https://orcid.org/0000-0003-2464-6845
Figures
Abstract
Well-being in children with a complex medical condition (CMC) impacts the way they view and communicate with their immediate environment as well as their development, and it is thus necessary to inquire about the contextual issues and different needs that a CMC carries. This pilot study aimed to identify factors of pediatric well-being from the experience of hospitalization and convalescence of youth with CMC and their caregivers, in a cross-sectional analysis using a selective methodology complemented by an indirect observational methodology. We analyzed the quality of life and well-being of youth with CMC using a validated KINDLR questionnaire. We collected 35 surveys: 11 from youth with CMC and 24 from caregivers from Spain. We focused the analysis on sociodemographics, well-being perceptions, and coping strategies variables. The results show that children aged between 3 and 6 years and their caregivers scored physical well-being the lowest out of all dimensions of well-being, and they scored family well-being the highest. Moreover, youth between the ages of 7 and 17 years and their caregivers scored school-related well-being the lowest. Coping strategies to deal with stressful situations differ between children and caregivers. While children mainly engage in social withdrawal, caregivers engage in cognitive restructuring and expressing emotions. However, we did not find a relationship between coping strategies and well-being perceptions. These results highlight the need to facilitate communication spaces with both families and health professionals where the voice of children is considered.
Citation: Muñoz-Violant S, Violant-Holz V, Rodríguez MJ (2023) Factors of well-being of youth with complex medical conditions from the experience of hospitalization and convalescence: A pilot study. PLoS ONE 18(5): e0285213. https://doi.org/10.1371/journal.pone.0285213
Editor: Omar M. Khraisat, Al-Ahliyya Amman University, JORDAN
Received: December 23, 2022; Accepted: April 18, 2023; Published: May 4, 2023
Copyright: © 2023 Muñoz-Violant et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Data Availability: The datasets generated and analyzed during the current study are included in the paper as Supporting information.
Funding: This pilot study has received funding from Fundació Privada Fútbol Club Barcelona (Spain) (Project number FBG-310741). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.
Competing interests: The authors declare that the study was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. The sponsor was not involved in the study design; the collection, analysis, and interpretation of the data; the writing of the report; or the decision to submit the article for publication.
Introduction
The number of children with complex medical conditions (CMC) continues to increase due to the reduction in infant mortality, access to new technologies, and the improvement in social and health markers [1, 2]. Following Cohen et al.’s [3] definition of medical complexity, CMC are here understood as the medical fragility and intensive care needs of children presenting either a multisystemic disease (congenital or acquired) or a severe neurological condition, both leading to a functional impairment of daily living. This group of patients also includes the pediatric population with special health care needs due to increased risk of chronic physical, developmental, behavioral, or emotional conditions [4]. Chronic conditions are characterized by four domains [3], namely needs, chronic conditions, functional limitations, and healthcare use, all of which contribute to health-related quality of life (HRQoL). Thirty years ago, children with these conditions could not survive; nevertheless, they can now be treated with fragile drug profiles and special care needs [3]. A 2020 study found that in Spain, around 15% of the sampled children accessing primary care had a chronic illness [5], and another previous study found that the most frequent pediatric chronic conditions were neurological (76.95%), gastrointestinal (63.78%), and respiratory diseases (61.72%), respectively [6]. In the specific case of cancer, 80% of affected European boys and girls survive until adulthood, but many of them require permanent medical follow-ups and substantial care needs [7–9], including hospitalization in intensive care units and greater technological assistance and highly specialized services [10–12].
HRQoL is a variable used to measure the impact of illness on quality of life (QoL). Previous research has shown that HRQoL is significantly low in children with CMC and their caregivers [13] due to various factors such as caregiver stress and burnout [14, 15], the hospitalization process [16, 17], and poor physical and mental health [18] caused by a condition of illness.
The hospitalization process often involves a traumatic loss of children’s daily life and significant physical discomfort and pain [19]. Consequently, these children regularly show poor well-being, psychological distress, and psychiatric symptomatology such as anxiety and depression [20–23]. Generally, the presence of secondary mental health problems leads to a higher number of hospital visits, worse treatment outcomes, and poor pain management [24]. Additionally, studies have suggested that both psychological distress and psychiatric conditions are associated with the dysregulation of the immune function in youth [25, 26].
