Introduction
Paediatric heart failure is a complex and burdensome condition that carries an incidence of 0.9–7.4 per 100,000 children. Reference Shaddy, George and Jaecklin1,Reference Peng, Rosenthal, Zafar, Smyth, VanderPluym and Lorts2 For children with CHD admitted to the hospital with severe heart failure, in-hospital mortality reaches 26%. For those awaiting heart transplantation, 22% will die before an organ becomes available. Reference Peng, Rosenthal, Zafar, Smyth, VanderPluym and Lorts2 Despite the increased use of advanced therapies and ventricular assist devices, there remains a high burden of morbidity and mortality among this population, Reference Peng, Rosenthal, Zafar, Smyth, VanderPluym and Lorts2 as well as impaired quality of life. Reference Cousino, Lim and Smith3 Understanding modifiable risk and resilience factors is critically important to improving physical and psychosocial outcomes in paediatric heart failure.
Psychological health and well-being is one such modifiable factor. Both negative and positive psychological health have important implications for cardiovascular health and cardiac-related outcomes. Reference Levine, Cohen and Commodore-Mensah4,Reference Kovacs, Brouillette and Ibeziako5 In the general population, negative psychological health, such as anxiety and depression, has been associated with an increased risk of acquired cardiovascular disease. Reference Levine, Cohen and Commodore-Mensah4 Research to date has largely focused on characterising risk of negative psychological health in paediatric heart disease. Among the general CHD population, significantly higher rates of depression, anxiety, post-traumatic stress, and medically related fear are seen in comparison to healthy counterparts. Reference Gleason, Deng and Khan6–Reference Kovacs, Sears and Saidi12 High rates of mental health diagnoses and significant patient and family psychosocial distress have been recognised in paediatric ventricular assist device and heart transplant populations. Reference Rea, McCormick, Lim and Cousino13,Reference Cousino, Schumacher and Rea14
Beyond mental health morbidity, health-related worries and stressors are understandably notable for patients and families living with CHD. Several studies have catalogued the great amount of worry that parents of individuals with CHD have for their children. This worry is not limited to the time of diagnosis but often expands to the future health of their child and of the rest of the family. Reference Biber, Andonian and Beckmann15–Reference Docherty, Miles and Holditch-Davis19 Similar findings are seen in those living with CHD. One study of young patients with CHD reported that 71% of individuals experienced anxiety, and 55% experienced worrisome thoughts, leading to a disruption in daily living. Reference Strange, Stewart and Farthing20
It is important to recognise that while mental health morbidity and health-related worries are common, these risks and experiences are not ubiquitous. Many patients do not experience negative psychological health in the setting of their advanced heart disease. Promotive and protective factors for positive psychological health, however, have not been well studied in paediatric heart disease. Resilience, which has been defined as the process by which one harnesses internal, external, and learned resources to cope with adversity, is one facet of positive psychological health. Reference Connor and Davidson21,Reference Rosenberg and Yi-Frazier22 Studies in patients with chronic illnesses and their caregivers have demonstrated that both patient and parental resilience is associated with better quality of life, greater social support, and lower distress. Reference Lau, Yi-Frazier, Bona, Baker, McCauley and Rosenberg23–Reference Teixeira, Coelho and Proença27 In a study of 332 individuals with CHD (17.2 ± 5.1 years), higher resilience was correlated with fewer hospital admissions, lack of a mental health diagnosis, increased exercise, and participation in peer support groups or disease-specific camps. Reference Glenn, Cousino, Wernovsky and Schuchardt28 In paediatric patients living with other non-cardiac chronic illnesses, resilient individuals were more likely to have higher transition readiness and better disease control. Reference Verma and Rohan29–Reference Yi-Frazier, Yaptangco and Semana31
While one’s resilience processes and resources are likely to have important implications for both psychological and physical health in young people with heart disease, the concept is largely understudied within this patient population. This federally funded single-center study aimed to qualitatively describe how adolescents and young adults with heart failure view their own personal challenges, worries, coping skills, and resilience. Parental perspectives on their child’s coping and resilience were also obtained. Due to the qualitative nature of this study, there were no apriori hypotheses. The overall objective of this qualitative investigation was to inform the design of patient-centered interventions focused on well-being, resilience, and positive psychological health paediatric advanced heart disease, as no psychological interventions have been developed or piloted in the growing paediatric heart failure population.
