The psychosocial situation of families caring for children with rare diseases during the COVID-19 pandemic: results of a cross-sectional online survey.

BACKGROUND: The COVID-19 pandemic is affecting many areas of life and has posed additional strains on the highly vulnerable group of caregivers of children with rare diseases (RDs). The psychosocial situation of the family caregivers deserves more attention, both in research and practice. The current study explores the distress level of caregivers of children with RDs, their psychosocial information needs, and caregiver-reported health-related quality of life (HRQoL) of children with RDs in times of the COVID-19 pandemic. METHODS: Data from a cross-sectional online survey conducted within the German CARE-FAM-NET project (children affected by rare diseases and their families-network) between March and August 2020 were examined. The study sample included 149 family caregivers, mostly mothers (83.2%) of 167 children with RDs. The survey assessed demographic and disease-related characteristics, distress and everyday problems of caregivers (Distress Thermometer for Parents; scale 0-10), psychosocial information needs (self-developed items; scale 0-100), and caregiver-reported HRQoL of the children with RDs (DISABKIDS Chronic Generic Measure, short-form; scale 0-100). Using descriptive statistics, we analyzed the psychosocial situation of families during the COVID-19 pandemic. We further conducted correlation analysis to investigate interrelations. RESULTS: The distress level among caregivers was high (M = 6.84, SD = 2.43); 89.6% reported clinical distress (≥ 4). Everyday problems (e.g., sleep problems, fatigue, being out of shape, fears, feeling tense or nervous, and worry) were frequent. Caregivers reported a wide range of psychosocial information needs. In about half of the children (49.5%), caregiver-reported HRQoL was low, while average HRQoL (M = 58.7, SD = 19.5) was comparable to parent-reported norm data of children with severe clinical conditions. Distress correlated positively with psychosocial information needs (r = 0.40), and negatively with the caregiver-reported HRQoL of the children (r = - 0.46). CONCLUSIONS: This study indicates a high psychosocial burden on family caregivers of children with RDs during the early COVID-19 pandemic, characterized by high distress levels and wide-ranging everyday problems, unmet psychosocial information needs, and reduced caregiver-reported HRQoL in children with RDs. The findings highlight the ongoing need for target group-specific, low-threshold support services (e.g., websites) during and after the pandemic.

Mental Health of Siblings of Children with Rare Congenital Surgical Diseases during the COVID-19 Pandemic.

INTRODUCTION: The COVID-19 pandemic has affected every aspect of our society, particularly vulnerable groups, such as families with children suffering from rare diseases. However, the psychosocial influences of COVID-19 on the healthy siblings of children with rare diseases have not been investigated yet. Thus, the study aimed to evaluate the mental health of healthy siblings of children with rare congenital surgical diseases during the COVID-19 pandemic. MATERIAL AND METHODS: Siblings of children with rare congenital surgical diseases were investigated cross-sectionally between April 2020 and April 2021. Data on mental health were collected using the parent-version of the Strengths and Difficulties Questionnaire (SDQ). RESULTS: Out of 104 families, 81 (77.88%) participated in the survey. Healthy siblings presented with comparable levels of emotional and behavioral difficulties compared with population norms before the COVID-19 pandemic. Compared with studies that surveyed child and adolescent mental health during the COVID-19 pandemic, parents of siblings in this study reported a significantly lower impairment rate. Psychosocial and disease-specific risk factors of the respective outcomes in healthy siblings were identified through regression analysis models. CONCLUSION: In general, health-care professionals should be aware of the possibility of siblings' mental health being at risk. Therefore, screening for psychosocial deficits may be essential in preventing psychiatric disorders in this population, especially during pandemics. TRIAL REGISTRATION: ClinicalTrials.gov: NCT04382820 (registered April 8, 2020).

Quality of life and mental health of children with rare congenital surgical diseases and their parents during the COVID-19 pandemic.

BACKGROUND: COVID-19 has affected our society at large, particularly vulnerable groups, such as children suffering from rare diseases and their parents. However, the psychosocial influences of COVID-19 on these have yet to be investigated. As such, the study's goal was to evaluate the health-related quality of life (HRQoL), quality of life (QoL), and mental health of children with rare congenital surgical diseases and their parents during the COVID-19 pandemic and lockdown measures. METHODS: A survey of n = 210 parents of children with rare congenital surgical diseases and a control group of n = 88 parents of children without rare diseases was conducted cross-sectionally between April 2020 to April 2021. Data on HRQoL, QoL, and mental health was collected using standardized psychometric questionnaires for children and parents presenting to the pediatric surgery department at a university hospital. RESULTS: Mothers of children with rare pediatric surgical diseases showed significantly lower QoL and significantly higher impairment in mental health than a control group and norm data. For fathers, this was solely the case for their QoL. Children's parent-reported HRQoL and mental health were partially impaired. Social and disease-specific risk factors of the respective outcomes in affected families were identified through regression analysis models. CONCLUSION: Parents of children with rare diseases report severe psychosocial impairment regarding themselves and their children during the COVID-19 pandemic. Therefore, affected families should receive attention and supportive care in the form of a family-center approach to alleviate the additional burden of the COVID-19 pandemic.

Perceived mental health in parents of children with rare congenital surgical diseases: a double ABCX model considering gender.

BACKGROUND: Previous research has supported the utility of the Double ABCX model of family adaptation for parents in various diseases. Nonetheless, it remains unclear how raising a child with rare congenital surgical diseases impacts the mental health of both mothers and fathers. METHODS: The potential predictors of maternal and paternal mental health in a German sample of 210 parents of children with rare congenital surgical diseases were investigated. Parents were investigated cross-sectionally utilizing standardized psychometric questionnaires that assessed factors attributed to parental adaptation within the Double ABCX model. RESULTS: Stressor pile-up, family functioning, perceived stress, and mental health were positively associated with mothers and fathers. However, further analyses revealed that family functioning, social support, and perceived stress fully mediated the positive association between stressor pile-up and mental health in mothers, but not fathers. CONCLUSION: Our findings suggest that parental adaptation to a rare congenital surgical disease in their children may be improved by increased intra- and extrafamilial resources and decreased perceived family-related stress in mothers, but not fathers. Our results may help to identify gender-specific factors that may guide clinicians and future interventions.