Traumatic stress in parents of children with congenital heart disease: a scoping review

Abstract

Congenital heart disease is the most common birth defect in the United States, with many of the affected infants requiring surgical and/or interventional procedures within their first year of life. The parental impacts of a child’s diagnosis, subsequent hospitalization, and transition to home after discharge are numerous and burdensome, and many experience symptoms of traumatic stress along this trajectory. The purpose of this scoping review was to summarize current available literature related to the traumatic stress experienced by parents of children with heart disease to better understand the prevalence, related factors, and consequences. The Joanna Briggs Institute Scoping Review Framework was implemented to identify 31 relevant peer-reviewed articles published between 2000 and early 2024, including 25 quantitative studies, 3 qualitative studies, and 3 systematic reviews or meta-analyses. This scoping review provides an overview of parent traumatic stress for clinicians caring for children with heart disease at every stage of their clinical course.

Congenital heart disease (CHD) is the most common birth defect in the United States, affecting approximately 40,000 (1%) live births each year, and 25% of these infants are born with critical heart disease requiring surgical and/or interventional procedures during their first year of life. ¹ While advances in medical and surgical care have dramatically improved survival rates for infants and children with critical CHD, the psychological impact on parents managing long hospitalizations, uncertain outcomes, multiple procedures and appointments, and high medication burdens is concerning. Parents of children with CHD are at risk for mental health disorders, including depression, anxiety, and traumatic stress. ²

Traumatic stress is the most prevalent form of psychological distress experienced by parents of children with heart disease. ^2–4 Symptoms of traumatic stress have been reported at the time of diagnosis, during the hospitalization, and after discharge. ^2,3 Thus, it is important for clinicians who interact with parents during all phases of care to understand the prevalence of and factors associated with traumatic stress. Early recognition of traumatic stress is essential to trigger additional support. This scoping review summarizes the current literature related to prevalence, related factors, and consequences of traumatic stress in parents of children with CHD. Gaps in knowledge and opportunities for future research are discussed.

Definition of traumatic stress

Trauma can be defined as “sets of circumstances that represent significant challenges to the adaptive resources of the individual and that represent significant challenges to the individuals’ way of understanding the world and their place in it.” ⁵ Traumatic medical events involving a child may trigger post-traumatic stress symptoms (PTSS) in parents, including re-experiencing, avoidance, and hyperarousal. Re-experiencing or thinking about the traumatic event can help with processing; however, flashbacks or nightmares may be intrusive or distressing and feel like one is reliving the trauma. Avoidance may be characterized as trying to stop thoughts or conversations about the trauma and may manifest new worries and fears. Additionally, while a new sense of caution may be appropriate after a traumatic event, these new fears and anxieties can be problematic if they interfere with daily life. Hyperarousal refers to an altered stress response, such that the response to a stimulus may be exaggerated and lead to physical symptoms of stress. ^6–7 Symptoms of traumatic stress may meet diagnostic criteria for acute stress disorder (ASD) or post-traumatic stress disorder (PTSD). ⁸ ASD describes traumatic stress symptoms lasting up to 1 month after the traumatic event, whereas PTSD describes symptoms that last more than 1 month after the traumatic event. ⁹

Materials and methods

Framework

The Joanna Briggs Institute Scoping Review Framework was selected because of its detailed guidance, inclusiveness of qualitative research, and alignment with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses—Extension for Scoping Reviews (PRISMA-ScR). ¹⁰ Accordingly, the following steps were taken: (1) identifying the research question, (2) identifying relevant studies, (3) study selection, (4) charting the data, and (5) collating, summarizing, and reporting results.

Identifying the research question

The research team and a clinical librarian developed a protocol and research purpose to explore, “what is known about the prevalence, related factors, and consequences of traumatic stress in parents of children with heart disease?”

Identifying relevant studies

PubMed, PsycInfo, and Scopus databases were searched for relevant publications between 2000 and early 2024. Search terms used in PubMed were parent (mesh) and heart defects, congenital (mesh) and traumatic stress (tiab) or stress disorders, traumatic, and acute (mesh). PsychInfo search terms were parents or caregivers or mother or father or parent and CHD or cardiac defect or heart defect or CHD and traumatic stress or PTSD or ASD. Scopus search terms were congenital and heart and disease and parent and traumatic and stress or acute and stress and disorder.