Methods
Study design and population
This qualitative methods study utilising semi-structured interviews was approved by the local Institutional Review Board. Single site recruitment was completed at a U.S.-based Midwest children’s hospital. Patients (12–24 years) with heart failure symptoms meeting criteria for NYHA Classes II–IV heart failure were eligible (i.e., limitations in physical activity, fatigue, palpitations, dyspnoea). Parents of patient participants meeting this inclusion criteria were also eligible. Adolescent and young adult patient and parent participants were excluded if (a) they had experienced suicidal intent or symptoms of psychosis in the past 6 months, (b) had moderate cognitive impairment or developmental disability (e.g., IQ below 55 per self, parent, or provider report), or (c) were not English-speaking. In addition, adolescent and young adult participants who (d) were intubated, unable to respond verbally, or with active delirium as diagnosed by a physician were excluded.
Eligible patients were identified and approached following a thorough review of our institutions heart failure and transplant outpatient clinic and inpatient cardiology census by the research team. After identification, we obtained approval from the patient’s primary cardiology care team before approaching the individual. For those patients who were interested, enrollment occurred in both the inpatient and outpatient setting. Patients of research team members were eligible for study participation; however, study enrollment procedures and the interview were conducted by a study team member who was not active in the care of these patients.
Purposive sampling Reference Etikan32 was utilised to recruit approximately: (1) 8 adolescent patients ages 12–16 years of age, (2) 8 adolescent/young adult patients ages 17–24 years of age, and (3) 14 parents of adolescent/young adult patients enrolled in study as well as to ensure diverse representation in the study sample. Adolescent and young adult-only or parent-only participation was permitted. A total sample size of 30 was targeted for achieving theme saturation. Reference Cousino, Miller and Smith33 The COREQ (COnsolidated criteria for Reporting Qualitative research) Checklist was used for describing methods and reporting results (Supplementary File).
Methods and measures
Written assent/consent was obtained from all participants and legal guardians prior to interview initiation. Semi-structured interviews were conducted by a nurse research coordinator across a variety of settings depending on patient location (i.e., inpatient cardiology room, cardiology clinic consult room, HIPAA-compliant telemedicine). Participants were reminded of the general overall purpose of the study as well as the interviewer’s role (i.e., nurse research coordinator) at the start of the interview. All interviews were audio recorded and transcribed for analysis purposes. Duration of interviews ranged from approximately ∼ 30–60 minutes. The semi-structured question guide was developed by the study team as informed by the literature and a related survey-based study in adolescents and young adults with heart failure Reference Hsieh and Shannon34 with additional review from experts representing psychology, cardiology, palliative medicine, developmental/behavioral paediatrics, adolescent medicine, and bioethics. The drafted question guide was also reviewed by patient/family volunteers with feedback obtained. Questions were developed to be short, open-ended, one-dimensional, and without medical jargon. Reference Cousino, Miller and Smith33 The adolescent and young adult interview included 12 question prompts, while the parent interview included 14 question prompts. Questions specific to this current analysis focused on (a) worries, (b) challenges, (c) sources of strength/support, (d) coping strategies, (e) defining resilience, and (f) fostering resilience. Caregiver interview analysis was focused on their perceptions of the patient’s worries, coping, and resilience. Interview transcriptions were stripped of personal identifying information to protect each participant’s identity before content analysis. Each patient and caregiver participant received a $50 gift card for compensation of their time completing study interview and the demographic/background survey.
Analysis
Descriptive statistics (i.e., frequencies and percentages) were performed to describe the sample. Content analysis was utilised to code interview data. Reference Glaser35 Three authors (TG, CS, MC) independently read each of the interview transcripts to obtain a holistic view of the data. Themes were generated using the constant comparative method, Reference Steiner, Abu-Rish Blakeney and Corage Baden36 and supporting quotes were identified. A coding book was developed by the senior author and study PI (MC) within Microsoft Excel. All transcripts were coded by two independent coders (TG, CS). Discrepant coding was reviewed and discussed. Reliability coding was completed on 20% of transcripts to enhance validity by a third coder (MC).
