Introduction
The World Health Organization defines palliative care as “an approach to care which improves quality of life of patients and their families facing life-threatening illness through prevention, assessment, and treatment of pain and other physical, psychological, and spiritual problems” (World Health Organization, 2002). “Palliative care for children (PPC) represents a special, albeit closely related field to adult palliative care” (World Health Organization, 1998, p. 8). It incorporates “total care” that addresses the child's body, mind, and spirit (World Health Organization, 1998), and it serves as a means of providing support to the family (Morgan, Reference Morgan2009). PPC is “recognised to be a specialised type of care requiring specific skills and knowledge” (Steele et al., Reference Steele2008, p. 229); nonetheless, healthcare professionals can rely on a paucity of research on which to base care (Committee on Palliative and End-of-Life Care for Children and Their Families, 2003; Steele et al., Reference Steele2008). As noted by the Committee on Palliative and End-of-Life Care for Children and Their Families, healthcare professionals and researchers are conscious of “shortfalls in clinical research involving palliative and end-of-life care for children” (Committee on Palliative and End-of-Life Care for Children and Their Families, 2003, p. 351). Because it can access and include patients’ perspectives, qualitative research (QR) can contribute (Sackett & Wennberg, Reference Sackett and Wennberg1997) to PPC quality improvement. In the past few decades, the number of qualitative studies on PPC has increased slightly (Akard, Reference Akard2013), as has interest in understanding the endeavor of QR in this area. Accordingly, the present article aims to understand the contribution of QR that has included children as participants.
Methods
Search strategy
We performed a systematic review and a meta-summary. A meta-summary is a method for aggregating findings that appear in exclusively qualitative reports and is suitable for acknowledging the “preponderance of evidence” (Thorne et al., Reference Thorne2004) in QR and highlighting what the qualitative results reveal. We used both Preferred Reporting Items for Systematic Reviews and Meta-Analysis (for tutoring in retrieving papers (Moher et al., Reference Moher2009) and the Enhancing Transparency in Reporting the Synthesis of Qualitative Research guidelines (Tong et al., Reference Tong2012) to report the results.
We searched for QR articles on PPC involving children ranging in age from 3 to 18 years in the electronic databases MEDLINE, CINAHL, PsycINFO, PsycARTICLES, and ERIC, with no limitations on publication date or language. A generic search was also performed using Google Scholar, ResearchGate, and Academia.edu. We also performed a manual search using the references of systematic reviews we retrieved, and we included articles for both cross-checking and identifying other potential QR articles. We performed a systematic review from July 2016 to December 2016. We discussed the search strategy and agreed on using keywords from three primary domains: PPC-related terms, AND participant-related, AND QR keywords. We summarize the search terms in Table 1. We managed to work collaboratively using an online platform (i.e., Google Drive) to share progress regularly.
Search domains and keywords
PPC, palliative care for children; QR, qualitative research.
Search outcomes
We retrieved a total of 1,240 articles by searching the databases and checking the articles’ references. We used an Excel file and inserted all titles and authors’ names. We manually removed duplicates (n = 504) and then reviewed 736 articles by title. The articles had to meet the following inclusion criteria: report of a QR and study conducted with children ranging in age from 3 to 18 years and experiencing PPC. We eliminated 324 articles that were considered irrelevant according to their titles because they did not mention any methodology related to QR. There were 340 articles that, according to their abstracts, did not meet the inclusion criteria (i.e., QR articles involving children experiencing PPC and ranging in age from 3 to 18 years). LG and GA retrieved the full texts and shared the articles using the online platform. LG proposed an assessment framework to describe the aims, research questions, method, and ethical issues and to evaluate the inclusion/exclusion for each full text. We wrote a file for each article and uploaded the files using the online platform for sharing. We were able to check the full text of 72 articles, of which 56 were excluded because they did not meet the inclusion criteria. A total of 14 QR articles reporting on 12 unique studies and two mixed-method articles were selected. The Preferred Reporting Items for Systematic Reviews and Meta-Analysis flow chart of the search process is shown in Figure 1.