Poor physical and mental health well-being negatively impacts the family environment, which can slow the recovery of children and adolescents with CMC [27–29]. Simultaneously, research has shown that the caregivers and family members of youth with CMC regularly experience psychological distress, post-traumatic stress, and symptoms of anxiety and depression following their children’s hospitalization [30–33]. These psychological effects among caregivers seem to be present across all diagnoses—cancer [34–36], heart diseases [37–40], and other illnesses—that require hospitalization at either intensive neonatal care units or intensive pediatric care units [41–43].
It is necessary to establish a supportive environment that considers the psychosocial needs of patients and their caregivers during the hospitalization process holistically [44]. In Spain, this environment must target improved communication with health professionals and the family members’ feelings of isolation and maladaptive emotions, such as fear and pain. Additionally, it is necessary to expand opportunities for patients for relationships with their peer group [45–49].
Another environmental factor that determines the QoL of youth with CMC is the school context, which is an opportunity for children to develop socially, emotionally, physically, and educationally [50]. Nevertheless, the school experience of children and adolescents with CMC is significantly impacted by the primary and secondary aspects of their illness, including hospitalization stays, physical and psychological discomfort, and feeling different from their peers [51, 52]. Furthermore, parental perceptions of QoL related to their children’s health can also influence school attendance [53]. An American National Survey with more than 400 children with congenital heart disease reported that they are three times more likely to miss more than 10 days of daycare/school a year [54]; consequently, school reintegration during and after treatment is paramount [55] for improving the QoL—and probably the self-esteem—of these patients. Self-esteem levels in youth with CMC are actually significantly lower than their healthy peers, particularly once hospitalization and treatment have ended [56, 57]. As low self-esteem can develop into poor satisfaction and anxiety and depressive symptomatology, it is essential to target it [58–60]. Finally, one study found self-esteem to predict the willingness to receive treatment [61].
For all these reasons, appropriate coping strategies for children with CMC and their caregivers that can ease their physical and mental health burden and improve their QoL must be investigated further. Consequently, this pilot study aimed to identify the factors of pediatric well-being from the experience of hospitalization and convalescence of youth with CMC and their caregivers. For the purpose of this study, “youth” indicates children and adolescents.
Materials and methods
Study design
This was a cross‐sectional pilot study with a selective methodology. It used the validated KINDLR questionnaire as elicitation of the responses, complemented along with an indirect observational methodology [62], and it focused on responses to open-ended questions. These responses were coded by consensus agreement [63] by the team’s three members following a well-established validated model [64]. Within the mixed-methods framework, qualitative elements (indirect observational data) were integrated with quantitative elements—the quantitative analysis techniques used, which are highly robust and suitable for qualitative data [65–67]. Fig 1 summarizes the methodological approach of the study.
Participants
We recruited 38 participants to answer a survey through convenience sampling and screened their eligibility according to the inclusion criteria detailed in Table 1.
Instruments and measurements
The survey collected sociodemographic variables and health variables as described below. We conducted a literature search to guarantee relevance to this population.
Sociodemographic variables for caregivers and children with CMC included the age of children with CMC, the number of children and family members at home, the presence of pets at home, children’s hospitalization state, and the maintenance of personal hobbies, among others (Table 1).
Health variables included the participants’ perception of QoL, well-being and disease, and coping strategies displayed to deal with stressful situations. We selected the KINDLR questionnaire to measure HRQoL in children and adolescents [68]; this instrument measures children’s HRQoL through self-reporting and can be used for both healthy children and in clinical practice. We selected this questionnaire after a literature search with the following inclusion criteria: (1) self-reported; (2) availability of versions for children, teenagers, and caregivers; (3) short time completion (10–15 min maximum); (4) questions formulated from a positive life situation; (5) availability of a validated version in Spanish or Catalan, and (6) robust psychometric properties (namely reliability as well as discriminant and construct validity coefficients). The KINDLR questionnaire meets all these inclusion criteria in its five versions: Kiddy-KINDLR for children aged 3–6 years, Kid-KINDLR for children aged 7–13 years, Kiddo-KINDLR for teenagers aged 14–17 years, and the KINDLR questionnaire for caregivers with children aged 3–6 or 7–17 years [69–73]. Its Spanish version shows acceptable reliability and validity coefficients and has been validated as an adequate assessment tool useful in clinical practice [74, 75].
The KINDLR questionnaire consists of 24 Likert-scale standardized items associated with six QoL dimensions [68, 76]: physical well-being, emotional well-being, self-esteem, family, friends, and school (this category includes questions about daily functioning). These six dimensions can be combined to produce a total score for general well-being. Furthermore, all versions of the questionnaire contain an additional subscale named “disease,” which aims to measure children’s QoL concerning their chronic illness and hospitalization. The version of the KINDLR questionnaire for caregivers of children between the ages of 3 and 6 years matches that for caregivers of children and teenagers aged 7–17 years.