Results
Participants
A total of 30 participants completed the study (N = 16 adolescent and young adult patients with heart failure and N = 14 caregivers). A total of 23 eligible patients were approached for the study with 16 consenting to study participating (70% enrollment rate). The most common reason for declining study participation was “not interested in participating in any research studies.” Among patients screened and deemed not eligible per pre-determined criteria, the most common reason for ineligibility was developmental delays impacting study participation (i.e., non-verbal). Consenting patient participants ranged in age from 13 to 22 years (M = 17.06; SD = 3.26), and 63% (N = 10) identified as male. Five patient participants identified as non-Hispanic Black, while one participant identified as Hispanic, multi-racial (37.5% self-identifying with racial or ethnic minority group). The majority of patient participants (62.5%) had heart failure in the setting of CHD, while the remaining participants (N = 6; 37.5%) had a primary cardiomyopathy diagnosis. Patient and caregiver demographics are shown in Tables 1 and 2.
Patient demographics
Caregiver demographics
Fourteen caregivers completed the study (N = 9 biological mothers, N = 1 stepmother, N = 4 biological fathers). Caregivers described their families as married/both parents at home (N = 7, 50%), mixed family home (N = 4, 29%), or single parent home (N = 3, 21%). A subset of caregivers (29%) reported a family income below the Federal Poverty Level ( <$25,000 in 2023), while one patient-only respondent reported the same, resulting in approximately 1/3 of the sample indicating living among impoverished living conditions. Among known parental education levels reported (N = 26), 5 parents had not completed high school (19%) and 9 parents held a Bachelor’s, Master’s, or Doctoral-level degree (35%).
Worries and challenges among adolescents and young adults with heart failure and their caregivers’ perception
Adolescents and young adults with heart failure reflected on worries and challenges that they face personally, while caregivers of adolescents and young adults shared their perceptions of worries and challenges their children face (Table 3). At times, given the semi-structured nature of the interview, parents went on to discuss their own worries and coping. Over half of the adolescents and young adults with heart failure (8/15, 53%) responded that living with heart disease causes little or no difficulties in their life. One participant responded, “I have nothing to compare it to, so it’s just normal for me.” In contrast, nearly all caregivers (12/14, 86%) responded that their child had a moderate or severe amount of difficulty in their life. When asked about specific worries that adolescents and young adults with heart failure experience, 30% of patients responded with having no specific worries. One participant responded, “I have no worries; when you’re sick you look at life differently.”
Examples of illustrative quotes
Causes of worry
For adolescents and young adults who acknowledged having worries, nearly half responded that major surgeries, hospitalisations, and therapies (e.g., interventions) were their main cause of concern (6/14, 43%). In response to what their main worry is, one patient respondent noted, “Everything. I’m scared I’m going to get into like a really, really sick state or you know this is going to go on worse and worse and I’m really not going to be able to do anything because it’s hard for me to do a lot of stuff right now. All these medical procedures and stuff… the transplant just very, very much worries me.” Caregivers generally perceive their children worry most about major complications such as stroke, death, or life-limiting adverse events (6/17, 35%). In response to what worries their child with heart failure most, one caregiver stated, “[Name] has talked about his/her/their funeral, he/she/they have talked about what he/she/they wants done with his/her/their body and he/she/they wants me to grant all his/her/their wishes [crying] and that’s really hard for him/her/them.” No caregiver reported their child as having no worries, despite 30% of patients reporting no worries specific to their heart disease.
Challenges faced
When asked about the challenges that they face, adolescents and young adults with heart failure mostly indicated declining physical health and ability as their biggest challenge (7/17, 41%). “One step forward, three back(wards),” as one participant recalled. Another common theme among patients was “not being able to do everything that my friends can do.” Several reported that their ongoing health concerns and challenges negatively impacted their peer relationships and connections with others (4/17, 24%). This was a major challenge that caregivers also perceived in their child, with most responses citing their child’s inability to do things that their peers do as a notable challenge they face (8/22, 36%). One caregiver recalled, “his/her/their biggest challenge is trying to keep up with his/her/their friends who do not have heart issues. You know, they’re playing sports, they’re swimming, they’re doing this, they’re doing that and then he/she/they can’t do all that and he/she/they wants to be out there with them….”