Qualitative Preferred Reporting Items for Systematic Reviews and Meta-Analysis.
Critical appraisal of the studies
Three authors (EB, MS, and LG) used Critical Appraisal Skills Programme (CASP, 2013) to highlight methodological strengths and weaknesses of the selected studies.
Meta-summary
LG, EB., MS, and VDM extracted the results/findings sections in each included study. All the text under the “results/findings” sections was extracted manually and entered in a table. LG performed the thematic analysis of findings to identify overarching themes (Sandelowski & Barroso, Reference Sandelowski and Barroso2003; Sandelowski et al., Reference Sandelowski, Barroso and Voils2007). The thematic techniques included (1) line-by-line coding, (2) the reduction of these codes into themes, and (3) calculation of the frequency (Sandelowski & Barroso, Reference Sandelowski and Barroso2003), as shown in Table 4. The main topics were discussed among all the authors. GA and VC detailed the final version of the themes.
Results
We found 16 qualitative studies addressing children's experience of PPC. We report the studies’ characteristics in Table 2.
Studies' characteristics
PC, palliative care.
Using the CASP, we found seven articles of high/moderate-to-high quality, in which authors openly discuss the research design and methods along with the methodological steps they have followed. There are three articles of moderate/moderate-to-low quality, as the description of the method lacks essential information (i.e., internal consistency, interview guide, data saturation). The main weaknesses of studies we assessed as low quality regard the ethical considerations and the data analysis process, which are not explicitly accounted. The appraisal is shown in Table 3.
Critical appraisal of the included articles
The entire sample of the retrieved studies involved 212 children. For 105 children, the studies lacked detailed age-related information because the authors had indicated large age ranges, whereas, for the remaining children, the developmental stage can be sketched as follows: 29 preschoolers, 48 primary schoolers, and 30 teenagers. The reported conditions of pediatric participants were oncologic or hematologic diseases (n = 60), nononcological illnesses (n = 63), and unspecific life-limiting or life-threatening situations (n = 89).
The participating children were recruited within the services they attended and were mainly involved as interviewees in 10 of 16 articles. Additional data were derived from ethnographic observations (two articles), a focus group, a questionnaire with several free-response questions, a play-based workshop in action-research, and a personal diary.
Meta-summary results
We concentrated the analysis for the meta-summary on 14 articles because three articles refer to the same study. The QR with PPC children focuses on eight major themes, which are listed in Table 4.
Meta-summary: themes and subthemes
Relationship with professional caregivers
The majority of QR with pediatric patients in palliative care (Amery et al., Reference Amery2009; Carter, Edwards & Hunt, Reference Carter, Edwards and Hun2015; Coad et al., Reference Coad2015; Flavelle, Reference Flavelle2011; Hsiao et al., Reference Hsiao, Evan and Zeltzer2007; Spalding & Yardley, Reference Spalding and Yardley2016; Swallow, Forrester & Macfadyen, Reference Swallow, Forrester and Macfadyen2012) describes the relationship with professional caregivers. This theme includes communication between children and doctors (Carter et al., Reference Carter, Edwards and Hun2015, Coad et al., Reference Coad2015; Hsiao et al., Reference Hsiao, Evan and Zeltzer2007); the bond with family support workers (Carter et al., Reference Carter, Edwards and Hun2015); a reflection of the relationship between the home nurse and Ed, who is the protagonist in the phenomenological case study (Flavelle, Reference Flavelle2011); and relations with professionals from a short-break service (Swallow et al., Reference Swallow, Forrester and Macfadyen2012) and a hospice (Amery et al., Reference Amery2009).