The participants were asked about coping strategies through open-ended questions; these strategies were measured following the three-level hierarchical structure model of the Coping Strategies Inventory [64] based on Folkman and Lazarus’ model [77] and validated in the Spanish population [78].
Data analysis
To analyze the data collected in the survey, we conducted descriptive analyses of the perception of QoL, well-being, and disease and of the participants’ coping strategies. We also performed comparisons between the cohorts and transformed qualitative data from coping strategies into quantitative data [79]. In the questionnaire, the participants were asked to write down the activities in which they engaged the most to cope with stressful situations. We analyzed the responses to this open-ended question following the three-level hierarchical structure model of the Coping Strategies Inventory [64] based on Folkman and Lazarus’ model [77]. This hierarchical model organizes coping strategies into two main categories: engagement and disengagement. Each category is then split into two categories: problem-focused and emotion-focused. Consequently, mid-level categories are problem-focused engagement (PFE), problem-focused disengagement (PFD), emotion-focused engagement (EFE), and emotion-focused disengagement (EFD) coping strategies. Each of these four categories is then split into two new categories, representing the final eight categories of coping strategies: problem solving (PS), which involves strategies focused on making and following a plan to overcome the faced challenge; cognitive restructuring (CR), which focuses on reappraising the situation in a way that encourages positivity; expressing emotions (EE), which are strategies focused self-disclosure of emotions and/or engagement in activities allowing people to be in contact with their internal states; social support (SS), which involves participants who turn to their social network to overcome challenges; problem avoidance (PA), which refers to focusing on certain tasks to avoid thinking about the stressful situation or conflict; wishful thinking, which expresses a desire that the situation would disappear or a miracle would happen; self-criticism (SC), which involves criticizing oneself for an event and feeling guilty; and social withdrawal (SW), which refers to avoiding time spent with others and refusing social contact. Three research members coded the responses by consensus agreement until saturation and mutual exclusion were reached [63]. Thus, we transformed the qualitative data into quantitative data [67] and analyzed the possible relationships between different coping strategies and perceptions of well-being.
The statistical analysis included testing the homogeneity of variance of KINDL scores using Levene’s test. Then, we analyzed the differences in KINDL scores for QoL and well-being and the disease variables between cohorts using Student’s t-test, one-way analysis of variance (ANOVA), two-way ANOVA, or two-way ANOVA with repeated measures adjusted for different demographic groups. When significant effects were detected in the ANOVA analysis, the Sidak post-hoc test was applied for pairwise multiple comparisons. We conducted a linear correlation study to analyze the possible relationship between well-being and disease perceptions and between the perception of well-being and the number of hospitalizations. Further, we analyzed the differences in the distribution of coping strategies between caregivers and children using Pearson’s χ2 test. Frequencies were presented as a percentage (%) of the total data collected; data were presented as mean ± standard error of the mean (SEM), and values of p < 0.05 were considered significant. We performed statistical data analyses using the statistical package SPSS Statistics v26 (IBM Corp. USA).
Procedure
The pilot study was conducted in Catalunya, Spain between February 2021 and May 2022; however, data collection was discontinued between February 24, 2021 and January 6, 2022 due to the COVID-19 pandemic. Participants received an invitation to participate in an online survey hosted on UB Forms (University of Barcelona), which took about 15 minutes to complete. Recruitment was conducted through (1) personalized letters sent to the pediatric units of hospitals and patient associations, (2) communication with health professionals, and (3) social media dissemination (LinkedIn, Twitter, Facebook, and WhatsApp). All participants were informed about the survey’s purpose and provided their written informed consent to participate in this study; adults signed their informed consent, and children gave their assent. Recruitment of youth was addressed to parents and caregivers, who eventually gave their consent to their children’s participation in the study. Parents willing to participate received an e-mail with a link to the online questionnaire addressed to the youth, and they were also asked to help their children decide if they gave consent to participate and understand and fulfill the questionnaire. All participants were reminded that they had the right to withdraw at any time, that their participation was entirely voluntary, and that their responses would be kept confidential. The survey did not explore sensitive, private, or political information.
