Introduction
Having a child with congenital heart disease (CHD) puts parents at risk for impaired mental health and reduced quality of life (QoL). When a child is born with CHD, the parents experience shock, disbelief, despair, and grief [
1]. Shortly after birth, many infants with CHD undergo medical and surgical interventions, intensive care unit (ICU) treatment, and overall prolonged hospitalization [
2]. Reasonably, this traumatic experience is associated with psychological strains for the parents. Parents describe that having a child with CHD leads to a burden of uncertainty and is overall a life-changing experience [
5].
Consequently, parents of children with CHD have a high prevalence of posttraumatic stress disorder symptoms (30%), depression and anxiety (25–50%), and severe psychological distress (30–80%) [
6]. Further, a systematic review demonstrated diminished health-related QoL (HRQoL) and general QoL in parents of children with CHD compared to parents of healthy children and children with other illnesses. QoL is a multidimensional construct for the subjective perception of affect and satisfaction with life across physical, social, and psychological domains of daily life [
7]. Factors that contribute to low QoL include stress, anxiety, depression, anger, hopelessness, sense of isolation, inadequate social support sources, financial burden, decreased vitality, impaired physical functioning, and sleeping problems. These factors most likely interact with each other and substantially impact the family’s functioning and everyday life. Risk factors previously identified for low QoL include the severity of CHD, lower child’s age, less social support, and fewer financial resources [
8]. Also, neurodevelopmental impairments, which are common in children with CHD [
9], may add additional burden to their parents’ daily life an may thus impact their QoL.
Most studies investigating well-being (i.e., mental health and HRQoL) in parents of children with CHD have used cross-sectional study designs and investigated well-being during or shortly after hospitalization. Few studies have investigated longitudinal changes in parental well-being. Although these studies have consistently found lower parental well-being during hospitalization and at discharge [
10‐
13], long-term outcomes have been mixed. Two studies showed an improvement of well-being to a normal level six [
10] and 12 months [
11] after hospital discharge. In contrast to these findings, a nationwide population-based cohort study in Norway found lower subjective well-being (measured as cognitive aspect, positive affect and negative affect) and higher symptoms of depression and anxiety, in mothers of children with complex CHD than in mothers of healthy children at 6, 18, and 36 months after the child’s birth [
14,
15]. To date, no study has investigated well-being in parents of children with CHD through the children’s development into adolescence. The complexity of the CHD, the parents’ social situation and potential neurodevelopmental impairments of the children may influence the long-term trajectory of parental well-being.
In this study, we investigated trajectories of psychological dimensions of HRQoL (i.e., mental HRQoL [m-HRQoL]) in mothers of children with CHD from hospital discharge after the first open-heart surgery until the children were 13 years of age. We further aimed to identify classes of maternal m-HRQoL trajectories and risk factors for low m-HRQoL over time.
We hypothesized that we would identify subgroups of trajectories of maternal m-HRQoL including a subgroup of mothers with good m-HRQoL and a subgroup of mothers with persistent poor m-HRQoL. We further wanted to explore whether additional subgroups of mothers experienced worsening or improving m-HRQoL over time. In addition, we hypothesized that having persistent poor m-HRQoL was associated with having a child with a complex CHD (i.e., univentricular CHD, > 1 cardiopulmonary bypass [CPB] surgery), low psychosocial resources (i.e., migration background, low education, sparse social support) and having a child with neurodevelopmental impairments (i.e., low IQ).
Discussion
This is the first study to longitudinally investigate psychological dimensions of HRQoL (m-HRQoL) in mothers of children with CHD over a period of 13 years. We found that m-HRQoL in mothers of children with CHD after their children’s first open-heart surgery was significantly lower than in a matched normative group of mothers. At discharge, 42% of mothers reported clinically relevant impairment of m-HRQoL. Subsequently, maternal m-HRQoL improved to a normal range (at child’s age 1, 10, and 13 years) and even above normal range (child’s age at 4, and 6 years). However, a large variability in m-HRQoL outcomes was observed, and LCGA identified two groups from differing trajectories: a subgroup with normal m-HRQoL (75% of mothers) and a subgroup with low m-HRQoL over time (25% of mothers). Sparse social support was associated with a more than three-fold increased risk of low m-HRQoL in mothers of children with CHD.
M-HRQoL of mothers of children with CHD compared to normative data
We found mothers of children with CHD to be at risk for low m-HRQoL when their child undergoes open-heart surgery. This is in line with previous studies [
10‐
13]. During the period of hospitalization and surgery, parents face several psychological strains, including the fear of losing their children, bonding issues, simultaneous care for other family members, and financial burdens [
3,
4]. Beside low m-HRQoL, previous studies also reported symptoms of posttraumatic stress, anxiety, and depression during the child’s hospitalization and shortly after [
6]. Another study showed that 16% of mothers and 13% of fathers assessed with a self-rated screening questionnaire met diagnostic criteria for a posttraumatic stress disorder after the child’s first open-heart surgery [
28].
