The psychosocial situation of family caregivers of children with RDs is severely strained, even more so in times of the COVID-19 pandemic. The current study found high distress levels among family caregivers, their manifold psychosocial information needs, and impaired caregiver-reported HRQoL in children with RDs.
Distress levels were remarkably high among the current sample of family caregivers of children with RDs. Clinical distress was reported by almost 90% of the caregivers. Notably, the mean distress score of the caregivers was well above the cut-off for clinical distress set by the authors of the original DT-P, highlighting the clinical relevance in this sample. Furthermore, caregivers experienced numerous problems in the domains of practical, physical, social/family, cognitive, emotional, and parenting issues, providing insights regarding the sources of distress. While all problem domain scores significantly correlated with overall distress, cognitive and practical problems showed the weakest and emotional problems the highest correlation, revealing possible targets for interventions. The majority of participants suffered from sleep problems, fatigue, being out of shape, weight problems, fears, feeling tense or nervous, worry, as well as problems regarding work/study, childcare/child supervision and the independence of their child, problems regarding leisure activities/relaxing, and keeping emotions under control.
The current findings are consistent with pre-pandemic studies demonstrating the immense burden and compromised psychosocial health of caregivers of children with chronic illnesses [46], and particularly in caregivers of children with RDs [12, 47, 48]. Given the negative impact of the COVID-19 pandemic on the mental health and well-being of children and their families in the general population [21, 22], it was reasonable to assume that the pandemic would particularly affect caregivers of children with RDs who were already highly strained before the pandemic. The assumption is further supported by findings of a significantly lower quality of life and significantly higher impairment in mental health in parents of children with a congenital RD compared to healthy controls and norm data in times of the early COVID-19 pandemic [28].
Family caregivers of children with RDs reported wide-ranging psychosocial information needs regarding the navigation through the health care system (e.g., financial aids related to the disease of the child), psychosocial strains in the family (e.g., coping with emotional strain), further support offerings (e.g., support options for everyday life), and the strengthening of oneself and the family system (e.g., strengthening of siblings). These findings suggest that the current provision of information by professionals or existing websites is perceived as inadequate by families and underscore the importance of additional support services. The current psychosocial information needs are consistent with previous studies that highlighted the need for more information about psychosocial support options and sociolegal counseling among family caregivers of children with RDs before the COVID-19 pandemic [6,7,8, 16,17,18, 49]. Since many of the pre-pandemic psychosocial counseling and care services were not available during the COVID-19 restrictions in Germany or were not used due to risk of infection, it can be assumed that psychosocial information needs increased even further during the course of the pandemic.
Most participants (136 of 140 participants, 97.1%) reported no helpful web pages devoted to psychosocial burdens, which is also consistent with the underrepresentation of psychosocial topics on specific RD information websites [18]. Other studies report that parents of children with RDs regularly use the internet as a source of information [17, 50] and that internet-sourced information was found to have a significant empowering effect on the parents [50]. These findings, along with the high demand for psychosocial information revealed by the current results, suggest that more informational websites on psychosocial matters are urgently needed. However, other possible explanations for this extremely high proportion should also be considered. It is possible that participants had difficulties assigning which topics belonged to “psychosocial burdens” and were therefore unable to name any specific websites. Moreover, caregivers may primarily search for specific topics via search engines and may not recall individual pages in their search history. Another possible explanation could be that caregivers tend to search more for practical information such as health and long-term care insurance benefits while neglecting their own psychosocial needs as they do not consider the internet to be useful for self-care-related purposes. Qualitative studies using interviews are warranted to investigate the role of online information in the care of families of children with RDs.
Almost half of the children with RDs over the age of four had an impaired HRQoL, as reported by their caregivers. The caregiver-reported HRQoL of the children with RDs was comparable to the parent-reported HRQoL of children with severe chronic diseases but substantially lower than that of children with mild to moderate chronic diseases [43]. However, it should be noted that the reference sample was not matched by age or gender, and no reference data for children under the age of 8 years exist for the measure. Because the age of the children in the current sample was not associated with caregiver-reported HRQoL scores, and norm differentiation is only recommended for background factors associated with the outcome variable, this can be considered appropriate [51]. Previous research indicates that the HRQoL of children with RDs was already impaired before the pandemic [52, 53] and that the quality of life of children generally decreased during the pandemic [21]. Therefore, it seems likely that the present finding is a combination of pre-pandemic impairment in HRQoL and additional impairment due to the pandemic. Note that the current data were collected at the onset of the pandemic and may not reflect its full impact on the HRQoL of children with RDs.
The distress level of the caregivers was positively associated with their overall psychosocial information need and negatively associated with the caregiver-reported HRQoL of the children with RDs, whereas psychosocial information needs and caregiver-reported HRQoL of the children were not found to be correlated. To our knowledge, no previous study has examined a direct relationship between distress levels and psychosocial information needs in family caregivers of children with RDs–although an existing relationship provides an interesting starting point for psychosocial support services in the form of information offers. The HRQoL of children with RDs appears to play an important role in caregiver burden, which is in line with previous research that has identified emotional and behavioral problems in children as important determinants of caregiver burden [12]. This also broadly fits with previous findings that the physical, emotional, social, and school functioning of children with RDs were associated with parental life satisfaction [52]. Altogether, the results presented also correspond well with previous research and theoretical frameworks on related constructs–for example, caregiver needs and models of stress, quality of life, or coping with stress [1, 9, 19, 54,55,56,57,58].
