Abstract
Background: Families of children with Pediatric Acute-onset Neuropsychiatric Syndrome (PANS) and Pediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcus (PANDAS) face a unique burden related to these frequently misunderstood illnesses. PANS and PANDAS cause a variety of neuropsychiatric symptoms that disrupt every facet of daily life for children and their families. The sudden onset of symptoms and unpredictability of flares are part of the burden. Families are also burdened by difficulty accessing care and treatment. Previous qualitative research indicated PANS/PANDAS presents numerous and ongoing challenges for families of affected children, but no known research has quantitatively measured family burden and identified the correlates and predictors. The purpose of this study was to measure family burden in families affected by PANS/PANDAS and to identify factors that correlate with and predict family burden. Methods: A cross-sectional descriptive correlational survey was used to identify correlates and predictors of family burden from the parent or primary caregiver of children with PANS/PANDAS perspective. The 19-item Burden Assessment Scale was used to measure family burden. Pearson product-moment correlation and multiple regression analysis were used to explore the relationships between illness-related variables, access to care, and uncertainty with the dependent variable family burden. Variables were entered into the regression model based on theoretical relevance, allowing assessment of the incremental contribution of each set of predictors to the variance in family burden. Qualitative responses were collected to provide additional context and support for the quantitative findings. Results: A total of 154 primary caregivers of children with PANS/PANDAS completed the online survey. Mean scores of 63.54 (SD = 8.54) on the Burden Assessment Scale, indicating high burden, were noted in 98.7% of respondents. Illness related factors significantly correlated with increased family burden included the diagnosis of PANS, PANDAS, or both, illness course, symptom severity, uncertainty, unpredictability, and missed school days. Access to care factors significantly correlated with increased family burden included time to diagnosis (r = .192, p = .017), number of providers to reach diagnosis (r = .349, p < .001), current number of providers (r = .226, p = .005), and out-of-pocket costs (r = .276, p < .001). Family related factors significantly correlated with increased family burden included number of affected children in the family (r = .17, p = .035) and greater perceived threat to family integrity (r = .658, p = < .001), Standardized regression coefficients indicated the number of providers to diagnosis (β = .198, p = .002) and increased threat to family integrity (β = .549, p < .001) were found to be persistent predictors of family burden. Qualitative data analysis supported delays in diagnosis and the need to see multiple providers to reach diagnosis contributed to family burden in PANS/PANDAS. Overall, findings support family burden is multifactorial, with illness-related factors, access to care related variables, and family-related factors all contributing significantly. Conclusions: The identification of increased number of providers to reach diagnosis and perceived threat to family integrity as predictors of family burden highlights the importance of early diagnosis and family support interventions for families of children with PANS/PANDAS. Nurses and healthcare providers are in a key position to identify families who may be at greater risk due to prolonged diagnostic processes or concerns about family stability. This study highlights opportunities for nurses to develop family-centered interventions aimed at reducing family burden and supporting families coping with PANS/PANDAS. Additionally, findings suggest a need for healthcare systems to address barriers that contribute to delayed diagnosis and financial hardship for families. Future research is needed to explore the relationships of the illness-related factors significantly correlated with family burden that were not significant predictors.