Pain and pain management
Pain and pain management (Borghi et al., Reference Borghi2014; Flavelle, Reference Flavelle2011; Kortesluoma et al., Reference Kortesluoma, Nikkonen and Serlo2008; Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004, 2006; Tamannai et al., Reference Tamannai2015) constitute another dominant theme. Living with pain has an impact on self-image (Borghi et al., Reference Borghi2014) and may produce fatigue (Flavelle, Reference Flavelle2011). Children can apply self-help strategies for pain (Borghi et al., Reference Borghi2014; Kortesluoma et al., Reference Kortesluoma, Nikkonen and Serlo2008) such as physical actions (i.e., massage, caressing, deep breathing, moving or changing the body posture, application of heat or cold, resting and relaxation, immobilization or exercise, eating and/or drinking, loosening tight clothes, and defecation) (Kortesluoma et al., Reference Kortesluoma, Nikkonen and Serlo2008). These actions offer them a sense of control over the pain. Besides, children follow what Kortesluoma et al. (Reference Kortesluoma, Nikkonen and Serlo2008) interpreted as cognitive and behavioral actions as they learn about what techniques may increase or reduce pain. Children can give meaning to their illness (Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2006) and can distinguish different pains and their origins (Tamannai et al., Reference Tamannai2015), such as pain caused by medical and diagnostic procedures and basic nursing (Borghi et al., Reference Borghi2014; Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004) and pain caused by accidents occurring in everyday activities. Children also experience pain they find inexplicable (Flavelle, Reference Flavelle2011; Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004), such as pain caused by aggressive actions of other people (Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004). Kortesluoma and Nikkonen (Reference Kortesluoma and Nikkonen2004) reported that one of the most intense types of pain children experienced was the physical pain and emotional suffering caused by being hit or verbally abused.
“Living beyond pain”
We titled this theme “living beyond pain” to indicate what children experiencing PPC consider important for them despite the pain and the disease (Borghi et al., Reference Borghi2014; Davies et al., Reference Davies2005). This theme emerges from six articles (Amery et al., Reference Amery2009; Borghi et al., Reference Borghi2014; Carter et al., Reference Carter, Edwards and Hun2015; Davies et al., Reference Davies2005; Flavelle, Reference Flavelle2011; Swallow et al., Reference Swallow, Forrester and Macfadyen2012). Children need to be engaged in activities such as sports and individual play (Borghi et al., Reference Borghi2014; Carter et al., Reference Carter, Edwards and Hun2015), video games, which authors consider an escape from illness (Flavelle, Reference Flavelle2011), or group play (Amery et al., Reference Amery2009; Carter et al., Reference Carter, Edwards and Hun2015; Davies et al., Reference Davies2005). Playing is seen as a way to forget the disease for a while (Amery et al., Reference Amery2009; Flavelle, Reference Flavelle2011). In an assessment of a short-break service for children with life-limiting conditions (Swallow et al., Reference Swallow, Forrester and Macfadyen2012), teenagers appreciated better access to leisure activities (i.e., hide and seek, treasure hunts, playing on the computer/the Internet, singing and listening to music) compared with being at home.
Relationship with the family
The relationship between pediatric patients and their families was mentioned in six qualitative studies (Borghi et al., Reference Borghi2014; Flavelle, Reference Flavelle2011; Gaab et al., Reference Gaab, Owens and MacLeod2013; Hinds et al., Reference Hinds2005; Kortesluoma et al., Reference Kortesluoma, Nikkonen and Serlo2008; Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2006). The relationship with the family is interlinked with the relationship with healthcare professionals and peers (Kortesluoma et al., Reference Kortesluoma, Nikkonen and Serlo2008). Children describe their family as the primary source of support (Borghi et al., Reference Borghi2014; Gaab et al., Reference Gaab, Owens and MacLeod2013; Hinds et al., Reference Hinds2005). Studies (Flavelle, Reference Flavelle2011; Gaab et al., Reference Gaab, Owens and MacLeod2013; Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2006) have found that the diagnosis affects the relationships among family members. Children often note that their feelings have changed from before the diagnosis (Gaab et al., Reference Gaab, Owens and MacLeod2013; Hinds et al., Reference Hinds2005). On the one hand, the clinical situation makes them feel closer to their parents, whereas, on the other hand, it augments children's dependency on family members (Flavelle, Reference Flavelle2011; Gaab et al., Reference Gaab, Owens and MacLeod2013).
