The World Health Organization estimates that 21 million children worldwide live with life-limiting or life-threatening conditions (LLTCs).1 Published studies2,3 show that every year more than 170 000 children with terminal illness in Europe need palliative care, while the number in Spain is estimated to be of 25 000 children. The LLTCs that affect children include solid tumour and blood cancers, neurological, neuromuscular, respiratory, metabolic, and chromosomal disorders and syndromes, malformations, infections and post-anoxic conditions, among others.4

According to international standards for paediatric palliative care (PPC),2 all palliative care programmes should have an established and systematic approach to the psychological and psychiatric aspects of care.5 The relationship between psychiatric disorders and other medical conditions is bidirectional, and the presence of psychiatric comorbidities is associated with greater functional impairment than would be expected otherwise.6 Although there are limited data on the paediatric population with palliative care needs, it is known that children and adolescents who have serious illness experience psychological distress.7,8 Some studies9,10 have found fear of being alone, loss of perspective and loss of independence in children with LLTCs or complex chronic diseases, and a high frequency of severe emotional distress and mood disorders. Anxiety, disruptive behaviours and emotion dysregulation are also common, with prevalence rates ranging from 7% to 50%.

The main objective of this study was to describe the psychological characteristics of young people with LLTCs in our setting and improve our understanding of the relationship between these characteristics and perceived wellbeing. Our main hypothesis was that emotional changes and maladaptive cognitive strategies have an impact on the child’s perceived wellbeing.

Of the 94 young people who met the inclusion criteria, 60 (63.83%) participated in the study. Fig. 1 presents a flow chart of the study participants.

Figure 1.

Flow chart of the sample (N = 60).

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Table 1 presents the characteristics of the sample. The mean age of the paediatric patients was 16.0 years (SD, 4.2; range, 9–24), 20 were female (33.3%), 41 were Spanish nationals (68.3%) and 34 were older than 14 years (56.7%).

Table 2 presents the data on the pWB of the patients and the different psychological variables assessed. On average, young people rated their pWB as 7 out of 10 (SD, 1.8; range, 3–10; median, 7.0) and 18.3% had difficulties in emotion regulation. When it came to psychosocial functioning, we found the highest risk of problems in the dimensions of problems with peers (46.7%) and emotional symptoms (45.0%). In addition, 30.0% of the young patients were at risk of anxiety and 22.0% at risk of depression. The coping strategy with the highest score was positive reappraisal (mean, 7.5; SD, 2.3; median, 8.0). The correlation coefficient for the strategies of catastrophizing and rumination was 0.72, which suggested that they could be measuring similar aspects. To avoid the inclusion of highly correlated variables and multicollinearity in the models, they were combined and included in the model as a one-dimensional factor.

Table 3 presents the results on pWB in relation to the other variables (demographic, disease-related and psychological). It shows a difference in the group aged more than 14 years (mean, 6.4; SD, 1.4) compared to the group aged less than 14 years (mean, 7.8; SD, 19) that was statistically (P = .003).

Among the disease-related variables, we found significant differences in the presence of exacerbated symptoms (P = .001).

When we reviewed the mean pWB values in relation to the assessed psychological characteristics, we found statistically significant differences. Lower mean pWB scores were associated with higher scores in emotional symptoms (mean difference [MD], 1.4; 95% CI, 0.6–2.2), and lower scores in emotion regulation (MD, 1.6; 95% CI, 0.5–2.8). Young people at risk of hyperactivity had a higher mean pWB compared to those not at risk (MD, 1.1; 95% CI, 0.0–2.3; P = .047). This result is not representative because there were few young patients with hyperactivity (n = 14) and most of them (n = 12) rated their pWB at 8 out of 10. Higher scores of depression were associated with lower mean pWB scores (MD, 2.2; 95% CI, 1.3–3.2; P < .001). We did not find significant differences in the mean pWB in relation to the risk of having anxiety (MD, 0.5; 95% CI, −0.5 to 1.6; P = .296).

Fig. 2 presents the results on the correlation between pWB and psychological variables. The variables that correlated most strongly with the self-reported pWB of young patients were the cognitive strategy of rumination (r = −0.57) and the risk of depression (r = −0.55). The pWB increased with increasing emotion regulation (r = 0.35) and increasing use of the cognitive strategy of positive refocusing (r = 0.25).

Figure 2.

Correlation of pWB with psychological variables (N = 60).

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Finally, to assess the predictive value of the variables that best explained the self-reported pWB of young patients, we fitted 3 regression models (Table 4). Model I considered demographic and disease-related variables. It showed that the combination of age greater than 14 years (coef = −0.98; P = .03) with the presence of symptom exacerbation (coef = −1.46; P = .01) explained 29.2% of the pWB variance. Model II considered the psychological variables, combining the rumination and catastrophizing cognitive strategies into a single variable due to the strong correlation discussed above. The variables risk of depression (coef = −1.49; P < .01) and the combined variable of rumination/catastrophizing (coef = −0.18; P < .01) were statistically significant. This model explained the variation in pWB with an R2 of 38.1%. To obtain a broader perspective, we decided to incorporate all the analysed variables in Model III. This last model showed that age greater than 14 years (coef = −0.78; P = .03), having exacerbated symptoms (coef = −1.04; p = 0.01), risk of depression (coef = −1.14; P = .01) and the cognitive strategies rumination and catastrophizing (coef = −0.17; P < .01) explained the variance in pWB with an R2 of 54.7%.