Research in times of the COVID-19 pandemic was primarily concerned with families of healthy children [21, 59], or children with disabilities in general [25], whereas little consideration was given so far to the psychosocial situation and needs of families with children with RDs [28]. However, the uncertainty about the impact of COVID-19 was even more threatening to people with chronic or pre-existing diseases and especially RDs [26, 27]. Furthermore, the German COVID-19 restrictions during the study period concerned particularly sensitive areas of life, including the closure of school and daycare, the cancellation of leisure activities, contact restrictions, restrictive visitation policies in medical facilities, and limited access to social support, which particularly affected individuals who needed special support and therapies. These pandemic-specific stressors, which added to pre-pandemic stressors, should be taken into account when interpreting the situation of the families.
The current results should be interpreted in light of the exploratory nature of the study and should be understood as a general inventory of the psychosocial situation of the families during the early COVID-19 pandemic. Even though the present research delivers interesting starting points for further support offerings, it is limited to providing a snapshot of the situation of family caregivers of children with RDs during the first year of the COVID-19 pandemic. Furthermore, the cross-sectional study design does not allow making inferences about causality, as the temporal link between the outcome and the exposure cannot be determined. More research with experimental or at least longitudinal study designs and setting up structural equation models or directed acyclic graphs [60] are needed to further investigate the nature of the discovered links. Moreover, embedding the presented results into theoretical frameworks on stress and needs in caregivers of children with disabilities (e.g., Perry’s stress framework for parents of children with disabilities [58]) would be desirable.
Analogous to the depression-distortion hypothesis [61, 62], the distress level of the caregivers may have biased their assessment of the HRQoL of their children [30, 31], which may have distorted the magnitude of the true association. However, cognitive biases are typical for people with confirmed depression, and a distinction should be made here from the present sample, in which no confirmed diagnoses but only elevated distress levels were found. Furthermore, not the psychopathology of the children, but their HRQoL was rated by the caregivers, and the “proxy problem” in HRQoL ratings may be smaller than previously assumed [63].
Some limitations regarding the representativeness of the sample restrict the generalizability of the results. First, the sample size is rather small considering the large population size in Germany. Nonetheless, smaller samples are relatively common in the field of RDs, and the current sample size is comparatively decent for a sample of family caregivers of children with RDs. Furthermore, it is plausible that mainly people who are involved in self-help or are otherwise proactive were reached. Notably, participants in the current study mostly included individuals with a high level of education. In addition, fathers caring for a child with RD were heavily underrepresented in the current sample, whose experiences and needs might differ from those of mothers–a problem that is quite prevalent in similar studies [64]. Future studies should include a more diverse field of participants–including children with RDs if possible–by acquiring through primary care providers or centers for RDs. In addition, the current results may not be generalizable to other countries with different health care or support systems. However, the consistency of the current results with those of previous studies from different countries suggests that the findings may be broadly applicable to other countries.
Practical implications
The current study should raise awareness of the unmet psychosocial care needs of family caregivers of children with RDs and their heavily strained situation among practitioners and policymakers, particularly during the COVID-19 pandemic. Collectively, the presented results suggest that there is an acute need for action to ensure sufficient psychosocial care for family caregivers of children with RDs. Importantly, the COVID-19 pandemic should not compromise the psychosocial care for the families. More pre- and intervention options for the families are needed and psychosocial supports should be offered to all families in the diagnosis of an RD of their child, while health care professionals should be aware of the high prevalence of clinical distress among caregivers. Comprehensive care programs should be complemented by low-threshold offerings that can be integrated into the daily lives of those affected. As an example, the CARE-FAM-NET project, which is currently being evaluated, may take an important step towards improving access to psychosocial services in Germany and serve as a model for other countries [35].
Our findings underscore the importance of psychosocial information for caregivers. Psychosocial information and support options should become part of the standard medical care that is automatically provided when a child is diagnosed or suspected of having an RD. The existing guideline in Germany on psychosocial care in pediatric oncology [65] could serve as a basis for the development of a similar guideline for RDs. In addition, the wide reach of (umbrella) patient organizations for RDs (e.g., ACHSE for Germany, NORD for the USA) should be leveraged to disseminate existing information services among families with children with RDs. A feasible avenue is to provide a modular website–such as the CARE-FAM-NET website [66], which we built for families with children with RDs using the current findings on psychosocial information needs. Any such website can cover different topics that are of relevance for the families and can be accessed individually depending on acute need. Furthermore, existing websites that provide medical information to families with children with RDs should be used to also provide psychosocial information, for example, by linking to quality-checked websites with psychosocial information. Finally, psychosocial care for family caregivers of children with RDs should be given greater focus within existing international collaboration projects, as multinational programs can have a great impact and significantly advance the dissemination of scientific knowledge. The existing European Reference Networks for RDs (ERNs) could, for example, develop a guide for health care professionals on the psychosocial needs of families affected by RDs alongside better access to psychosocial care to promote resilience.