Support and coping among adolescents and young adults with heart failure and their caregivers’ perception
Adolescents and young adults with heart failure and their caregivers answered open-ended questions about their support system and coping (Table 3).
Support systems
All participants, both patients and caregivers, were able to identify one or more sources of patient-specific support. There was a strong consensus among both patients (26/32, 81%) and caregivers (17/23, 74%) that family and friends provided the highest level of support to adolescents and young adults with heart failure. One patient spoke on their support, “My family is great, my parents have been there with me through my entire life, making sure my health was a priority was something that I’m super thankful for.” On support systems for their child, one caregiver responded, “Through us, he/she/they are pretty open…he/she/they just talks to us and his/her/their friends at school…his/her/their sister is his/her/their rock.” Nearly 16% of all patient responses included their healthcare team as a major source of support. “I have a good support team, but mainly it’s the people at the hospital, the doctors and nurses, the people I’ve had on my team since like two (years old).”
Coping mechanisms
Both patient (9/19, 47%) and caregiver (9/21, 43%) respondents reported that adolescents and young adults with heart failure mostly cope through hobbies, music, art, and activities. “Music and puzzles, poetry, anything that allows me to express my emotions and I think puzzles are… it allows me to turn my brain off. So, it allows me to forget that I’m in the hospital or I’m sick or something like that,” one patient responded. Notably, three adolescent and young adult participants endorsed having no coping skills while one said, “I just push those feelings away.” Only one caregiver perceived their child to have no coping skills.
Resilience among adolescents and young adults with heart failure and their caregivers
Resilience defined
Initially, half of the patient respondents were unable to define resilience; however, after a definition was provided by the interviewer, all understood the term and were able to discuss their viewpoints and experiences specific to resilience (Table 3). Most (5/10, 50%) patient reflections on the term focused on “strength, fighting, and sticking with it”. Two reflected on their definition of resilience, “…somebody who can bounce back from a lot of stuff, someone who’s, who’s been through a lot of stuff but also they know that it’s just another obstacle in the road” and “…not letting things get to you or being strong.”
Examples of resilience
Both patients and their caregivers were prompted to reflect on a time during which the adolescents and young adults demonstrated resilience. Nearly all patient and caregiver responses focused on an experience related to a medical therapy, surgery, or hospitalisation. When asked to recall a time their child demonstrated resilience, “always” was a noteworthy response among caregivers of adolescents and young adults with heart failure (3/11, 27%).
Final advice to others
Patients were questioned about how other individuals with a similar heart condition to theirs could foster and build resilience. All patients were able to provide responses, with the majority of them focused on remaining positive and optimistic (7/14, 50%), which in turn would build resilience. Two patients recommended, “…just live how you want to” and “…just stay the course. It’s a tough road but in the end I’m very normal.” Seeking family and peer support was another common theme that adolescents and young adults reported as a way to build resilience. Patients were lastly asked for any final advice from others who are experiencing similar journeys (Table 3), with one patient summarising their experience, “It gets easier, it gets easier. After a while you kind of just, it becomes your normal routine. It’s a part of our life, it’s the life we were dealt, and it was the cards we were dealt and we have to play the hand we were given. It’s going to take a long time, it’s going to be a long road but it gets easier…a lot easier.”
Discussion
Adolescents and young adults with heart failure face a variety of challenges, both medically and psychosocially. While risks for both medical and psychological comorbidities are high among this patient population, we also know negative outcomes are not ubiquitous. Reference Peng, Rosenthal, Zafar, Smyth, VanderPluym and Lorts2,Reference Cousino, Lim and Smith3 It is important to increase the understanding of promotive and protective factors with regards to psychological health, particularly given associations between mental health and cardiac outcomes. Reference Levine, Cohen and Commodore-Mensah4,Reference Kovacs, Brouillette and Ibeziako5 To our knowledge, this is the first study among a racially and socioeconomically diverse sample of adolescents and young adults with heart failure and their caregivers to gain insight into how they view their own personal challenges, difficulties, coping skills, and resilience through qualitative methods.
