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

Eve Namisango,Richard Harding,Fliss E M Murtagh,Melanie Abas,Katherine Bristowe,Julia Downing,Matthew J Allsop,Irene J Higginson

doi:10.1007/s40271-018-0333-5

Abstract

The design and provision of quality pediatric palliative care should prioritize issues that matter to children and their families for optimal outcomes. 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 informthe development of a relevant framework of health outcomes. This is a systematic literature review across multiple databases for identification of eligible primary evidence. Data sources includedPsycINFO, 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. 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 peoplewith 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 followingdomains 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. 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 identifiedacross the literature (i.e.,the involvement of young people in research, evidence for developing countries, and a focus onnonmalignant conditions.

Highlights

An overarching theme identified in young people with malignant and non-malignant illnesses is the issue of multi-dimensional, complex symptoms and concerns which interact and occur across the disease trajectory
This review aims to appraise the global evidence on symptoms and concerns that matter to young people and their families to identify meaningful core person-centred health outcomes in young people with LLC and their families
The objectives of this review are to: i) appraise the methodological quality and extent of research literature, detailing patient, caregiver, family, and health provider reports of symptoms and concerns across disease trajectories for young people living with LLC and their families; ii) identify the gaps that exist in the research literature, and; iii) synthesise reports of symptoms and concerns, using a conceptual framework to identify the domains of importance in the development of outcome assessment measures for young people with LLC

Summary

Objectives

This review aims to identify symptoms concerns and outcomes, that matter to children and young development of a relevant framework of health outcomes. This review aims to appraise the global evidence on symptoms and concerns that matter to young people and their families to identify meaningful core person-centred health outcomes in young people with LLC and their families. The objectives of this review are to: i) appraise the methodological quality and extent of research literature, detailing patient, caregiver, family, and health provider reports of symptoms and concerns across disease trajectories for young people living with LLC and their families; ii) identify the gaps that exist in the research literature, and; iii) synthesise reports of symptoms and concerns, using a conceptual framework to identify the domains of importance in the development of outcome assessment measures for young people with LLC

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