A Systematic Review Highlighting Meaningful Health Outcomes: The design and provision of quality pediatric palliative care should prioritize issues that matter to children and their families for optimal outcomes.
The number of children and young people (“young people”) (aged 0–23 years) living with life-limiting and life-threatening conditions (LLC) is increasing worldwide [1, 2, 3]. A crucial component of care for young people with LLC is palliative care, which seeks to improve the quality of life of patients and their families facing problems associated with life-threatening illness. Recent global estimates have set the annual number of young people who need palliative care at any point during their disease trajectory at 21 million, with 8 million requiring some form of specialist palliative care .
For high-income countries such as the UK and America, the prevalence of LLC in young people is increasing , with most deaths caused by trauma, congenital conditions, extreme prematurity, and other acquired illness . In resource-limited settings, the case mix of young people with LLC also includes conditions such as HIV, which continues to pose a public health concern and is typically accompanied by a high symptom burden (e.g., pain, weight loss, lack of appetite, feeling sad, and difficulty sleeping).
According to the Joint United Nations Programme, 2.9–3.5 million children are living with HIV infection, with sub-Saharan Africa shouldering 91% of the global burden . The situation is further exacerbated by the increasing incidence and prevalence of various types of cancer among young people, with over 80% of deaths occurring in resource-limited settings . Besides HIV and cancer, complex chronic, neonatal, and other noncommunicable diseases contribute to mortality and morbidity among the children in resource-limited settings .
The current delivery of palliative care for young people typically runs parallel to existing healthcare systems, without integration of existing and specialty services . The provision of quality care to young people with LLC requires the critical establishment of robust evidence on the symptoms and concerns that matter to the patients and their families . Despite the need for this evidence, information on meaningful outcomes for young people with LLC is currently limited [12, 13]. This need for patient-level data is more pressing than ever amidst the worldwide demand to address the absence of person-centered outcome measures in measuring quality of pediatric care [13, 14].
Research with young people with LLC is hampered by several methodological challenges. These include clinical considerations such as participants being very ill, limited access to potential participants , and limited capacity to generate self-reports because of the less developed (or impaired) verbal and cognitive skills of this population .
As symptoms and concerns affect children’s beliefs, expectations, and perceptions, it becomes important for self-reports from young people to be prioritized wherever possible . The inclusion of the perspectives of young people and their families is critical to ensure that outcome measures are meaningful to them and their families . However, the level of young people’s involvement in research remains limited, and self-reports of their outcomes and experiences are not commonly reported . Innovative and feasible approaches for engagement of young people in research that will shape their care should be prioritized.
This review aims to appraise the global evidence on symptoms and concerns that matter to young people and their families to identify meaningful person-centered health outcomes in young people with LLC and their families.
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.
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.
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.
An overarching theme identified in young people with malignant and nonmalignant illnesses is the issue of multidimensional, complex symptoms and concerns that interact and occur across the disease trajectory.
Our findings demonstrate considerable overlap in themes of illness experiences across diagnostic groups, settings of care and geographical location, alongside identifying common behaviors.
This review presents an evidence-based child/family framework of symptoms, concerns, and health outcomes spanning three levels: child, family, and quality of services. This framework can encourage the development of pediatric palliative care outcome measures to inform service audits, research, and evaluations.
There is evidence that young people aged ≥ 6 years can self-report on symptoms, concerns, and health outcomes, but reduced communication and cognitive abilities also remain a challenge. Developing person-centered child-appropriate information and communication tools that are more inclusive of patients with special needs should thus be prioritized.
We highlight the limited involvement of young people in research, a disparity in evidence coverage for developing countries, and a lack of evidence for nonmalignant conditions.
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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