hasson et al 2020 ; Hawwash et al 2018 ; Manafo et al 2018 ; Mörelius et al, 2020 ) specifically focused on HICs, two reviews (McGregor et al 2014 ; Tomlinson et al 2011 ) focused on LMICs, five reviews (Badakhshan et al 2018 ; Booth et al 2018 ; Bryant et al 2014 ; Garcia et al 2015 ; Reveiz et al 2013 ) focused on specific countries or regions of countries and three (Bourne et al 2018 ; Odgers et al 2018 ; Terry et al 2018 ) did not specify geographic settings.…”

“…Three reviews (Booth et al 2018 ; Cadorin et al 2020 ; Mörelius et al, 2020 ) found that consensus building methods, such as Delphi processes, multi-staged surveys and focus groups, were commonly used to develop priorities. However, a key limitation of many studies was the lack of engagement with patients and carers or professional groups outside of healthcare sectors, such as social workers and teachers, in the research prioritization exercise.…”

Therapeutic letters: A qualitative study exploring their influence on the hope of parents of children receiving pediatric palliative care in Portugal

“…It is estimated that there are about 21.6 million children worldwide with life‐limiting or life‐threatening diseases (Downing et al, 2018), and specifically in Portugal, at least 6000 children are diagnosed with complex chronic disease annually (Minister of Health, 2014). The health needs of this population associated with the complex chronic conditions (CCCs) represent an emerging public health phenomenon globally (Booth et al, 2019; Shapiro et al, 2017). A CCC implies disability, functional limitation, and reserved prognosis, in addition to dependence on hospital and/or community health services (Lindley & Fortney, 2019).…”

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

“…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].…”