The project’s methods and experimental protocols were evaluated and approved on March 13, 2020 by the Ethics Committee of the University of Barcelona (Spain) before the research began (Institutional Review Board approval number-IRB00003099). The study followed the regulations established by the European Union (EU) 2016/679 of the European Parliament and of the Council from April 27 on the protection of natural persons about the processing of personal data and their free movement. It also followed the Spanish Ley Orgánica 3/2018, from December 5 on the protection of personal data and digital rights.
Results
Selection of participants meeting the inclusion criteria and final cohort
We excluded three (7.8%) surveys due to missing data, and 35 responses remained; 0% of participants answered “I don’t know/I prefer not to answer” in all analyzed variables except for “Maintain her/his hobby” in the caregiver survey, where missing data reached 8.3%. Table 2 shows the participants’ detailed demographic characteristics.
Caregivers’ perception of youth’s well-being and disease
The caregivers’ KINDL score for the perception of their children’s general well-being was 70.40 ± 2.88. We analyzed the differences in the perception of the six dimensions of QoL by the youth’s age groups. In the two-way ANOVA with repeated measures, we found a significant effect of age, with F(1,13) = 8.130; p = 0.0128, but no effect of the QoL dimension or interaction between both parameters, with F(5,65) = 2.222; p = 0.0626 and F(5,41) = 2.133; p = 0.0807, respectively. Therefore, although certain trends are visible, we detected no significant differences between the different dimensions of youth’s QoL as perceived by caregivers. Conversely, caregivers of children aged 3–6 years had a higher perception of QoL in most of its dimensions than the caregivers of youth aged 7–17 years (Fig 2a). This difference was also evident when the KINDL scores for general well-being were compared: the score was 23.4% higher among caregivers of children aged 3–6 years (t = 2.981; p = 0.0069; Fig 2b).
The histograms represent the following: (a) the effects of youth’s age on the caregivers’ perception of the six dimensions of QoL defined in the KINDLR questionnaire; *, p < 0.05, age effect, two-way ANOVA; (b) the effect of youth’s age on caregivers’ perception of youth’s general well-being; **, p < 0.01, different from 3 to 6 years, Student’s t-test; (c) effect of the youth’s hospitalization situation on the caregivers’ perception of the six dimensions of QoL defined in the KINDLR questionnaire; *, p < 0.05, different from physical well-being with hospital discharge, Sidak post-hoc test; (d) the effect of the youth’s hospitalization situation on the caregivers’ perception of the youth’s general well-being; (e) linear correlation analysis between the caregivers’ perception of well-being and that of children’s disease; (f) comparison of the caregivers’ perception of well-being and disease depending on the age group of their children; *, p < 0.05, two-way ANOVA. The histograms present the mean ± SEM.
Then, we analyzed the effect of hospitalization on the KINDL scores of QoL that caregivers gave to their children. A two-way ANOVA with repeated measures revealed significant differences between QoL dimensions, F(5,60) = 2.826; p = 0.0234; without a significant effect of hospitalization status, F(1,12) = 0.090; p = 0.7689; but with a strong interaction between both parameters, F(5,48) = 5.345; p = 0.0006. According to the post-hoc analysis, physical well-being had a lower KINDL score compared with the other dimensions (Fig 2c). In addition, the interaction between both parameters indicated that the group of caregivers of hospitalized youth is responsible for this decrease (Fig 2c). When analyzing the KINDL score for general well-being, the effect of physical well-being was offset by the other dimensions, and no differences were observed when comparing the scores given by the caregivers of hospitalized and discharged youth (t = 0.0523; p = 0.6056; Fig 2d).
Among the participants, 13 caregivers stated that their children were hospitalized, and answered the KINDL questions about disease perception with a mean value of 56.30 ± 4.79, which was 25% lower than the score obtained for the caregivers’ perception of youth’s well-being (t = 2.504; p = 0.0123). Then, we analyzed the effect of the children’s age on the differences between well-being and disease perceptions. The two-way ANOVA showed differences between the perceptions of well-being and disease of caregivers, F(1,33) = 5.754; p = 0.0223, and also a significant effect of age, F(1,33) = 6.705; p = 0.0142, but no interaction between both parameters, F(1,33) = 1.449; p = 0.2372. This indicates that caregivers of children aged 3–6 years had higher perceptions of both the well-being and disease of their children than those of youth aged 7–17 years (Fig 2f). In particular, we performed a correlation study between the perception of well-being and the perception of disease presented by the caregivers of hospitalized youth and found no linear relationship between both parameters (r2 = 0.0031; p = 0.854), which suggests that caregivers’ perceptions of well-being and disease are independent, without a clear relationship between them (Fig 2e).
