From the Department of Neuroscience, Divisions of Child and Adolescent Psychiatry and Psychiatry, and the Department of Surgical Sciences, Division of Plastic Surgery, Uppsala University.
Plast Reconstr Surg. 2020 Aug;146(2):355-365. doi: 10.1097/PRS.0000000000007009.
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.
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.
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.
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.
颅缝早闭是婴儿期最常见的颅面畸形之一，需要手术治疗。非综合征性颅缝早闭儿童的整体精神发病率数据仍然有限。本研究调查了非综合征性颅缝早闭的精神障碍风险。
作者回顾了瑞典在 1973 年至 1986 年和 1997 年至 2012 年期间所有非综合征性颅缝早闭患者的登记队列（n=1238）。非综合征性颅缝早闭队列与匹配的社区队列（n=12380）和未受影响的全同胞（n=1485）进行了比较。作者通过使用调整围产期和躯体因素、季节和出生年份、性别、父母社会经济因素以及父母精神疾病的 Cox 回归来调查精神障碍、自杀未遂和自杀的风险。
患有非综合征性颅缝早闭的儿童患任何精神障碍的风险较高（调整后的 Cox 衍生风险比，1.70；95%CI，1.43 至 2.02），包括智力障碍（调整后的 Cox 衍生风险比，4.96；95%CI，3.20 至 7.70）、语言障碍（调整后的 Cox 衍生风险比，2.36；95%CI，1.57 至 3.54）、神经发育障碍（调整后的 Cox 衍生风险比，1.30；95%CI，1.01 至 1.69）和其他精神障碍（调整后的 Cox 衍生风险比，1.43；95%CI，1.11 至 1.85）。在未调整的分析中，患有非综合征性颅缝早闭的全同胞更有可能被诊断为任何精神障碍，包括智力障碍、语言障碍和神经发育障碍，而非未受影响的同胞。在调整了混杂因素后，任何精神障碍和智力障碍的风险仍然较高。
与无非综合征性颅缝早闭的儿童相比，患有非综合征性颅缝早闭的儿童表现出更高的任何精神障碍风险。这种风险不能完全用家族影响（即遗传或环境因素）来解释。
临床问题/证据水平：风险，III。
