Original Title: Intervenciones psicológicas en población pediátrica susceptible de recibir cuidados paliativos y su familia

Authors' objectives: The purpose of this report is to evaluate the effectiveness, safety, and cost-effectiveness of psychological interventions for paediatric populations eligible for PPC and their families. It also aims to address ethical, legal, organizational, and social aspects, as well as the research needs and standard outcome measures associated with these interventions.

Authors' results and conclusions: RESULTS: EFFECTIVENESS/SAFETY The evaluation of effectiveness and safety was based on the results of 68 studies (4646 participants: 1747 children; 2859 parents). Most focused-on children eligible for PPC and/or their families (parents), with only two studies explicitly conducted in the PPC context. The diseases studied included cancer (57 studies), cystic fibrosis (6 studies), and sickle cell disease (5 studies). Regarding the populations targeted, 34 studies focused on parents, 31 on children, and 3 on both children and their parents. Most psychological interventions combined approaches, predominantly cognitive-behavioural therapy, though a wide variety of interventions were included (e.g., cognitive therapy, acceptance and commitment therapy, mindfulness, dignity therapy, play therapy, filial therapy, etc.) and techniques (e.g., progressive muscle relaxation, deep breathing, guided imagery, etc.). The most common comparator was usual care, followed by no intervention or delayed intervention (e.g., waitlist). The quality of evidence for virtually all the outcomes assessed and rated as critical and important for decision-making has been classified as very low (⨁◯◯◯). This is primarily due to the risk of bias in individual studies, the small sample sizes used, the high imprecision of confidence intervals, the heterogeneity of populations and interventions evaluated, and the limited number of studies identified for each outcome assessed. Overall, MAs conducted on children eligible for PPC show a significantly favourable effect of adding psychological interventions to usual care. This includes reductions in symptoms of depression, anxiety, distress during invasive medical procedures (e.g., lumbar punctures, bone marrow biopsies), general pain intensity and pain during invasive medical procedures, and increased caloric intake in children with cystic fibrosis (p < 0.05) following the intervention. Effects on anxiety, pain, and distress during medical procedures appear to persist in some short-term follow-up periods. However, for other outcomes such as hope, sleep problems, emotional, social, and behavioural adjustment, conduct problems, and self-esteem, the results are inconsistent across studies. In other hand, MAs conducted on parents show a significantly favourable effect of adding psychological interventions to usual care, improving symptoms of depression, anxiety, distress, post-traumatic stress symptoms, coping, and hope (p < 0.05) following the intervention. Effects on depression, anxiety, and post-traumatic stress symptoms also appear to persist in some short-term follow-up periods. However, for other outcomes such as quality of life, stress, resilience, the emotional impact of the child’s illness experience, psychological well-being, and psychiatric symptoms, the results are inconsistent across studies. Regarding safety, no studies reported adverse effects related to psychological interventions. In summary, based on the available evidence, it is currently not possible to draw solid and generalizable conclusions about the effectiveness of psychological interventions for paediatric populations eligible for palliative care and their parents due to the heterogeneity of the identified studies. Nevertheless, despite these limitations, the available results suggest a positive balance in favour of adding various psychological interventions—primarily cognitive-behavioural approaches—to the usual care of children eligible for PPC and their parents. COST-EFFECTIVENESS AND ECONOMIC ANALYSIS The SR of economic evaluations did not identify any studies meeting the established selection criteria. Using the algorithm for conducting HTA economic evaluations and considering the effectiveness and safety results, as well as available resources, a full economic evaluation or budget impact analysis was not conducted due to the lack of high-quality data needed to make reliable estimates. ETHICAL, LEGAL, ORGANIZATIONAL AND SOCIAL ASPECTS Ten SRs with an overall low quality were included. The main findings indicate that psychological interventions within the framework of PPC 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 geographical barriers, leading to significant inequalities in access. Consultations with professionals and patient representatives confirm that the most acceptable and feasible interventions are those that: 1) Are personalized, adapting to the needs of children and their families at each stage of disease management; 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 for paediatric populations eligible for PPC and their families is of very low quality for practically all the outcomes evaluated. This is due to the risk of bias in individual studies, small sample sizes, high imprecision in confidence intervals, heterogeneity in populations and interventions assessed, and the limited number of studies identified for each outcome. Nevertheless, despite these limitations, the available results and their clinically relevant effects suggest a positive balance in adding various psychological interventions to the standard care for children eligible for PPC and their families. • For children eligible for PPC, the results indicate that adding psychological interventions to standard care appears to have a favourable effect on reducing symptoms of depression, anxiety, and distress during invasive medical procedures, as well as general pain intensity and pain during invasive medical procedures. It also seems to increase caloric intake, particularly in children with cystic fibrosis. Some of these effects appear to persist in the short-term follow-up (e.g., anxiety, pain, and distress during procedures). • For families of children eligible for PPC, the results suggest that adding psychological interventions to standard care appears to have a favourable effect on reducing symptoms of depression, anxiety, distress, post-traumatic stress symptoms, and on improving coping and hope. Some of these effects also seem to persist in the short-term follow-up (e.g., depression, anxiety, and post-traumatic stress symptoms). • Regarding safety, no adverse effects from the psychological interventions were identified. • No published economic evaluations were identified that assessed the cost-effectiveness of psychological interventions in the context of PPC. • Psychological interventions in PPC should comprehensively address the emotional needs of children and their families, offering flexible and continuous psychological support tailored to family dynamics and the stages of the disease. • Specialized training and the effective integration of multidisciplinary teams are essential to ensure interventions that foster trust and well-being in families. • It is crucial to reduce geographic, economic, and organizational inequalities through public policies that promote equity in access to and availability of psychological services within the PPC framework. • In conclusion, more robust methodological studies are needed to evaluate the effectiveness and safety of the assessed interventions and to gather resource usage and cost data to inform their cost-effectiveness. Additionally, more qualitative studies are needed to explore the perceptions, experiences, expectations, values, and preferences of paediatric patients and their families, as well as the healthcare professionals assisting them.