This review included clinical trials, qualitative studies, and systematics reviews with samples that included parents of children with heart disease and measures or descriptions of traumatic stress. Exclusion criteria included studies of parents of children with exclusively non-cardiac illnesses or injuries, children with heart disease, adult patients with heart disease, and those that did not specifically examine traumatic stress. Bibliographies were also reviewed to find other relevant references. All publications were managed in Covidence.

Study selection and data abstraction

This search yielded 1492 studies. After 218 duplicates were removed, the title and abstract were screened on 1274 articles. Of the 1274 studies screened, 54 were included in the full-text review, and 11 were excluded for the wrong outcomes and 12 for the wrong patient population. Thirty-one articles were included in the final review. See Figure 1 for the PRISMA diagram of this process. One reviewer conducted the study selection and abstraction.

Figure 1. Flowchart of PRISMA diagram depicting the literature review and inclusion process.

Results

The articles (n = 31) were published between 2000 and early 2024, with 14 published since 2020. Twenty-five were clinical trials, three were qualitative studies, and three were review studies (see Table 1–3). Of the three review papers included, one published by Woolf-King and colleagues in 2017 focused specifically and exclusively on the mental health outcomes of parents of children with heart disease. ² This review cited five studies that focused specifically on traumatic stress and those were also included in this review. Two other review papers were included because their samples included parents of children with heart disease, one focused on parents of children with chronic physical illness and the other on parents of children following paediatric surgery. ^11,12

Table 1. Key findings of included quantitative studies (n = 25)

ASD = acute stress disorder; CHD = congenital heart disease; ED = emergency department; ICU = intensive care unit; PCICU = paediatric cardiac intensive care unit; PICU = paediatric intensive care unit; PTSD = post-traumatic stress disorder; PTSS = post-traumatic stress symptoms; QOL = quality of life; SV = single ventricle.

Table 2. Key findings of included qualitative studies (n = 3)

ASD = acute stress disorder; CHD = congenital heart disease; ICU = intensive care unit; PICU = paediatric intensive care unit; PTSD = post-traumatic stress disorder.

Table 3. Key findings of included review studies (N = 3)

ASD = acute stress disorder; CHD = congenital heart disease; ICU = intensive care unit; PTSD = post-traumatic stress disorder; PTSS = post-traumatic stress symptoms.

Prevalence

Traumatic stress symptoms have been identified in 25–88% of parents at the time of diagnosis of CHD. ^13,14 Rychik and colleagues found that 39% of pregnant mothers had PTSS after fetal diagnosis of CHD. Another study found that 85% of parents had PTSS 4 weeks after the diagnosis and 27.3% continued to have PTSS 4 months after diagnosis. ⁴ In a mixed methods study of 29 parents, 88% of mothers and 66% of fathers developed PTSD after prenatal diagnosis of hypoplastic left heart syndrome (HLHS), and 100% of mothers and 75% of fathers experience PTSD after postnatal diagnosis of HLHS. ¹⁵

After their child’s cardiac surgery, 83% of parents endorsed at least one symptom of traumatic stress, with mothers reporting higher levels of traumatic stress symptoms than fathers. ³ After the Norwood operation, 50% of mothers and 39% of fathers experienced PTSD. ¹⁶ In other reports, ASD was reported in 25% of parents 3–4 weeks after their child’s cardiac surgery. ^17,18

Two studies found that 25% of parents met the criteria for PTSD at the time of hospital discharge. ^18,19 Other studies report 16–17% of mothers and 6–13% of fathers meet the criteria for PTSD at the time of hospital discharge. ^20–22 In one report, all parents of infants with single ventricle anatomy between Stage 1 and Stage 2 operations were noted to have PTSS at the time of discharge. ¹⁹ One month after discharge, 33% of mothers and 18% of fathers had ASD. ³ Three months after surgery, 19% of parents reported PTSS. ²³ Bainton and colleagues found that the prevalence of PTSD in parents of infants with single ventricle heart disease decreases in mothers (50–27%) and fathers (39–24%) between the Norwood operation and 16 months of age. PTSD lasting greater than 4 months was found in 9–25% of all parents of children with heart disease (see Table 1). ^24–27