In our study, low-maternal m-HRQoL at discharge improved to a normal or even above-normal range at the child's age of 1 to 13 years. However, although an overall improvement in maternal m-HRQoL occurred, m-HRQoL varied substantially over time. Studies with short-term follow-ups have reported mixed findings. Longitudinal studies found that initially low parental HRQoL improved to normal levels 6 months after discharge, and increased anxiety during hospitalization eventually improved to normal levels > 12 months after discharge [
10,
11]. In contrast, other studies have found persistently lower well-being and increased depressive and anxiety symptoms during early childhood [
14,
15,
29]. A cross-sectional study including parents of children with CHD attending preschool showed substantial variation in parental psychological distress, with 27% of mothers reporting high levels of stress and 19% reporting defensive low levels of stress [
30].
Improvement of parental well-being after a traumatic and stressful event, such as having a hospitalized child undergoing open-heart surgery, could result from a range of psychological processes. First, parents could develop and adopt coping strategies to adjust to the cardiac condition of their child and associated stressors. Indeed, Demianczyk et al. used semi-structured interviews to identify frequently used coping strategies in parents of children with CHD. Coping strategies include amongst others (1) self-care to help reduce stress, termed active coping, (2) seeking emotional support among their social network, (3) accepting the situation and trying to find peace, and (4) being spiritual and having faith. In Demianczyk et al.’s study, parents also reported coping strategies that are unique to having a critically ill child, such as self-education, trust in the medical team, connecting with other parents of children with CHD, ensuring presence at the bedside, and spending time away from the bedside for self-care [
31]. Another reason for improved m-HRQoL over time could be that parents may develop “posttraumatic growth”: positive personality changes after a life-changing event [
32]. Indeed, a study recently demonstrated that parents of children with CHD report moderate to strong posttraumatic growth arising from their experience with CHD [
33]. However, associations between posttraumatic growth and improved well-being have not been investigated and therefore warrant further research. Lastly, a “response shift” could be another psychological process explaining improved parental well-being. A response shift indicates that someone has adapted their internal standards and values of well-being after experiencing a traumatic event; the phenomenon has previously been introduced as accompanying improved HRQoL in parents of children with CHD after open heart surgery [
10].
Interestingly, in our study, mean m-HRQoL in mothers of children with CHD was even higher than the Swiss norms when the child was in preschool (i.e., 4 and 6 years of age). One explanation for such a “honeymoon phase” could be that in this phase, most children are in good cardiac condition and the burden of potential neurodevelopmental problems may only manifest later at school age, when cognitive and social demands increase. Therefore, parents might feel relieved to return to a normal life and therefore feel exceptionally well during this period in comparison to the stressful time during their children’s hospitalization and open-heart surgery. Again, adaptive coping strategies, posttraumatic growth, and response shift may foster positive well-being during this phase. However, this hypothesis has not been tested, and future qualitative studies may usefully explore age-related changes in the subjective well-being of parents of children with CHD.
Different trajectories of m-HRQoL over time and risk factors for low m-HRQoL
We found substantial variation in m-HRQoL among mothers of children with CHD. Indeed, LCGA identified two qualitatively distinct subgroups of m-HRQoL trajectories: 75% of mothers belonged to a normal m-HRQoL subgroup, and 25% of mothers belonged to a low m-HRQoL subgroup. In the low m-HRQoL subgroup, m-HRQoL significantly decreased over time with a relatively small effect size. This finding underlines the need to identify those at risk for low and decreasing m-HRQoL to provide early psychosocial support. We found that persistent low m-HRQoL was associated with sparse social support. This is in line with previous studies that identified social support as one of the most consistent and strongest predictors of parental well-being and resilience [
34]. Another study found that a combination of family-related predictors, which include dyadic adjustment, social support, parenting stress, and posttraumatic stress symptoms, explained approximately 75% of the variance in QoL outcome 4 months after discharge [
35]. A systematic review summarized how the family factors of family strain, parental perceptions, and coping were stronger predictors of parental well-being than the illness severity and physical limitations of the child [
8]. In agreement with their finding, we did not observe an association between low m-HRQoL over time and CHD severity measured as number of surgeries and univentricular CHD. Lotto and colleagues suggest that the parental perception of CHD severity differs from the CHD severity defined by the cardiologists. They found poor agreement between parental risk estimation and objective measures of surgical risk [
36]. Indeed, a parent’s subjective appraisal of their child’s health may affect their well-being more strongly than the actual clinical risk. Future studies should consider this difference when evaluating predictors of parental well-being.
