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Nonsyndromic Craniosynostosis Is Associated with Increased Risk for Psychiatric Disorders
Karin K Tillman1, Jonas Höijer1, Mia Ramklint1
1From the Department of Neuroscience, Divisions of Child and Adolescent Psychiatry and Psychiatry, and the Department of Surgical Sciences, Division of Plastic Surgery, Uppsala University.
Insights
Children with nonsyndromic craniosynostosis face a significantly higher risk of psychiatric disorders, including intellectual and language impairments. This increased risk persists even after accounting for familial factors, suggesting non-genetic influences.
Area of Science:
- Pediatric neurology
- Psychiatry
- Genetics
Background:
- Craniosynostosis is a common craniofacial malformation requiring surgical intervention in infancy.
- Limited data exist on the psychiatric morbidity associated with nonsyndromic craniosynostosis.
- This study addresses the gap in knowledge regarding psychiatric disorder risk in affected children.
Purpose of the Study:
- To investigate the risk of psychiatric disorders in individuals with nonsyndromic craniosynostosis.
- To compare psychiatric morbidity in affected individuals with community and sibling cohorts.
- To identify specific psychiatric conditions associated with nonsyndromic craniosynostosis.
Main Methods:
- A register-based cohort study in Sweden (1973-2012) included 1238 individuals with nonsyndromic craniosynostosis.
- Comparison with a matched community cohort (12,380 individuals) and unaffected full siblings (1485 individuals).
- Cox regression analysis adjusted for various perinatal, somatic, and familial factors was employed to assess psychiatric disorder risk.
Main Results:
- Nonsyndromic craniosynostosis is linked to a 1.70-fold increased risk of any psychiatric disorder.
- Significant associations were found with intellectual disability (HR 4.96), language disorders (HR 2.36), and neurodevelopmental disorders (HR 1.30).
- Increased risk for psychiatric disorders, particularly intellectual disability, was observed in affected siblings compared to unaffected ones, even after adjustments.
Conclusions:
- Children with nonsyndromic craniosynostosis exhibit a higher risk for psychiatric disorders.
- Familial influences do not fully explain this elevated risk.
- The findings highlight the need for comprehensive psychiatric monitoring in children with nonsyndromic craniosynostosis.
Background:
Craniosynostosis is one of the most common craniofacial malformations demanding surgical treatment in infancy. Data on overall psychiatric morbidity among children with nonsyndromic craniosynostosis remain limited. This study investigated the risk of psychiatric disorders in nonsyndromic craniosynostosis.
Methods:
The authors reviewed a register-based cohort of all individuals born with nonsyndromic craniosynostosis in Sweden between 1973 to 1986 and 1997 to 2012 (n = 1238). The nonsyndromic craniosynostosis cohort was compared with a matched community cohort (n = 12,380) and with unaffected full siblings (n = 1485). The authors investigated the risk of psychiatric disorders, suicide attempts, and suicides by using Cox regression adjusted for perinatal and somatic factors, season and birth year, sex, parental socioeconomic factors, and parental psychiatric disorders.
Results:
Children with nonsyndromic craniosynostosis had a higher risk of any psychiatric disorder (adjusted Cox-derived hazard ratio, 1.70; 95 percent CI, 1.43 to 2.02), including intellectual disability (adjusted Cox-derived hazard ratio, 4.96; 95 percent CI, 3.20 to 7.70), language disorders (adjusted Cox-derived hazard ratio, 2.36; 95 percent CI, 1.57 to 3.54), neurodevelopmental disorders (adjusted Cox-derived hazard ratio, 1.30; 95 percent CI, 1.01 to 1.69), and other psychiatric disorders (adjusted Cox-derived hazard ratio, 1.43; 95 percent CI, 1.11 to 1.85). Full siblings with nonsyndromic craniosynostosis were more likely, in the crude analyses, to be diagnosed with any psychiatric disorder, including intellectual disability, language disorders, and neurodevelopmental disorders compared with nonaffected siblings. The higher risk for any psychiatric disorder and intellectual disability remained after adjusting for confounders.
Conclusions:
Children with nonsyndromic craniosynostosis demonstrated higher risks of any psychiatric disorder compared with children without nonsyndromic craniosynostosis. This risk cannot fully be explained by familial influences (i.e., genetic or environmental factors).
Clinical Question/Level Of Evidence:
Risk, III.
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