Stressors and symptoms

Lisanti et al. found that 3 months after discharge, postnatal diagnosis and parental role alteration predicted PTSS, as well as anxiety and depressive symptoms. ²³ In their qualitative descriptive study, Harvey et al. described six themes extrapolated from mother’s journal entries related to stress, including feeling intense fluctuating emotions, navigating the medical world, dealing with the unknown, facing the possibility of a child dying, finding meaning and spiritual connection, and mothering through it all. ²⁸ These themes overlap with some of the themes described by Kosta et al. in their qualitative study exploring parental experiences after their infant’s cardiac surgery. These themes include uncertainty, structural and systemic issues related to the microenvironment within the unit and macro-environment within the hospital, relationships with staff, social networks, communication of important information, and individual coping strategies. ²⁹ Consistent with Harvey et al., Cantwell-Bartl et al. found that the main stressor for parents was fear of their child dying. ¹⁵ Other stressors described by Cantwell-Bartl and colleagues were the appearance of their child, ICU environment, hearing about their child’s poor prognosis and/or complications, and witnessing another child’s death (see Table 3).

Few studies reported on the prevalence of specific symptoms of traumatic stress; however, Bainton et al. found that intrusive thoughts and hyperarousal symptoms were the most commonly reported symptoms among parents. ¹⁶ Another study found that receiving a prenatal diagnosis compared to a postnatal diagnosis increased the likelihood of avoidance symptoms of mothers and fathers and hyperarousal symptoms in fathers. ³

Demographic factors associated with traumatic stress

Both mothers and fathers experience high levels of stress related to their child’s diagnosis, surgery, and recovery; however, more mothers experience PTSS than fathers. ^{2,16,20–22,15,30,31} (see Table 1) Mothers also experienced higher levels of PTSS. ^3,30,31 Golfenshtein and colleagues reported that at the time of hospital discharge, the number of PTSS was associated with parent education and insurance, and 4 months later, the severity of PTSS and PTSD was predicted by ethnicity and number of children in the household, respectively. ⁶ Mothers of very young children were also found to have higher levels of stress (see Table 1). ²⁰

Psychosocial factors associated with traumatic stress

Muscara et al. reported that psychosocial factors explained 36% of the variance in parents with ASD, compared to demographic factors that explained only 4.5% of the variance. ³² Currie found that higher psychosocial risks, mediated by the stress response, were predictive of PTSS 4 months after diagnosis. ¹⁴ After fetal diagnosis, those who experienced denial or were unresolved to the diagnosis had higher rates of PTSD. ^13,14

Stokes et al. reported that parents' acute stress responses to unexpected complications during the hospitalization, including prolonged mechanical ventilation, were significantly associated with post-traumatic stress scores 2 years after the hospitalization. ³³ Acute stress reactions explained 44% of the variance of parents' long-term PTSS. ³³ Unexpected complications causing an acute stress response in parents had a greater impact on long-term mental health outcomes, including PTSD, than the patients’ diagnosis, perioperative factors, or demographics. ³³ Several studies found that high levels of anxiety, reduced coping, perceived lack of social support, and low parenting satisfaction were associated with PTSD (see Table 1). ^{11,12,16,17,22} Helfricht et al. report that traumatic stress severity at the time of discharge was associated with PTSD after 6 months after discharge. ¹⁷ Additionally, parenting stress at discharge was associated with both the number and severity of PTSS. ⁶ Four months after discharge, parenting stress predicted PTSD. ⁶

Clinical factors associated with traumatic stress

Wray and colleagues examined over 2000 parents and described 2 pathways of psychological well-being of parents defined by the complexity of heart disease, specifically that parents of children with mild heart disease report fewer PTSS than parents of children with cardiomyopathy or single ventricle heart disease. ³¹ This finding was consistent with another study of parents 4 months after discharge that found that having a child with single ventricle physiology was predictive of PTSD. ⁶ Complexity of surgery was associated with more acute stress symptoms in two studies ^11,22 and another found that specifically, fathers of children with high clinical acuity had higher levels of acute stress. ²⁰ However, other studies found little to no relationship between clinical variables and PTSS or PTSD (Table 1). ^21,33,34 The number of infant medications at the time of discharge was associated with the severity of PTSS, and the need for tube-assisted feedings at discharge predicted PTSD. ⁶ Infant growth trajectory over the first 4 months was associated with PTSS and parenting stress. ³⁵ Four months after discharge, the number of emergency visits was associated with the severity of PTSS. ⁶ In a recent study examining parent biomarkers of stress, Lisanti et al. found that parents' salivary cortisol area under the curve with respect to the ground measured at discharge predicted PTSS 3 months later. ³⁶