25% of mothers of children with CHD reported chronically low m-HRQoL. Further studies are needed to evaluate how these mothers can be supported and investigate the impact of family-based interventions on their long-term well-being. The importance of parental well-being has been underlined by research showing that low maternal mental health is associated with neurodevelopmental impairments in children with CHD [
37‐
39]. Few studies have investigated interventional approaches to supporting mental health in parents of children with CHD. A systematic review demonstrated that parent interventions during the children’s stay on the ICU may improve maternal coping, mother–infant attachment, parenting confidence, satisfaction with clinical care, and infant mental health (see systematic review by [
40]). Another study demonstrated that skin-to-skin care during the stay on the ICU can mitigate the negative effects of parental role alteration by reducing maternal physiological and psychological stress responses. This was observed in reduced salivary cortisol levels and anxiety symptoms [
41]. Two randomized controlled trials showed that educational intervention programs after hospitalization improved maternal QoL and anxiety symptoms [
42,
43]. These studies demonstrate promising interventional approaches to promoting parental mental health during and after hospitalization. Nevertheless, health policies that prioritize parental mental health care for parents with critically ill children remain lacking [
40]. Further, reasons why parents do not proactively seek mental health support can be manifold and include (a) the belief that psychological support is outside the care team’s scope of practice, (b) the inclination to “stay strong” and fear negative judgment, and (c) the desire to focus care resources on the child [
44]. Thus, substantial effort and standardized screening procedures may be required by the clinical care team to facilitate mental health support for parents of children with CHD.
Our study has shown that many mothers report critically poor m-HRQoL at the time of hospitalization. Although some of these mothers recover and report normal m-HRQoL throughout their child’s development, others continue to have low m-HRQoL. We have shown that CHD complexity may not adequately predict the trajectory of maternal m-HRQoL, but perceived social support can. Screening for psychosocial resources during hospitalization may be suitable for early identification of mothers at risk for persistent low m-HRQoL. Future studies are needed to investigate screening tools for identifying mothers at risk to target psychological interventions on those who need them most.
Limitations
This longitudinal study used m-HRQoL data on mothers of children with CHD, compared their m-HRQoL to matched normative data, and investigated the prediction of two m-HRQoL trajectories. However, some limitations should be taken into account when interpreting our results. First, mothers included in this analysis had significantly higher education than mothers who did not complete any m-HRQoL questionnaires. Therefore, our results may not be fully generalizable to the whole population of mothers of children with CHD. Second, we used m-HRQoL to measure maternal psychological adjustment over time. Thus, our results cannot provide any information about specific mental health problems, such as depression, anxiety, and posttraumatic stress. Future longitudinal studies are needed to investigate specific psychological disorders and thus select specific treatments and support. Further, we have not collected data on maternal m-HRQoL during pregnancy, but approximately 40% of CHD diagnoses are made prenatally [
45]. Parents with a prenatal CHD diagnosis may be more deeply affected than parents who receive a postnatal CHD diagnosis [
46]. The long-term effects of impaired mental health during pregnancy warrant further investigation and are important for defining the optimal timing of mental health interventions for parents of children with CHD.
Another limitation is that the normative data used for comparison with mothers of children with CHD was cross-sectional. Thus, we were unable to compare longitudinal trajectories between mothers of children with CHD and mothers of healthy children. Nevertheless, we were able to use a matched normative sample, and we corrected for age to control for normal age-related changes in m-HRQoL.
Over the course of 13 years, missing m-HRQoL data was relatively high. 58% of mothers filled in 4 or more questionnaires. Missing data may introduce bias. To maintain power and account for relations in the data, missing data was estimated with imputation by chained equation [
25].
Lastly, we only investigated mothers of children with CHD and did not include fathers in this analysis because we lacked sufficient responders. Previous cross-sectional studies have found that fathers of children with CHD also report low m-HRQoL and symptoms of posttraumatic stress, depression, and anxiety [
6,
8]. Nevertheless, fathers are substantially underrepresented in studies investigating parental well-being [
8]. In addition, we did not assess siblings and we did not assess if siblings had any chronic conditions. Future studies should also include siblings of children with CHD, because their well-being may also be substantially affected and they could further influence parental outcome.
Conclusion
In mothers of children with CHD, trajectories of m-HRQoL from hospital discharge throughout the child’s development are heterogenous. A subgroup of mothers with continuously low m-HRQoL was identified. Overall, mothers of children with CHD are at risk for low m-HRQoL in the initial period when their children undergo open-heart surgery. Importantly, a significant subgroup of mothers continues to have low m-HRQoL throughout their children’s development into adolescence. Sparse social support was associated with an increased risk of low m-HRQoL over time, whereas CHD severity was not predictive of m-HRQoL. Studies of family-oriented approaches targeting families with low psychosocial support are needed to reinforce parental well-being in the long term. Future longitudinal studies investigating parental well-being should also consider fathers and siblings, because they are often underrepresented in mental health studies and may have different HRQoL trajectories from mothers with, consequently, differing needs.
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