Authors' recommendations: Based on the findings of this report, a weak recommendation is made in favour of including psychological interventions in the routine care of paediatric populations eligible for PPC and their families within the healthcare system. This recommendation is grounded in the available evidence on effectiveness, safety, and consideration of ethical, legal, organizational, and social aspects at the time this report was prepared. However, the current evidence is of low or very low quality, limiting the certainty of these findings. Additionally, the feasibility of widespread implementation of these interventions in routine clinical practice presents certain challenges, and the costs associated with their implementation could not be analysed. As a result, this recommendation is contingent upon the generation of new scientific evidence to strengthen the quality and consistency of data on effectiveness and safety; evaluate the economic impact and sustainability of these interventions in real clinical settings; and explore strategies to overcome organizational barriers and improve their implementation in routine clinical practice. To achieve this, further high-quality research with low risk of bias is needed, including economic analyses and qualitative studies focused on the experiences and needs of paediatric patients, their families, and the healthcare professionals who care for them.

Authors' methods: EFFECTIVENESS AND SAFETY A systematic review (SR) of the literature was conducted by independent reviewers, consulting the following databases: MEDLINE, Embase, CINAHL, APA PsycINFO, CENTRAL, and CDSR up to July 2024. The selected studies included SRs with or without meta-analyses (MA) (from 2010 to 2024) and randomized controlled trials (RCT) (up to 2024) published in Spanish or English that assessed the effectiveness and safety of psychological interventions in children eligible for PPC and/or their parents. The initially considered outcome measures were: quality of life, mental health, emotional and psychological well-being (e.g., anxiety, depression, stress, coping, feeling of burden, etc.), and adverse events related to psychological interventions in the child and/or their family or primary caregivers. Additionally, outcomes included the reduction of disease-related physical symptoms (e.g., fatigue, pain, nausea, vomiting, sleep quality) in the child. The risk of bias in the studies was assessed using the RoB-2 tool. Where possible, a quantitative synthesis of the results was performed through meta-analysis using the Review Manager software, version 5.4. The evaluation of the quality of evidence and the grading of the strength of recommendations 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 SR of economic evaluations of psychological interventions in the context of PPC was conducted. Economic evaluations (either alongside primary studies or model-based) were sought if they reported any of the following outcomes: incremental cost-effectiveness ratio, costs expressed in monetary units, and benefits expressed in quality-adjusted life years, life years gained, monetary units, or any of the outcome measures included in the effectiveness section. The methodological quality was planned to be assessed using the checklist by Drummond et al. (2005). Data extraction and narrative synthesis with tabulated results were also foreseen. An economic evaluation was planned, with its design determined by the application of the algorithm for conducting economic evaluations in HTA. This algorithm is based on the availability of scientific evidence, the minimum necessary information, and the resources available. ETHICAL, LEGAL, ORGANIZATIONAL AND SOCIAL ASPECTS The evaluation framework of the EUnetHTA Core Model 3.0 was adapted, along with the criteria established by the Spanish Network of Health Technology Assessment Agencies (RedETS) of the National Health System (NHS). A systematic review (SR) was designed, building on the SR of effectiveness and safety described earlier. SRs were included if they provided relevant analytical elements for this section, as well as SRs of qualitative studies and meta-ethnographies. The AMSTAR-2 scale was used to assess the quality of the reviews. A narrative synthesis was performed, considering the relevance and consistency of the results. Additionally, a consultation was conducted with clinical experts from various professional backgrounds and patient representatives to address issues related to the ethical, legal, organizational, and social aspects of psychological interventions in the context of PPC.

Project Status: Ongoing

Anticipated Publish Date: 2024

URL for published report: https://sescs.es/intervenciones-psicologicas-poblacion-pediatrica-cuidados-paliativos/

English language abstract: An English language summary is available

Publication Type: Full HTA

Country: Spain

Organisation Name: Canary Health Service

Contact Address: Dirección del Servicio. Servicio Canario de la Salud, Camino Candelaria 44, 1ª planta, 38109 El Rosario, Santa Cruz de Tenerife

Contact Name: sescs@sescs.es

Contact Email: sescs@sescs.es