The primary objective of our study was to describe the main psychological features exhibited by young people with LLTCs and to improve the understanding of the relationship between these features and perceived wellbeing. The results show that the most prevalent psychological features were problems with peers, emotional symptoms, and anxious-depressive symptoms. The most frequently used “adaptive” cognitive strategies were positive reappraisal and refocus on planning, and the most frequently used “maladaptive” strategies were rumination and catastrophizing. We also found a lower pWB in patients aged 14 years and older, with exacerbated symptoms, with risk of depression and who used the rumination and catastrophizing cognitive strategies.

Our results indicate that patients with LLTCs aged 14 or over reported a poorer pWB compared to younger patients. Other studies show similar results in adolescents and young adults with palliative care needs, whose wellbeing is poorer compared to younger children, with higher rates of depression and anxiety, loss of independence, fear and guilt and a higher prevalence of complex pain.18 This could be a reflection of adolescents’ superior cognitive capacity for logical and abstract thinking, which leads to increased questioning of one’s own values and beliefs, and a greater understanding of the disease that may give rise to more illness-related stressors compared to younger children.19 Children aged 7 to 11 are better at applying their reasoning skills to concrete information compared to abstract concepts20 like pWB. Although more studies are necessary, it is nevertheless widely accepted that sociocultural factors can have an influence on young people’s pWB.21

The presence of exacerbated symptoms was found to be a significant and influential variable in the perception of decreased wellbeing. Other studies have found similar results, with an association between disease progression and higher physical symptom scores.9 Young people with LLTCs express a desire for physical comfort, including pain relief and being able to eat, walk and sleep.19

In our study, the risk of depression and the use of the rumination or catastrophizing cognitive strategies were associated with a poorer pWB and found to be significant in the regression model. Other studies show that palliative care needs substantially increase the risk of experiencing symptoms of anxiety and depression in young people by an estimated 20% to 50%.21 The cognitive strategies used most commonly by young people in our study were adaptive strategies, specifically positive reappraisal and refocus on planning. Even so, we did not find a direct relationship between their use and the pWB. However, the use of maladaptive strategies, such as rumination and catastrophizing, was associated with a decrease in pWB. Other studies show that adaptive coping strategies can be key to improving wellbeing,22 and have found a stronger relationship between these cognitive emotion regulation strategies and the report of depressive symptoms.13 In our study, a high percentage of young people (46.7%) reported problems with their peers. Social interaction with friends is an important coping tool for adolescents and young adults.21 Forty-five percent of the young patients in our study exhibited emotional symptoms. It is known that emotional distress is another psychological challenge faced by young people21 and that higher levels of emotion regulation are associated with a better psychological adjustment to their situation and a better mental health.23 This increases their risk of developing emotional and behavioural problems compared to their healthy peers.24 Young people with LLTCs describe many psychological and emotional repercussions such as anger, worry, sadness and an overwhelming desire to be happy.25 Various studies describe that children may express optimism to maintain a positive mindset,26 that they find it difficult to rate their wellbeing negatively27 or that it may be a way of preserving an appearance of normality. Some children, however, pretend to be brave, possibly to motivate themselves or to protect their parents from feeling guilty or sad.28

Professionals can optimize the wellbeing of young people and their families by identifying the issues that matter to them and working together to set goals and the steps to achieve them. Simple tools and training to support professionals may help them implement this approach.29

Despite the importance of its findings, there are limitations to our study. First of all, the sample size, use of a cross-sectional design in a non-random sample and the fact that the study was conducted in a single centre hinders the generalizability of the results. Reproducing the study in larger samples and analysing similar populations would be important. Nevertheless, a prospective study in a sample of 60 patients of these characteristics is a particularly strong representation of the population of interest. Secondly, the use of sociodemographic variables could be problematic. The wide age range of the participants may be a confounding variable in the interpretation of the results, as well as in the application of the different self-report scales. Thirdly, the wellbeing assessment was simplified into a single question, but future studies should encompass evaluation of physical, social, psychological and spiritual aspects. Furthermore, most children with LLTCs are aged less than 8 years and/or have cognitive disorders, so a potential future line of research would be to confirm these results in children with these characteristics.

Young people with LLTCs experience a substantial psychological impact, including problems with peers, emotional symptoms, and anxious and depressive symptoms. The self-perceived wellbeing was poorest in patients aged 14 years of older, with exacerbated symptoms, at risk of depression and who used the rumination and catastrophizing cognitive strategies. These results illustrate the need to work directly with these young people and to guide the psychological evaluation and management with instruments and therapeutic approaches that respond to these characteristics. After obtaining these results, future research could pursue the generalization of these findings in the Spanish population.

DTP conceived and designed the study, designed the data collection instruments, collected data, and drafted and revised the manuscript.

JL and ECR conceived and designed the study, coordinated and supervised data collection, and participated in the critical review of the intellectual content of the manuscript and its editing.

SNV participated in the critical review of the intellectual content of the manuscript and contributed to its drafting and editing.

CB and MG performed the analysis of the results and contributed to the drafting and editing of the manuscript.

The first draft of the manuscript was written by Daniel Toro-Pérez and all authors commented on previous versions of the manuscript. All authors read and approved the final version of the manuscript.

We obtained the informed consent of all the patients who participated in the study and their parents.

All the procedures that contributed to this study adhered to the ethical standards of the competent national and institutional human research committees and with the Declaration of Helsinki of 1975 as revised in 2008. The study was approved by the Medical Research Ethics Committee of the Sant Joan de Déu Hospital (file PIC-158-20; 02/06/2020).

No funds, grants, or other support were received by the authors during the preparation of this manuscript.