In the current study, patients often defined resilience as “being strong,” “bouncing back,” and “persevering through any situation.” Patients generally had positive thoughts about the term and were often able to describe times that they demonstrated resilience throughout their medical journey, which is similar to findings supported by a recent qualitative study on resilience in adult CHD patients. Reference Moreira, Bouissou Morais Soares, Teixeira, Simões e Silva and Kummer37 Perhaps most notably, findings demonstrated that a majority of adolescents and young adults with heart failure are, by their own definition, resilient, with more than half indicating that living with advanced heart disease causes little or no difficulties in their life. This finding is supported by a recent study which demonstrated that individuals living with CHD scored higher on a commonly used measure of resilience when compared to their healthy counterparts Reference Glenn, Cousino, Wernovsky and Schuchardt28 Similar findings were seen in a cohort of children and adolescents with chronic kidney disease with resilient outcomes being similar between those with chronic kidney disease and their healthy controls. Reference Yi, Zebrack, Kim and Cousino38
Prior research on the concept of post-traumatic growth, or the positive psychologic change that can occur in the face of a traumatic event, may provide insight into the current study findings. Reference Zebrack, Kwak and Salsman39–Reference Patel, Lai, Goldfield, Sananes and Longmuir41 In our study, patients and their caregivers were able to easily recall a time during which they were resilient, and in almost all cases, the ‘identifiable event’ was related to a medical procedure or adverse event. This begs the question as to whether those who experience a traumatic health-related event, which many of the study patients described, can in turn, develop positive coping strategies and resilience-building processes. In adolescents and young adults with cancer, post-traumatic growth was observed in many subjects as a result of cancer-related stressors. Reference Zebrack, Kwak and Salsman39–Reference Patel, Lai, Goldfield, Sananes and Longmuir41 The authors of this study posited that these events may partially foster resilient processes and outcomes, in comparison to other ‘healthy’ individuals, who may not experience such events.
Caregivers of study patients, in contrast, perceived their child with heart failure as experiencing higher overall heart disease-related burden and disease-specific worries and difficulties. This finding has been supported by others who demonstrated discrepancies in youth and parent reports of psychosocial quality of life and emotional health. Patel and colleagues revealed that among children with CHD, lower parent–child agreement was observed specific to emotional functioning, internalising behaviors, and anxiety, similar to our findings. Reference Uzark, Jones, Slusher, Limbers, Burwinkle and Varni42 In other samples of children and teenagers with CHD, discrepancies between patient–parent dyads in specific indices, such as psychosocial quality of life and emotional functioning, have been observed. Reference Berkes, Pataki and Kiss43,Reference Cousino, Pasquali and Romano44 Further, significant discrepancies were noted between what adolescents and young adults with heart failure desired in terms of their healthcare communication and medical decision-making compared to what their parents perceived they wanted, perhaps suggesting that many patients with advanced heart disease “can handle more” than adults in their life may perceive. Reference Hsieh and Shannon34
Among both patients and caregivers in our study, a support structure was identified as an important component to adolescent and young adult coping and resilience building. The significance of a support structure and its positive impact on resilience has been studied heavily, both in caregivers of medically complex children Reference Lau, Yi-Frazier, Bona, Baker, McCauley and Rosenberg23,Reference Rosenberg, Wolfe and Bradford24 and in patients with CHD. Reference Silva, Vaz and Areias26–Reference Glenn, Cousino, Wernovsky and Schuchardt28 In patients with CHD, peer support has been identified as an unmet need for a subset of patients. Reference Kovacs, Bendell, Colman, Harrison, Oechslin and Silversides45,Reference Rosenberg, Bradford and McCauley46 Few respondents in our study noted patients as having unhealthy or no identified coping skills. For many, coping strategies often included engagement in hobbies, music, drawing, and art. Survey-based studies have reported interest among young people with heart disease in coping and stress management information/education. In a large international survey of 1,200 patients with CHD and their caregivers, one in three stated that materials specific to promoting their own mental health and coping would be helpful. Reference Kovacs, Bendell, Colman, Harrison, Oechslin and Silversides45 This has been seen in adults living with CHD as well, with a recent study reporting 1/3 of study respondents had interest in coping and managing stress related to their heart disease. Reference Rosenberg, Bradford and McCauley46
