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Symptoms and Concerns Among Children and Young People with Life-Limiting and Life-Threatening Conditions: A Systematic Review Highlighting Meaningful Health Outcomes

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Abstract

Background

The design and provision of quality pediatric palliative care should prioritize issues that matter to children and their families for optimal outcomes.

Objective

This review aims to identify symptoms, concerns and outcomes that matter to children and young people (“young people”) with terminal illnesses and their families. Findings from the systematic review will inform the development of a relevant framework of health outcomes.

Method

This is a systematic literature review across multiple databases for identification of eligible primary evidence. Data sources included PsycINFO, MEDLINE, Embase, CINAHL, OpenGrey, and Science Direct Journals, searched from 1 August 2016 to 30 July 2017. The study also incorporates consultations with experts in the field, citation searches via Scopus, and a hand search of reference lists of included studies.

Results

Of the 13,567 articles that were evaluated, 81 studies were included. Most of these studies (n = 68) were from high-income countries and foused on young people with cancer (n = 58). A total of 3236 young people, 2103 family carers, 108 families, and 901 healthcare providers were included in the studies. Young people did not contribute to data in 30% of studies. Themes on priority concerns are presented by the following domains and health outcomes: (1) physical (n = 62 studies), e.g., physical symptoms; (2) psychological (n = 65), e.g., worry; (3) psychosocial (n = 31), e.g., relationships; (4) existential (n = 37), e.g., existential loss; and (5) “other” (n = 39), e.g., information access.

Conclusion

Burdensome symptoms and concerns affect young people with malignant and nonmalignant conditions and occur across the disease trajectory; pediatric palliative care should not be limited to the end-of-life phase. A child–family-centered framework of health outcomes, spanning the patient, family, and quality of service levels is proposed to inform service development. Future research should address gaps identified across the literature (i.e., the involvement of young people in research, evidence for developing countries, and a focus on nonmalignant conditions.

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Acknowledgements

The authors extend sincere thanks to Denise Bradley, St Christopher’s Hospice, UK, for her support towards developing the search strategy. A note of thanks is extended to Simon Etkind, Cicely Saunders Institute King’s College London, UK, for the useful comments on the health outcomes framework and the manuscript. Thanks also to Melanie Merriman, Moses Bateganya, and Chang Victor for the useful comments on the manuscript.

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Authors and Affiliations

  1. Cicely Saunders Institute, King’s College London, London, UK

    Eve Namisango, Katherine Bristowe, Irene J. Higginson & Richard Harding

  2. Academic Unit of Palliative Care, Leeds Institute of Health Sciences, University of Leeds, Leeds, UK

    Matthew J. Allsop

  3. Cicely Saunders Institute of Palliative Care, Policy & Rehabilitation, King’s College London, London, UK

    Fliss E. M. Murtagh

  4. Wolfson Palliative Care Research Center, Hull York Medical School, Hull, UK

    Fliss E. M. Murtagh

  5. Health Services and Population Research Department, Institute of Psychiatry, Psychology and Neuroscience, King’s College London, London, UK

    Melanie Abas

  6. International Children’s Palliative Care Network, Bloemfontein, South Africa

    Julia Downing

  7. Palliative Care Unit, Makerere University, Kampala, Uganda

    Julia Downing

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  1. Eve Namisango
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  6. Irene J. Higginson
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  7. Julia Downing
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Contributions

EN conceptualized and designed the study, reviewed the literature, abstracted and analyzed the data, and drafted the initial manuscript. Professor RH, Professor FMM, and Dr. KB reviewed the protocol, data abstraction tools, data analysis framework, and the results. Professor IJH and Dr. MA reviewed the research questions, search strategy, and review findings. Dr. MA reviewed the protocol, abstracted data, and carried out data analysis in association with the lead reviewer. Professor JD reviewed the analysis framework and appraised the interpretation of the review findings. All authors contributed to the final manuscript.

Corresponding author

Correspondence to Eve Namisango.

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Conflicts of interest

Eve Namisango, Katherine Bristowe, Matthew J Allsop, Fliss EM Murtagh, Melanie Abas, Irene J Higginson, Julia Downing, Richard Harding have no conflicts of interest.

Funding

The authors thank the Open Society Foundation, which funded this review through an unrestricted grant.

Data availability statement

The data extraction table used to compile all details from included studies accompanies this article as a supplementary file. EndNote files used for compiling articles during searching and reviewing can be requested from the corresponding author with a reasonable request outlining the intention for their use.

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Namisango, E., Bristowe, K., Allsop, M.J. et al. Symptoms and Concerns Among Children and Young People with Life-Limiting and Life-Threatening Conditions: A Systematic Review Highlighting Meaningful Health Outcomes. Patient 12, 15–55 (2019). https://doi.org/10.1007/s40271-018-0333-5

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