Consequences of parental post-traumatic stress

Parents with full or partial PTSD were more likely to experience lower mental health-related quality of life. ^18,37 In a multivariable model, Lisanti et al. found that the combination of the relationship quality between partners, social support, parenting stress, and PTSS explained 74% of the variance in quality-of-life scores. ³⁷ Emergency room visits and unplanned visits to the cardiologist and primary care provider were associated with increased symptoms of PTSD (see Table 1). ³

Parent PTSS were also associated with overprotective parenting and subsequent emotional and behavioural problems among their children. ²⁴

Discussion

A stress response to a traumatic experience can be adaptive; however, persistent or intense symptoms may interfere with daily life and impact overall well-being and quality of life of parents of children with heart disease. The purpose of this scoping review was to report on what is known about traumatic stress in parents of children with heart disease. Studies sought to describe the prevalence, timing, associated factors, and consequences of traumatic stress. Periods of heightened traumatic stress for parents were identified at the time of diagnosis, during the hospitalization, and at the time of discharge. While the majority of parents report reduced PTSS over time, 16–25% of parents experience persistent PTSS lasting longer than 16 months. ^16,27,32 These findings reflect slightly higher rates than those recently reported by Whyte-Nesfield et al., who found that 12.5% of parents of children with unexpected PICU admissions experience PTSS 18–30 months post-discharge. ³⁹ In a group of 159 parents of children from the PICU, oncology, or cardiology divisions, Muscara identified three distinct profiles of parents over an 18-month period and termed them resilient, recovery and chronic. Thirty-three percent of parents were described as resilient and experienced low distress through all time points. The recovery group accounted for 52% of parents who had high distress at the diagnosis that decreased with time over several months. The chronic group accounted for 13% of parents, and they experienced high levels of distress at all time points through 18 months. ³²

Recognizing particularly vulnerable time points and factors associated with parent traumatic stress may prompt timely screening to facilitate risk stratification and tailored individualized interventions that optimize outcomes for parents and children with heart disease. Given the consequences of poor adjustment to the diagnosis, additional screening and psychological support may be warranted when communicating the diagnosis to parents. In their review paper, Tacy and colleagues illuminate the complex nature of counselling parents through a prenatal diagnosis of CHD, from the perspective of the provider and parents. ⁴⁰ During these conversations, parents are processing new medical information, managing uncertainty, and experiencing psychological distress. These early conversations significantly influence the adjustment and psychological well-being of parents and families; however, training for providers and best practice have not been established. Empathetic and trauma-informed communication may support psychological adjustment to the diagnosis and a collaborative and trusting partnership between parents and providers. ⁴⁰

The prevalence of PTSS during hospitalization and the sustained nature of these symptoms throughout admission warrant attention. ^3,30 Given the impact of the acute stress response on PTSD, the inpatient admission is an opportunity to identify parents at risk for ASD and PTSD and provide additional support to cope with the stress of the hospitalization. Psychosocial risks, including baseline anxiety, depression, poor coping, parental role alteration, parenting stress, lower education, and acute stress response, were commonly reported. While the complexity of the disease was not consistently reported as a risk factor, parents of children with single ventricle heart disease, tube-assisted feedings, and a high medication burden were at higher risk of developing PTSS. ^6,20,19,31 Early recognition of risk can be achieved by building standardized screening and subsequent support programmes (see Table 1). ^{4,6,13,14,16,21–23,33,34,17}

Specific triggers of stress are also important to guide interventions. Infants and children in the ICU are fragile, require continuous monitoring, and are often connected to life-saving equipment. While necessary, this environment poses many barriers to healthy bonding and parenting and can trigger both traumatic and parenting stress. Consistent with qualitative findings, ^12,15,28 the Paediatric Cardiac Intensive Care Unit Parental Stress Model asserts that parental stress emerges from three specific categories of stressors: infant, parent, and environment. ⁴¹ Exposure to these common stressors in the ICU setting combined with the significant challenges to one’s adaptation resources and the existential threats to their child’s well-being are a likely precursor to traumatic stress. ^15,28,29 This framework may be useful to employ in the development of parent support programmes aimed at addressing acute stress in parents during their child’s hospitalization.

