Each year, around 170,000 children in Europe die who require palliative care due to various diseases, according to the World Health Organization. In Spain, it is estimated that 25-30 out of every 10,000 children have serious illnesses that limit or threaten their lives, resulting in 930 – 1500 annual deaths. Pediatric Palliative Care (PPC) begins from the diagnosis of a potentially life-threatening illness and can be combined with other treatments. Psychological care in this field is key to improving quality of life, well-being, and reducing physical and emotional symptoms in children and their families. Although all autonomous communities in Spain have regional plans that include psychological care, its implementation remains limited due to a shortage of specialized health professionals and a lack of definition regarding what such care will consist of.
Objective
This report aims to evaluate the effectiveness, safety, and cost-effectiveness of psychological interventions in the pediatric population eligible to receive PPC and their families, as well as the ethical, legal, organizational, and social aspects, and the research needs and standard outcome measures associated with these interventions.
Methodology
Effectiveness and Safety
A systematic review (SR) of the literature was conducted by independent peer reviewers consulting the following databases: MEDLINE, Embase, CINAHL, APA PsycINFO, CENTRAL, and CDSR up to July 2024. Systematic reviews with or without meta-analyses (MA) (from 2010 to 2024) and randomized controlled trials (RCTs) (up to 2024), published in Spanish or English, that evaluated the effectiveness and safety of psychological interventions in children eligible for PPC and/or their families were selected. The initially considered outcome measures were: quality of life, mental health, emotional and psychological well-being (e.g., anxiety, depression, stress, coping, feelings of burden, etc.), and adverse events associated with the psychological intervention in the child and/or family/main caregivers; as well as reduction of physical symptoms related to the illness (e.g., fatigue, pain, nausea, vomiting, sleep quality, etc.) in the child. Risk of bias of the studies was assessed with the RoB-2 tool. When possible, a quantitative synthesis of the results was performed by meta-analysis using Review Manager version 5.4.
The quality of evidence assessment and strength of recommendation grading were carried out following the methodology of the international working group Grading of Recommendations Assessment, Development and Evaluation (GRADE).
Cost-effectiveness and Economic Analysis
A systematic review of economic evaluations of psychological interventions in the context of PPC was performed. Economic evaluations (parallel to primary studies or model-based) reporting any of the following outcomes were sought: incremental cost-effectiveness ratio, costs expressed in monetary units, and benefits expressed as quality-adjusted life years, life years gained, monetary units, or any of the outcome measures included in the effectiveness section. Methodological quality was to be assessed using the checklist by Drummond et al. (2005), as well as data extraction and a narrative synthesis with tabulation of results. An economic evaluation was planned, with its design determined by the application of the algorithm for conducting economic evaluations in health technology assessment (HTA), which is based on the availability of scientific evidence, minimum required information, and available resources.
Aspectos éticos, legales, organizativos y sociales
Se adaptó el marco evaluativo del Core Model 3.0. de EUnetHTA, así como los criterios establecidos por la Red Española de Agencias de Evaluación de Tecnologías Sanitarias (RedETS) del SNS. Se planteó una RS cuyo alcance partió de la misma RS de efectividad y seguridad descrita anteriormente. Se incluyeron RS si aportaban elementos de análisis relevantes para este apartado, RS de estudios cualitativos y meta-etnografías. Para la evaluación de la calidad se utilizó la escala AMSTAR-2. Se realizó una síntesis narrativa teniendo en cuenta criterios de relevancia y coherencia de los resultados. De manera adicional se llevó a cabo una consulta a expertos clínicos con diferentes perfiles profesionales y representantes de pacientes sobre diferentes cuestiones relacionadas con los aspectos éticos, legales, organizativos y sociales de las intervenciones psicológicas en el contexto del CPP.
Results
Effectiveness and safety
The evaluation of effectiveness and safety is based on the results of 68 studies (4646 participants: 1747 minors and 2859 parents and/or caregivers). Most were conducted with minors eligible for PPC and/or their families, and only two studies were explicitly conducted in the PPC context. The conditions evaluated were cancer (57 studies), cystic fibrosis (6 studies), and sickle cell disease (5 studies). Regarding the population intervened, 34 studies were conducted with families (primary caregivers, i.e., parents), 31 with minors, and 3 with both minors and their families. Regarding the psychological interventions evaluated, most cases involved a combination of psychological approaches, predominantly cognitive-behavioral, although a wide variety of interventions (cognitive therapy, acceptance and commitment therapy, mindfulness, dignity therapy, play therapy, filial therapy, etc.) and techniques (progressive muscle relaxation, deep breathing, guided imagery, etc.) were used. The most frequent comparator was usual care, followed by no intervention or delayed intervention (“waitlist”). The quality of evidence for almost all outcome measures evaluated and classified as critical and important for decision-making was rated very low (⨁◯◯◯), mainly due to risk of bias in individual studies, small sample sizes, wide confidence intervals, heterogeneity of populations and interventions evaluated, and the low number of studies identified per outcome measure. Overall, meta-analyses conducted in minors eligible for PPC showed a significantly favorable effect of adding psychological intervention to usual care in reducing symptoms of depression, anxiety, distress or discomfort during invasive medical procedures (e.g., lumbar punctures, bone marrow biopsy, etc.), general pain intensity and during invasive medical procedures, as well as in increasing caloric intake in minors with cystic fibrosis (p < 0.05) post-intervention. Results for anxiety, pain, and distress or discomfort during medical procedures appear to be maintained in some short-term follow-up periods. However, for other pain-related measures (e.g., feelings of hope, sleep problems, emotional, social, and behavioral adjustment, behavioral problems, self-esteem), results were inconsistent across studies. On the other hand, meta-analyses conducted with families showed a significantly favorable effect of adding psychological intervention to usual care in improving symptoms of depression, anxiety, distress or discomfort, post-traumatic stress symptoms, coping, and feelings of hope (p < 0.05) post-intervention. Results for depression, anxiety, and post-traumatic stress symptoms appear to be maintained in some short-term follow-up periods. However, for other outcome measures (e.g., quality of life, stress, resilience, emotional impact of the minor’s illness experience, psychological well-being, psychiatric symptoms), results were inconsistent across studies. Regarding safety, no study reported adverse effects derived from psychological intervention. In summary, the quality of the available evidence for psychological interventions for pediatric populations eligible for palliative care and their families is very low, given the heterogeneity and quality of identified studies. Nevertheless, despite these limitations, the available results and their clinically relevant effects suggest a positive balance for adding different psychological interventions, predominantly those with a cognitive-behavioral approach, to usual care for minors eligible for PPC and their families.