Finally, we performed a demographic study and analyzed the caregivers KINDL scores for well-being and disease based on whether their children with CMC had siblings, lived with other family members, or had a pet at home, and whether caregivers maintained their hobbies (Table 3). We found no differences in the perception of well-being among caregivers depending on whether the children had siblings; however, we detected a greater perception of youth’s well-being among caregivers who lived without other relatives at home, those who did not have pets at home, and those who kept their hobbies (Table 3). In all these situations, we found no significant differences in the caregivers’ perception of the youth’s disease (Table 3).
Youth’s perception of their own well-being and disease
We analyzed the differences in the perception of the six QoL dimensions by the youth’s age groups, and in the two-way ANOVA with repeated measures, we found no significant effect of age, F(1,6) = 3.103; p = 0.1286; QoL dimensions, F(5,30) = 1.870; p = 0.1293; or interaction between the two parameters, F(5,11) = 0.701; p = 0.6341 (Fig 3a). However, when the KINDL scores for general well-being were compared, we observed this score being 28.7% smaller in the group of youth aged 7–17 (t = 2.591; p = 0.0292; Fig 3b). The KINDL score indicating the perception of own well-being of the 11 youth participating in the study gave an average value of 69.5 ± 5.9.
The histograms represent the following: (a) the effect of age on the perception of the six dimensions of QoL defined in the KINDLR questionnaire; (b) the effect of youth’s age on their own perception of general well-being; *, p < 0.05, different from 3 to 6 years, Student’s t-test; (c) the effect of gender on the perception of the six dimensions of QoL defined in the KINDLR questionnaire; (d) the effect of gender on the perception of general well-being; (e) linear correlation analysis between the youth’s perception of well-being and the number of hospitalizations; (f) comparison of the perceptions of well-being and disease of the hospitalized 7–17-year-old youth; *, p < 0.05, different from well-being, Student’s t-test. The histograms present the mean ± SEM.
We also analyzed the possible differences in QoL perception based on the youth’s gender. In the two-way ANOVA with repeated measures, we found no significant effect of gender, F(1,6) = 0.075; p = 0.7930; of the QoL dimensions, F(5,30) = 1.307; p = 0.2847; or of the interaction between both parameters, F(5,11) = 0.284; p = 0.9117 (Fig 3c). Furthermore, we also found no differences when comparing the general well-being scores between females and males (t = 0.319; p = 0.7564; Fig 3d).
Next, we analyzed the effect of hospitalization on the youth’s perception of their own QoL and well-being. To estimate the possible influence of hospitalizations on youth’s well-being, we conducted a correlation analysis between their perception of well-being and the number of hospitalizations they had experienced. We did not find a linear correlation between the parameters (r2 = 0.033; p = 0.6737; Fig 3e), which indicates that, under our conditions, the youth’s perception of well-being did not have a clear linear relationship with the number of hospitalizations they experienced. The KINDLR questionnaire for children aged 3–6 years has no disease section; consequently, we compared perceptions of well-being and disease only in hospitalized youth aged 7–17 (Fig 3f). In this group, the KINDL score for the disease perception gave an average value of 38.88 ± 5.82, which is 33.1% smaller than the score obtained for the perception of well-being in the same age group (59.22 ± 6.89; t = 3.061; p = 0.0376).
Finally, given that 93% of youth with CMC participating in the study had siblings, we could not estimate the effect that this presence had on their perception of well-being. We analyzed the effect of the presence of pets at home and found no influence on this perception (KINDL score 71.04 ± 7.16 for pets at home and 65.30 ± 10.11 for no pets at home; t = 0.430; p = 0.6769).
Comparison of youth’s perceptions of their well-being and disease with the caregivers’ perceptions
The version of the KINDLR questionnaire for children aged 3–6 years differs from those of the other youth groups in the number of items and type of scale. However, the versions of the questionnaire for youth aged 7–17 years and for caregivers quantify the perception of QoL, well-being, and disease equally. Therefore, we made a comparative analysis between the KINDL scores of youth aged 7–17 answering the questionnaire (n = 7) and the scores of caregivers of youth of the same age (n = 12). We first compared the KINDL scores of both groups in each of the six QoL dimensions (Fig 4a). The two-way ANOVA analysis with repeated measures showed no differences between the perception of youth and that of caregivers, F(1,19) = 0.330; p = 0.572, but it showed a significant effect of the QoL dimension, F(5,95) = 3.543; p = 0.006, without interaction between both parameters, F(5,95) = 0.329; p = 0.894. This indicates that youth have a perception of their own well-being similar to that of their caregivers (Fig 4a). In addition, both groups present differences in their perceptions of the QoL dimensions. Specifically, the family dimension had the highest KINDL score (77.11 ± 4.47); this was significantly greater than that of the school dimension (p = 0.021; Sidak post-hoc), which comparatively presented the lowest score (52.99 ± 5.08).