Patients were given the opportunity to provide any final thoughts and advice to other patients and caregivers who are navigating the paediatric heart failure journey. Optimism and positivity were common themes among the responses. This finding is analogous to that reported in a recent qualitative study of resilience in adult CHD patients, where themes such as purpose and gratitude commonly emerged among study participants. Reference Moreira, Bouissou Morais Soares, Teixeira, Simões e Silva and Kummer37
Despite recently published findings on the importance of resilience and positive psychological influences among children and young adults with heart failure, no intervention programmes exist at this time to promote resilience or positive well-being in this at-risk population. Among other chronic disease processes, implementation of such programmes has shown promise. Reference Rosenberg, Yi-Frazier and Eaton47–Reference Kovacs, Grace, Kentner, Nolan, Silversides and Irvine49 Within the adult congenital cardiac community, psychosocial interventions have been deemed feasible and potential benefits to individuals’ mindfulness and coping Reference Luberto, Wang, Li, Pagliaro, Park and Bhatt50,Reference Cousino, Dusing and Rea51 has been demonstrated. Results of this qualitative study underscore the importance of community/social support, hobbies (i.e., passion and purpose), optimism, and gratitude in terms of patient coping and resiliency building. Building upon these findings, Cousino et al. recently described the development of the WE-BEAT well-being education programme, which utilises the principles of community, mindfulness and relaxation, purpose and identity, adjustment, and self-gratitude, to foster resilience in paediatric heart disease (52). Although further research is needed to demonstrate the psychological impact of these programmes, we hope that the findings of this current manuscript can provide further insight into the needs of paediatric heart failure patients as it relates to well-being and resilience intervention programmes.
While the present findings can inform future studies and intervention design, we acknowledge several study limitations. Our interviews were conducted with patients and caregivers from a single paediatric heart failure centre in the Midwest. As a result, despite our notable representation of participants across racial/ethnic and socio-economic strata, as well as high representation of paternal caregivers, our results may not be as generalisable in comparison to multi-center, multi-national studies. Heart failure is a heterogeneous diagnosis with a vast range of illness severity, treatment, and disease course. Some of our patient participants were admitted and actively awaiting heart transplant, while others were followed outpatient and prescribed one or two heart failure medications. As such, the heart journey experiences of the participants likely varied a great deal. Illness severity and treatment course were not considered in terms of qualitative analysis. Additionally, the study site has integrated mental health services, including psychology, social work, and child life, for patients with heart disease, and all patients requiring advanced cardiac therapies (i.e., ventricular assist device, transplant) meet with psychology and social work as a standard of care. The role of these psychological supports on patient-reported data in this study is not easily parsed out. Findings may be different across centres or patient populations where there is less access to preventative and promotive mental health care. Lastly, this study was performed in the midst of the SARS-CoV-19 pandemic, a time during which the lives of young people living with heart disease was greatly impacted. Reference Kovacs, Bendell, Colman, Harrison, Oechslin and Silversides45 This may have impacted results.
Despite limitations, this research serves as a foundation for future studies of both risk and resilience specific to psychological well-being in paediatric advanced heart disease. This study adds diverse patient and caregiver voices to this field of science while also emphasising psychosocial experiences at advanced stages of paediatric heart disease, as opposed to focusing on specific diagnostic or lesion groups. Overall, adolescent and young adult patients in our study did not view their life as significantly burdened by their heart failure, especially when compared to parental perceptions. Social support, particularly in family and friends, and positive coping skills, such as maintaining optimism, were noted by most in the study. While it remains critical that mental health morbidity is addressed and mitigated in paediatric heart disease, interventions focussed on identifying, enhancing, and fostering resilient processes and outcomes for both patients with heart disease and their caregivers are the exciting next step for the field.
Acknowledgements
Thank you to the young people and their caregivers who participated in this study and openly shared their lived experiences for the advancement of clinical care and science.
Funding statement
This work was supported by funding from the National Heart, Lung, and Blood Institute awarded to Dr Cousino (K23HL145096).
Competing interests
The authors have no conflicts of interest to disclose.
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