Psychosocial Outcomes in Children with and without Non-Syndromic Craniosynostosis: Findings from Two Studies

Abstract

Objective To evaluate the hypothesis that children with craniosynostosis and their parents have differences in psychosocial outcomes, as compared with an unaffected control group. Design Two studies were conducted, both which followed children born with and without craniosynostosis. Study 1 ascertained affected children from clinics and Study 2 ascertained affected children from a population-based study of birth defects. Participants Study 1 included 22 children with single-suture craniosynostosis and 18 controls, ages 4–5 years. Study 2 included 24 children with non-syndromic craniosynostosis and 124 unaffected controls, ages 5–9 years. Main Outcome Measures Outcome measures included the Child Behavior Checklist (CBCL), Social Competence Scale (SCS), Pediatric Quality of Life Inventory (PedsQL™), and Parenting Stress Index (PSI). Results We observed lower scores on measures of health-related quality of life in cases versus controls, with adjusted effect sizes ranging from −0.72 to −0.44 (p<0.05) on summary measures. Small but statistically non-significant increases in behavioral problems were observed in cases versus controls, with no apparent differences in social competence or parenting stress. Conclusions Results provide preliminary evidence suggesting that children with non-syndromic craniosynostosis may have elevated risk for psychosocial difficulties, particularly health-related quality of life. Continued follow-up through pre-adolescence and adolescence is warranted.

Craniosynostosis: Caring for Infants and Their Families

“…All of these factors can potentially affect parents' responsiveness and adaptation to an infant who has a craniofacial abnormality. 25 In studies [25][26][27][28] of the impact of syndromic craniosynostosis on quality of life, parents' scores for children with syndromic or complex cranio synostosis were significantly lower than scores for parents of children without the abnormality. Mothers of children with the abnormality reported greater stress if they perceived their child's condition as noticeable to others.…”

Parental Perceptions of Neurodevelopment in Toddlers Following Craniosynostosis Repair

“…[14][15][16] By school age, patients with nonsyndromic craniosynostosis have significantly lower IQ and math computation scores, increased behavioral problems, lower attention and executive function, and higher rates of developmental abnormalities. 4,9,14,[17][18][19] Parents of males reported significantly higher scores when rating whether the child was different for the better after surgery. Several parents of females reported low scores, 1 or ''strongly disagree,'' which increased the standard deviation and affected results.…”

Disorders of Sex Development: Lessons to be Learned from Studies of Spina Bifida and Craniofacial Conditions

“…Research methodologies mentioned earlier will help us all to look more objectively at whether or not the treatments, surgeries, and counseling that are done truly help to improve the quality of life of patients and other family members over time. In the area of craniofacial care, longitudinal cohort studies are underway to examine youth with diagnoses such as single suture synostosis [40]) and qualitative research is ongoing to determine if the clinical work done to support children and families is "on track" [33,41]. This research will examine whether the current standard of care (i. e., having social workers meet all families and children after birth and following them over 20 years) is useful to families.…”