The histograms represent (a) a comparison of the perception of the six QoL dimensions defined in the KINDLR questionnaire between caregivers and youth aged 7–17 years; *, p < 0.05, Sidak post-hoc; a comparison of the perception of general well-being (b) and disease (c) between caregivers and hospitalized youth aged 7–17; *, p < 0.05 different from caregivers, Student’s t-test. Histograms show the mean ± SEM.
Subsequently, we compared the perceptions of general well-being and disease presented by hospitalized youth aged 7–17 (n = 5) with those of caregivers of youth of the same age (n = 11). Under our conditions, we did not observe differences when comparing the general well-being scores between both groups (t = 1.529; p = 0.1457) (Fig 4b); on the contrary, we found that the perception of disease in youth was 34.2% lower than that detected in caregivers (t = 3.228; p = 0.0061; Fig 4c).
Relationship of well-being perception and coping strategies
All participants in the study showed various coping strategies to deal with stressful situations. Most of the strategies reported by caregivers belonged to the PA and CR categories (40.9% and 36.4% of the total, respectively), while EE strategies were reported in 18.2% of cases and SS in 4.5%. Interestingly, caregivers engaged in very few EFD activities, with no SC and SW strategies reported (Fig 5a). Coping strategies used by youth differed from those of caregivers: SS was greatly used by 36.4% of youth, followed by SW and PA strategies, each with a frequency of 27.3% (Fig 5a); finally, 9.1% engaged in CR, and 0% reported PS and SC strategies.
Analysis of the relationship of well-being perception with the coping strategies displayed to deal with stressful situations. (a) Graphical representation of the percentage of participants in each coping strategy category (see text for details). (b) Histograms of the contingency tables showing comparisons of coping strategies by group of participants; *, p < 0.05 values greater than expected, #, p < 0.05, values smaller than expected, Pearson’s χ2. (c) Histograms show the KINDL score of the perception that caregivers have of their children’s well-being for every category of coping strategy. (d) Histograms show the KINDL score of the youth’s perception of well-being for every category of coping strategy. Eng. = engagement, Diseng. = disengagement; PF = problem-focused; EE = emotion-focused; PFE = problem-focused engagement; EFE = emotion-focused engagement; PFD = problem-focused disengagement; EFD = emotion-focused disengagement.
We compared the frequencies of the strategies reported by caregivers and youth and found statistically significant differences in the frequency distribution of the groups (χ2 = 51.421, p < 0.0001; Fig 5b). Among all the differences in distribution, only caregivers reported EE strategies, and only youth displayed SW strategies. In addition, youth reported adopting SS strategies more frequently, while caregivers adopted CR strategies (Fig 5b). When we analyzed the frequency of coping strategies classified by secondary and tertiary scales, we also observed differences. Thus, youth displayed emotion-focused strategies (χ2 = 40.888, p < 0.0001) and disengagement strategies (χ2 = 35.738, p < 0.0001) more frequently than caregivers, who mostly displayed problem-focused strategies (Fig 5b); consequently, EFD and PFE strategies were more frequent among youth than among caregivers (χ2 = 44.731, p < 0, 0001).
Then, we analyzed the possible differences in the well-being KINDL score of youth associated with coping strategies. The one-way ANOVA showed no differences in the KINDL scores of caregivers’ perception of the youth’s well-being in any of the coping strategy classification levels (Fig 5b); thus, we found no differences in the mean KINDL score between coping strategies when classified into eight categories, F(3,19) = 0.184, p = 0.906. This was also true for either the comparison between engagement and disengagement strategies, F(1,19) = 0.214, p = 0.809; the comparison of emotion-focused vs. problem-focused strategies, F(1,21) = 0.271, p = 0.766; or middle-level classification, F(3,18) = 0.242, p = 0.866. Similar results were obtained when we analyzed the effect of coping strategy on the youth’s perception of their own well-being. We observed no effect of the coping category in the mean KINDL scores—F(3,10) = 0.251, p = 0.859—of the eight coping strategies: F(1,9) = 0.758, p = 0.406 for the disengagement vs. disengagement classification; F(1,10) = 0.124, p = 0.733 for emotion-focused vs. problem-focused classification; and F(3,10) = 0.251, p = 0.859 for middle-level classification (Fig 5c).