Treatment
A fifth theme is the view children have regarding treatment (Flavelle, Reference Flavelle2011; Hinds et al., Reference Hinds2005; Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004; Tamannai et al., Reference Tamannai2015). Children feel and comprehend both the adverse effects of medications (Flavelle, Reference Flavelle2011) and the benefit of pain relief medications (Tamannai et al., Reference Tamannai2015). Children are aware of the effects treatment may induce (Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004). Moreover, children can recall all of their past treatment options (Hinds et al., Reference Hinds2005).
Evaluation of the service provision
Another theme emerging from the articles is the children's evaluation of service provision (Amery et al., Reference Amery2009; Davies et al., Reference Davies2005; Noyes et al., Reference Noyes2013; Swallow et al., Reference Swallow, Forrester and Macfadyen2012). A tool for future care planning has been assessed by children, parents, and healthcare professionals (Noyes et al., Reference Noyes2013). Similarly, children and parents had the opportunity to evaluate the equipment and, generally, the atmosphere of a short-break service (Swallow et al., Reference Swallow, Forrester and Macfadyen2012), the strengths and weaknesses of children's palliative care service in Africa, including staff attitudes (Amery et al., Reference Amery2009), and a Canadian free-standing hospice programme (Davies et al., Reference Davies2005).
Death and consequences of end-of-life decisions
The meanings children give to their end-of-life situation (Flavelle, Reference Flavelle2011; Gaab et al., Reference Gaab, Owens and MacLeod2013) and the perception of consequences that the decisions or treatments may bring (Hinds et al., Reference Hinds2005; Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004) emerged as a theme. Children mentioned death and indicated that they were aware of having limited time (Gaab et al., Reference Gaab, Owens and MacLeod2013). Ed wrote about the end in a humorous way (Flavelle, Reference Flavelle2011). A child affirmed that he knew he would die if he lost blood (Kortesluoma & Nikkonen, Reference Kortesluoma and Nikkonen2004); other patients identified their death as an outcome of their decision (i.e., getting sick from an experimental medication, delaying death, and dying) (Hinds et al., Reference Hinds2005).
Relationship with peers
The last theme addressed in QR with PPC patients involves peer relationships (Borghi et al., Reference Borghi2014; Gaab et al., Reference Gaab, Owens and MacLeod2013; Swallow et al., Reference Swallow, Forrester and Macfadyen2012). It includes critical communication with friends (Borghi et al., Reference Borghi2014), the desire and the difficulty of speaking and having a “normal” conversation with healthy friends (Gaab et al., Reference Gaab, Owens and MacLeod2013) and the ability and willingness to talk freely with peers who share the same condition (Swallow et al., Reference Swallow, Forrester and Macfadyen2012). If allowed to confront others, children are more likely to feel understood (Gaab et al., Reference Gaab, Owens and MacLeod2013). Nonetheless, relationships with others may cause feelings of discrimination (Gaab et al., Reference Gaab, Owens and MacLeod2013).
Discussion
A meta-summary of findings was conducted to understand the contribution of QR on PPC in which the voices of patients were solicited. We retrieved 16 qualitative studies concerning eight major themes (and 22 related subthemes). The relationship with professional caregivers, pain and its management, “living beyond pain,” and the relationship between pediatric patients and their families are dominant themes in the literature. These themes are consistent with research on children's perspectives regarding their treatment and service provision. Finally, four studies discussed the meanings children give to their end-of-life situation and the consequences of clinical decisions, whereas three articles mentioned the relationships children in PPC have with their peers.
