Psychosocial Outcomes of Parents of Children with Hirschsprung Disease Beyond Early Childhood.

PURPOSE: The lifelong impact of Hirschsprung disease (HD) upon children and their families is increasingly well recognized. Parental psychosocial wellbeing and family functioning are determinants of psychological and health-related outcomes in children with chronic conditions. We performed a cross-sectional cohort study to evaluate the psychosocial functioning of parents/caregivers of children with HD, beyond early childhood. METHODS: Parents/caregivers of children with HD, aged 4-14 years, managed at a tertiary pediatric surgical center were surveyed. Parent psychosocial outcomes, including adjustment to illness and family response, were assessed using four validated measures: Family Management Measure (FaMM); Parent Experience of Child Illness (PECI); Patient Reported Outcomes Measurement Information System (PROMISR) anxiety; and PROMISR depression. The Pediatric Quality of Life Inventory (PedsQL) was administered to assess child quality of life (proxy-report). RESULTS: Forty parents (mean age 38.7 ± 5.6 years) of children with HD (mean age 8.0 ± 2.5) participated. Parents expressed greater long-term uncertainty (PECI) and poorer perceived condition management ability (FaMM) than comparator chronic disease cohorts. Other scores for parental adjustment to their child's condition (PECI) and family response (FaMM) were comparable to reference cohorts. Symptoms of anxiety and depression were prevalent in our cohort (52.5 % and 42.5 % respectively); however, the proportion with moderate - severe PROMISR anxiety (χ2 = 2.50, p = 0.114) and depression (χ2 = 0.156, p = 0.693) scores did not significantly differ from the expected population distribution. Proxy-reported child quality of life (PedsQL) was significantly reduced relative to healthy children (p = 0.0003), but comparable to those with physical health problems with special healthcare needs (p = 0.624). CONCLUSIONS: Parents of children with HD experience long-term uncertainty and have poorer perceived condition management ability than parents of children with other chronic childhood illnesses. This work highlights the importance of targeted parental education and support beyond primary surgical management, and provides a benchmark for this cohort, against which subsequent intervention-based studies may be assessed. LEVEL OF EVIDENCE: II.

Is Social Categorization the Missing Link Between Weak Central Coherence and Mental State Inference Abilities in Autism? Preliminary Evidence from a General Population Sample.

We explore the relationship between the 'theory of mind' (ToM) and 'central coherence' difficulties of autism. We introduce covariation between hierarchically-embedded categories and social information--at the local level, the global level, or at both levels simultaneously--within a category confusion task. We then ask participants to infer the mental state of novel category members, and measure participants' autism-spectrum quotient (AQ). Results reveal a positive relationship between AQ and the degree of local/global social categorization, which in turn predicts the pattern of mental state inferences. These results provide preliminary evidence for a causal relationship between central coherence and ToM abilities. Implications with regard to ToM processes, social categorization, intervention, and the development of a unified account of autism are discussed.