Discussion
This pilot study compared the perception of children and adolescents with CMC in the situation of hospitalization and convalescence and that of their caregivers. Our results support the beneficial effects of the family environment on youth’s QoL; the main findings, in particular, support the independent relationship between caregivers’ perceptions of well-being and disease. In addition, the perception of well-being in children with a CMC impacts the way they view, relate, and communicate with their immediate environment.
Regarding QoL perception, we found no significant differences between the QoL perceived by youth and that perceived by their caregivers, which means that caregivers view youth’s QoL according to how they view themselves. This is a highly positive finding as research has previously shown that parental catastrophizing leads to parental overprotection [80], higher child’s disability and low school attendance [81, 82], and restriction of their children’s ability [83]. Caregivers of children aged 3–6 years had a higher perception of well-being in most of its dimensions than caregivers of youth aged 7–17 years, and these lower levels in the older cohort are also found in the measures of youth’s perceived own well-being. A possible explanation to this finding is that as children become adolescents, youth’s limitations due to their illness become more obvious. This interpretation could also explain why physical well-being is reported as the lowest out of all other dimensions. Nevertheless, we did not find a correlation between the caregiver’s perceptions of well-being and disease.
Furthermore, we found no differences in the caregivers’ perception of well-being depending on whether the children had siblings; however, we detected a greater perception of youth’s well-being in caregivers who lived without other relatives at home, those who did not have pets at home, and those who kept their hobbies. This could be the result of added stress as the household dynamics get more complex: a pet adds another dependent entity, and relatives at home may be an added financial burden, especially if they are grandparents who need care. Three-generational households may strengthen perceived social support, but the positive effects may be dependent on the quality of the relationships and the financial burden [84].
When we explored coping strategies, we found that youth coped with stressful situations by engaging in the social support category, followed by strategies of the social withdrawal and problem avoidance categories. Interestingly, no youth reported strategies in the express emotions category. Types of coping strategies can influence the HRQoL of youth with CMC. For example, in a previous study, adaptive coping styles and social support were negatively correlated with psychological distress and anxiety in children with malignant tumors [85]. Additionally, we found no clear relationship between the youth’s perception of well-being and the coping strategies displayed. However, some tendencies were observed as youth displaying strategies of emotion-focused engagement—particularly social support—presented slightly lower well-being scores than youth presenting problem-focused disengagement, particularly problem avoidance. These results align with the previous literature reporting that children with CMC who express emotional reactions are negatively related to HRQoL [86, 87], while acceptance and avoidance are positively correlated [87, 88].
We found substantial differences between caregivers and youth in the coping strategies used to deal with stressful situations. Most of the strategies that caregivers engaged in belonged to the problem avoidance, cognitive restructuring, and—to a lesser extent—express emotions categories, while barely reporting emotion-focused disengagement strategies, which indicates that in our sample, caregivers preferred to display problem-focused strategies that individually manage the stressful situation than emotion-focused strategies that involve receiving understanding and comfort from others. Previous studies support this finding (e.g., [89]). Although this preference for problem-focused strategies may be associated with lower well-being outcomes [90], studies have reported contradictory effects of coping styles on caregivers’ psychological distress. For example, increased social support has been associated with lower caregiver motivation in parents of youth with type 1 diabetes [91]; in contrast, emotional support has also been associated with the well-being improvement of caregivers of children with rare diseases [92]. Indeed, fostering caregivers’ social support and cognitive restructuring strategies may be relevant in increasing parenting self-efficacy to successfully address children’s illnesses by helping them in emotion management [93]. As increased parental self-efficacy in health contexts has been reported to improve caregivers’ confidence and self-esteem [94], cognitive restructuring may constitute an adaptive coping strategy for improving caregivers’ well-being.
