Parenting stress among parents of children with congenital diaphragmatic hernia

The core findings in this study were that parents of children born with CDH who required ECMO support and/or had a long hospital stay showed a high level of parental stress. Children who required ECMO support and had a long hospital stay represented a more severely ill group of children (Table 6), which indicates a correlation between severity of the child’s malformation and the level of parental stress. Additionally, mothers and fathers scored differently, with the mothers scoring higher parental stress than the fathers. We also found an association between parental stress and receiving a prenatal diagnosis of CDH. A parent’s educational level was associated with parental stress in some of the factors.

According to a Swedish nationwide representative sample, a total score of all the five scale scores was calculated: mean 2.52 (SD 0.56). The mean for role of restriction was 3.42 (SD 0.82), while all the other scales had a mean below 3, ranging between 2.05 and 2.61 (SD range between 0.68 and 0.94) [28]. An interesting result is that the study population in general, and all the examined subgroups, reported a total lower parental stress than the Swedish nationwide representative sample, with the exception of parents of children who required ECMO support. Even though becoming a parent to a critically ill child is a severely challenging life event, there are many other regulating stressors that influence parental stress such as social support, single parenting, domestic work load, parity, care-taking issues, the mother’s age and educational level [29, 30]. Becoming a parent to a child with CDH could not be stated as a single isolated down-regulating stressor of parental stress according to the findings in this study. We did, however, find risk factors for parental stress within our study group.

More than half of the children with CDH in this study were detected prenatally, which seemed to influence negatively on parental stress within the Parental Health factor. It is commonly known that CDH is a life-threatening condition that often leads to an intensive, uncertain start in life, and the assumption that foreknowledge of a congenital malformation is beneficial for parents-to-be is questioned. Skari et al. found that a prenatal diagnosis of a congenital malformation is a single independent predictor of acute parental psychological distress after birth when compared with parents who received a postnatal diagnosis [21]. Moreover, children with a prenatal diagnosis of the malformation seem to have a more severe condition compared with infants diagnosed after birth [20]. Furthermore, Kaasen et al. showed that maternal psychological distress shortly after the detection of a fetal malformation is related to the severity of the anomaly, diagnostic and prognostic ambiguity, and gestational age [18]. Severity of the malformation, including ambiguity, has similarly been described to inflect the paternal response [31]. Aite et al. studied couples undergoing prenatal consultations due to a surgical correctable congenital malformation and found no linear correlation between the severity of a malformation and the extent of parental anxiety. However, the number of antenatal consultations could reduce the level of parental anxiety [32]. Subsequently, they studied parents’ emotional and cognitive reactions and stated that antenatal information, both written and visual, should be given several times during an ongoing pregnancy because of the intense emotional distress that parents-to-be experience at diagnosis and their ability to assimilate information affects [33]. In our clinic, parents are offered several consultations during pregnancy with a multidisciplinary team, which includes a pediatric surgeon and specialist nurse, an obstetrician, a midwife and, if needed, a psychologist. What additional information parents are impacted by is, however, impossible to know because of the magnitude of information available today. In a study of parents of children born with a congenital heart defect, parents experienced the amount of reachable information as overwhelming and asked for easily accessible and reliable information sources via the Internet [34]. Fonseca et al. stated that parent’s prior knowledge is important to assess to clarify any potentially incorrect information [35].

Over the last few decades, medical care has rapidly developed for infants born with CDH with increasing survival rates up to 80–90%, and a further late mortality after the first year of life of less than 5% [36]. Long-term sequelae are often related to the severity of the malformation, whereas children who require ECMO treatment are more affected than others [37]. Even in this study, children who required ECMO treatment represented a more severely ill group, compared with those without the need of ECMO treatment. This is shown in the higher rates of prenatal diagnosis, patch repair and the longer length of hospital stay (LOS) (Table 6). Since parental stress was high within this group of parents, there might be an association between parental stress and the severity of the child’s condition. Lewis et al. investigated post-traumatic stress disorder in parents of children supported with ECMO and found a substantial number of parents who were affected by it [38].

Even though there are several prenatal measurements for predicting postnatal outcome, there are no guarantees for the future, and a child’s health depends very much on how he or she is faring after birth. Skari et al. found that mortality and the presence of associated anomalies were consistent with psychological distress at follow-up [21]. We could not confirm any differences in parental stress over time when comparing parents with younger versus older children, which could indicate that it might not change over time. However, it would have been interesting to adjust for factors such as prenatal diagnosis, associated anomalies and ECMO treatment to be able to specify any differences in parental stress over time.

The scoring of the parental stress of fathers in this study is consistent with previous findings, where fathers reported significantly lower scores compared with mothers [26, 31]. Widarsson et al. showed in a study of 320 mothers and 315 fathers of healthy children that mothers with a low educational level, without a role model and with a poor sense of coherence had a higher level of perceived parental stress [39]. Even though fathers report lower total parental stress than mothers, Skreden et al. found that they report significantly more social isolation [26]. According to a study by Fonseca et al. mothers and fathers benefitted from different kinds of social support to reduce parental stress, but there was diffusion between both parents’ adjustments, suggesting that parents affect each other and have great impact in the partner’s level of parental stress [40]. It is well known that mothers who experience a negative childbirth subsequently have fewer children and a longer interval to their second birth [41]. Additionally, a previous history of traumatic birth indicates a significantly higher risk of developing clinically important psychological distress [42].