According to this systematic review and the meta-summary results, the voice of children, when they are able to talk and express themselves, appear to be misrepresented within the scientific panorama (Akard, Reference Akard2013; Hinds, et al., Reference Hinds2007). Emerging PPC research priorities (Baker et al., Reference Baker2015; Steele et al., Reference Steele2008) would be adequately met by conducting more QR “with” (and not only “on”) pediatric patients (Committee on Palliative and End-of-Life Care for Children and Their Families, 2003; Stevenson et al., Reference Stevenson, Achille and Lugasi2013). Further QR is needed to investigate what matters most for children receiving PPC (Snaman et al., Reference Snaman2018; Steele et al., Reference Steele2008), assess evidence-based practice guidelines in PPC (Baker et al., Reference Baker2015), and comprehend the children's role in making treatment decisions about their palliative and end-of-life care (Baker et al., Reference Baker2015). A recent example of dealing qualitatively with those emerging priorities is a study protocol about specialized outpatient palliative care, which has been conceived to provide information through qualitative narrative interviews with pediatric patients on what matters most to them receiving specialized outpatient palliative care (Ulrich et al., Reference Ulrich2018).
Accessing the voices of children through QR may also contribute to helping parents communicate with their child about the child's life-threatening illness and the likelihood of death (Baker et al., Reference Baker2015). These research implications are consistent with recently published articles (Badarau et al., Reference Badarau2017; Snaman et al., Reference Snaman2018; Weaver et al., Reference Weaver2016), which list topics of urgent consideration. Among them, effective communication (Snaman et al., Reference Snaman2018), and shared decision-making (Badarau et al., Reference Badarau2017; Weaver et al., Reference Weaver2015) are pressing subjects.
We also recommend that researchers and clinicians conduct QR about how to take advantage of children's spontaneous pain management to define its effectiveness, how the relationship with professional caregivers can make important differences in the provision of PPC, the role of play and leisure for children experiencing PPC, the way intrafamiliar relationships change during the care process (Snaman et al., Reference Snaman2018) and how to address the interdependency among PPC patients and family members.
Strengths and limitations
Among the limitations of the systematic review, we note that, although we followed a rigorous process in retrieving the articles, articles may have been missed. To address this limitation, we searched five comprehensive databases and performed a generic search using academic social networks (i.e., Google Scholar, ResearchGate, and Academia.edu) in addition to checking references in articles. The systematic review was limited to articles from peer-reviewed journals and those that were published in English. Many studies appear in different languages and in non–peer-reviewed journals (i.e., dissertations, doctoral theses, and gray literature), which we have not included. We understandably did not include qualitative PPC articles investigating the experience of children who were not able to communicate for themselves; therefore, we attempted to avoid bias in reviewing and appraising the studies using more than two members in each of the review stages, and the online platform that we used helped the team in cross-validation and checking tutoring. Regarding the meta-summary, we decided not to differentiate the findings according to children's age because of the scarceness of the number of items and detailed age-related information in most of the retrieved articles. Nonetheless, we are aware that children have particular physical, cognitive, and social development trajectories that change over time; therefore, when the QR body is increased, there will be a need to distinguish children according to age and related needs. Thus, we performed a thematic analysis that led us to collect themes from the included qualitative studies. Although the themes were discussed and validated by all of the authors, they might not represent all possible interpretations. To the best of our knowledge, this study is the first meta-summary of the current contribution of QR on PPC in which the voices of children were solicited and considered.
Conclusion
Conducting this systematic review and meta-summary revealed that QR with children about their experience in PPC is an emerging and promising field. We support the need for more QR, especially regarding the research relaunches we highlighted. It is our understanding that conducting QR with children in PPC will be pivotal for comprehending patients’ psychological, social, and spiritual needs (World Health Organization, 2002) and for improving PPC interventions and, consequently, children's quality of life (Huang et al., Reference Huang2010; O'Quinn & Giambra, Reference O'Quinn and Giambra2014). In fact, QR findings may make the first-person perspective on PPC emerge and concur with respect to incorporating “total care” that addresses the child's body, mind, and spirit (World Health Organization, 2002).
Acknowledgments
The authors received no financial support for the research, authorship, and publication of this article.
LG and GA conceived and designed the systematic review and the meta-summary. All authors discussed the search strategy and agreed on final terms list for the search. All authors revised the article critically and approved the release sent to the journal.
Conflicts of interest
None.