The differences between coping styles displayed by caregivers and youth emphasize that they do not tend to cope with stress jointly, which is an important finding because the way in which youth and caregivers deal with a CMC is not only affected by disease-related stress but also by stress perceived from each other [95]. Children’s low emotional functioning particularly affects the psychosocial aspects of the caregivers’ QoL, which in turn determines the way that the child confronts the CMC. Indeed, family-based support and affection are derived from QoL and well-being [96]. In addition, reinforcement of the bonding integration and connectivity between family members can enhance QoL [97]; thus, dyadic coping [98] emerges as an important agent to deal with the CMC-related stress of both youth and caregivers, although it has still been poorly studied in child–caregiver relationships. However, a study reported that emotion-oriented dyadic coping has been associated with HRQoL in children with CMC [95]. Moreover, children’s physical well-being has been linked to the caregivers’ use of acceptance coping strategies [86] commonly displayed by caregivers in our study (cognitive restructuring strategies). However, the same study reports that this acceptance coping is not positively linked to the caregivers’ own psychosocial well-being [86]. Nevertheless, communication between youths and caregivers appears crucial in understanding each other’s feelings and in providing appropriate dyadic coping with positive effects on the children’s HRQoL and well-being. Furthermore, helping children and caregivers to jointly deal with CMC-related stressful situations may not only empower youth and benefit their HRQoL, but it may also improve the caregivers’ QoL.
Limitations and future directions
This pilot study has certain limitations. First, sampling was non-randomized with the convenience selection criterium. Moreover, as recruitment was discontinued due to COVID-19 restrictions, the sample was small, which lessened the power of the statistical analysis of certain parameters, such as the illness perception of children aged 3–6 years and the frequency of some coping strategies. In the analysis of these parameters, the study’s statistical power only reached 29.5%, which may mask possible differences and crossover effects between the groups. Nonetheless, the sample was sufficient to achieve statistically significant results (25% effect with a significance level of 0.05) in the perception of well-being and identification of coping strategies. In this line, this pilot study helps to establish the sample size and characteristics for further larger studies analyzing the HRQoL of children with CMC. Moreover, as the surveys were accessed online, families without reliable access to the Internet or an electronic device may have been missed. Finally, self-reported responses may have been impacted by a poor view of the participants’ internal states; however, using a survey tool to measure well-being and psychological distress is a common practice in this field.
The understanding of hospitalization and care processes constitutes a barrier for the QoL and well-being of youth with CMC and their caregivers as the quantity of complex information to assimilate is overwhelming [99–101]. Personalized actions to improve communication with youth with CMC and with their caregivers are necessary to improve their perception of family well-being and help them display adaptive coping strategies [102, 103]. The literature includes various studies addressing the impact of hospitalization in CMC from the youth’s point of view (e.g., [104]) on their needs (e.g., [105]) and supportive care (e.g., [106]). However, a gap still exists with regard to some concrete issues, such as hospitalization and readmission [107] or isolation [104], and the same is true for suffering, spiritual pain, or emotional stress regarding further procedures—especially those involving needle procedures [108]. Future studies should conduct comparative longitudinal studies with and without intervention-related parenting and child- and family-centered care, which would allow to see the features that improve QoL and well-being in the family group.
Conclusions
Our findings show the paramount importance of evaluating the bidirectional impact of the experiences of youth with CMC and their support system; furthermore, they highlight the consequences that such dynamics have on the QoL of this youth. So far, little research has asked youth to participate directly, and investigations have mostly revolved around health professionals and caregivers. This preliminary study shows the importance of directly exploring the experience of youth as well as facilitating communication spaces with both the family and health professionals where the voice of children is considered. In addition, coping strategies differ between caregivers and youth; therefore, mental health professionals should recognize these differences and work accordingly. Finally, policymakers should integrate these findings when developing and implementing guidelines around the best practices of pediatric care. Our findings recognize and reiterate the importance of listening to the voice of children and adolescents and their families regarding pediatric care practices. We hope that this pilot will spearhead the development of further research exploring the experiences of children in the same way that certain research projects have been doing with parental experiences [109]. The literature in this field can positively impact the way health professionals care for patients and the quality of the healthcare system.
Acknowledgments
We warmly thank all the marvelous youth and families that kindly and selflessly participated in this pilot study, and are very grateful to M. Paula Céspedes (LaCardio–Fundación Cardioinfantil, Colombia) for helpful scientific advice and discussion and her participation as a member of the research team. We also acknowledge the support of the Department of Research and Universities of the Generalitat de Catalunya to the Group of Research and Innovation in Designs (GRID) Technology and multimedia and digital application to observational designs (Code: 2021 SGR 00718; VVH as a member, and SMV as an external collaborator) and to the group of Molecular Mechanisms and Therapeutical Approaches in Brain Pathologies (Code 2021 SGR 01086; MJR as a member). We also thank the International Observatory in Hospital Pedagogy, University of Barcelona, Spain.
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