Finally, given the subset of parents that experience persistent PTSS, and even meet the diagnostic criteria for PTSD, screening and ongoing support are needed after hospital discharge. Screening for PTSS and recognition of psychosocial risk factors and specific sources of stress may help inform a tailored approach to providing necessary support. Addressing these specific needs of parents experiencing PTSS may reduce unplanned utilization of healthcare and support healthy parenting and child development. ^24,38

Few psychological support interventions for parents of children with heart disease have been developed and tested, and only one measured traumatic stress. ⁴² Medoff-Cooper and colleagues tested the effect of a telehealth home monitoring programme for parent-infant dyads discharged after neonatal cardiac surgery. Four months post-discharge, there was no significant difference in PTSD scores between the intervention and control group who received usual care. ²⁵

Other intervention studies focused on anxiety, depression, parenting stress, coping, worry, and quality of life. For example, Callahan et al. described the effect of early palliative care on parenting stress, anxiety, and depression. The intervention focused on four specific domains including bonding, feeding, memories and emotional support, and psychological and spiritual support. ⁴³ Overall stress in the intervention group was decreased; however, there was no difference between anxiety and depression scores between the groups. ⁴³ McCusker and colleagues developed a psychoeducational intervention that supported the mother-infant relationship, parenting skills, and emotional processing of their experience. Six months after the intervention, mothers in the intervention group demonstrated improvement in anxiety, worry, and appraisal of their situation. ⁴⁴

In the neonatal intensive care unit (NICU), Ghaedi-Heidari and colleagues found that the mindfulness-based stress reduction intervention was associated with higher post-traumatic growth scores. ⁴⁵ The application of trauma-informed care and Adverse Child Experiences framework are emerging in the NICU literature; however, very few results are available related to the implementation of these strategies. ^46–48

A significant gap exists with regard to interventions that support parents of children with heart disease through acute stress responses and traumatic stress symptoms. Additional research is needed to develop programmes to support parents through trauma and address the other known psychological consequences of parenting a child with heart disease, including parenting stress, anxiety, and depression. ² Given the impact of psychosocial factors on psychological outcomes, interventions tested in particularly vulnerable populations, for example, those with low levels of education, low resources, poor social support, and non-English speakers, are needed. ⁴⁹

Limitations

There are limitations of this scoping review to be acknowledged. Given that this was a scoping review and not a systematic review, the quality of the studies was not critically appraised, including the potential for bias. Various methodological approaches to studies were included, and different measures of traumatic stress were used across studies making comparisons difficult.

Future research

While research about traumatic stress in parents of children with CHD is limited, the body of evidence has grown substantially over the past 5 years, and findings are concerning for both parents and their children. Rates of traumatic stress among parents of children with heart disease are considerably higher than national rates ^{3,4,6,13,14,16,18,20,21,19,23,24,27,38,17,50} and contribute to negative outcomes for both parents and children. ^{6,18,21,24,35,37,17} Developing an understanding of the full psychological profile, including the factors related to post-traumatic growth in parents of children with heart disease, may help inform interventions that support trajectories towards healthy coping and growth. ^24,49,51 Testing interventions that are informed by screening results and knowledge of the trajectory and triggers of traumatic stress will be important next steps. ⁴² Interventions should aim to improve parent self-efficacy and support healthy coping when faced with acute stressors. ⁴⁹ Given the high rates of traumatic stress symptoms and the known triggers of stress during the hospitalization, this period of time may be an opportunity to modify the subjective experience and reduce the risk of developing traumatic stress symptoms. ⁴² While impossible to alleviate the lifelong impact of CHD on parents and children, targeted interventions may create a foundation that fosters healthy adaptation to traumatic medical experiences.