The systematic review of economic evaluations did not identify any study that met the established selection criteria. Following the algorithm for conducting economic evaluations in health technology assessment (HTA) and considering the resources available at the time this report was prepared, a full economic evaluation and budget impact analysis were not conducted, mainly due to the absence of quality data that would allow estimates with a sufficient level of certainty.
Ethical, legal, organizational, and social aspects
Ten systematic reviews were included, with an overall low quality. The main findings indicate that psychological interventions within the PPC framework are essential to address the emotional and psychosocial impact faced by children and their families. These interventions should focus on providing continuous emotional support, resilience strategies, effective communication, and tools to manage complex emotions such as anticipatory grief. Despite their importance, access to these interventions is limited by organizational, economic, and geographic barriers, creating significant inequalities in access. Consultation with professionals and patient representatives confirms that the most acceptable and feasible interventions are those that: 1) Are personalized, adapting to the needs of minors and families at each stage of disease care; 2) Incorporate multidisciplinary teams trained to work in a coordinated manner respecting the specificity of each professional role.
Conclusions
- The available evidence on the effectiveness and safety of psychological interventions in pediatric populations susceptible to receiving palliative care and their families is of very low quality for virtually all outcome measures evaluated due to the risk of bias in individual studies, small sample sizes, high imprecision of confidence intervals, heterogeneity of populations and interventions evaluated, and the limited number of studies identified per outcome measure. Nevertheless, despite these limitations, the available results and their clinically relevant effects suggest a positive balance in adding different psychological interventions to the usual care of minors susceptible to receiving PPC and their families.
- In minors susceptible to receiving PPC, the results show that adding psychological interventions to usual care appears to have a favorable effect in reducing symptoms of depression, anxiety, distress or discomfort during invasive medical procedures, overall pain intensity and during invasive medical procedures, as well as increasing caloric intake, specifically in minors with cystic fibrosis. Some of these results are maintained in short-term follow-ups (anxiety, pain, and distress or discomfort during medical procedures).
- Regarding the families of minors susceptible to receiving PPC, the results show that adding psychological interventions to usual care appears to have a favorable effect in reducing symptoms of depression, anxiety, distress or discomfort, post-traumatic stress symptoms, coping, and hope. Some of these results are maintained in short-term follow-ups (depression, anxiety, and post-traumatic stress symptoms).
- Regarding safety, no adverse effects derived from psychological interventions were found.
- No published economic evaluations assessing the cost-effectiveness of psychological interventions in the context of PPC were identified.
- Psychological interventions in PPC should comprehensively address the emotional needs of minors and their families, offering flexible and continuous psychological support adapted to family dynamics and stages of the disease.
- Specialized training and effective integration of multidisciplinary teams are essential to guarantee interventions that generate trust and well-being in families.
- It is crucial to reduce geographical, economic, and organizational inequalities through public policies that promote equity in access and availability of psychological services within the PPC framework.
- In conclusion, more studies with robust methodology are required to evaluate the effectiveness and safety of the interventions assessed, as well as to collect data on resource use and costs to inform their cost-effectiveness. Additionally, more qualitative studies are needed to explore the gender perspective of these interventions and the perceptions, experiences, expectations, values, and preferences of pediatric patients and their families, as well as the healthcare professionals who assist them.
Recommendations
Based on the results obtained in this report, a weak recommendation is issued in favor of including psychological interventions in the usual care of the pediatric population susceptible to receiving PPC and their families within the healthcare system. This recommendation is based on the available evidence regarding effectiveness, safety, and the consideration of ethical, legal, organizational, and social aspects at the time of preparing this report. However, the quality of the current evidence is low or very low, which limits the certainty of these findings. Additionally, the feasibility of implementing these interventions widely in routine clinical practice presents certain challenges, and the costs derived from their implementation have not been analyzed. Therefore, this recommendation is subject to the generation of new scientific evidence that strengthens the quality and consistency of data on effectiveness and safety; evaluates the economic impact and sustainability of these interventions in real clinical settings; and explores strategies to overcome organizational barriers and improve their implementation in routine clinical practice. For this purpose, more high-quality research with a low risk of bias is required, including both economic analyses and qualitative studies focused on the experiences and needs of pediatric patients, their families, and the healthcare professionals who assist them.
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- Full report:
