Objective

Children with orofacial clefts (OFC) may have an increased risk of poor mental health. This study aimed to investigate the risk of psychiatric diagnoses in individuals with OFC, stratified by cleft type.

Method

A nationwide register-based cohort of all individuals born with nonsyndromic OFC in Sweden between 1973 and 2012 (n = 7,842) was compared to a matched cohort (n = 78,409) as well as to their unaffected siblings (n = 9,637). The risk of psychiatric diagnoses, suicide attempts, and suicides was examined by crude and adjusted Cox regression models. Effect modification by sex was investigated with interaction terms in the models.

Results

Children with cleft lip (CL) had a significantly higher risk of any psychiatric disorder, intellectual disability, and language disorders; children with cleft lip and palate (CLP) had, in addition, an increased risk of autism spectrum disorder (ASD). Children with cleft palate only (CPO) had risk increases for the same diagnoses as children with CL and CLP, but with higher hazard ratios, and also for psychotic disorders, attention-deficit/hyperactivity disorder (ADHD), and other behavioral or emotional disorders in childhood. Sex stratification indicated higher risk increases among females in CL and CLP but not in CPO. Siblings without OFC were less likely to be diagnosed with any psychiatric disorder, intellectual disability, language disorder, ASD, or ADHD compared to their siblings with OFC.

Conclusion

Children with nonsyndromic clefts had a significantly higher risk of neurodevelopmental disorders. This risk is unlikely to be explained by familial influences such as inherited genetic or shared environmental factors.

中文翻译：

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口腔颌面裂隙儿童神经发育障碍的风险增加。

客观的

口面部裂口（OFC）的儿童可能会增加精神健康状况差的风险。这项研究旨在调查按裂痕类型分层的OFC患者进行精神病诊断的风险。

方法

比较了1973年至2012年间瑞典所有非综合征性OFC出生的全国性登记队列（n = 7,842），相匹配的队列（n = 78,409）以及未受影响的兄弟姐妹（n = 9,637）。通过粗略和调整后的Cox回归模型检查了精神病诊断，自杀未遂和自杀的风险。在模型中使用交互作用项研究了按性别进行的效果修改。

结果

唇裂（CL）儿童患任何精神病，智力障碍和语言障碍的风险明显较高；此外，唇left裂（CLP）儿童患自闭症谱系障碍（ASD）的风险增加。仅具有left裂的儿童（CPO）的风险与CL和CLP的儿童相同，但危险比更高，而且患有精神病，注意力缺陷/多动症（ADHD）以及其他行为或情感障碍在童年时期。性别分层表明，CL和CLP中女性的风险增加更高，但CPO中女性没有。与拥有OFC的兄弟姐妹相比，没有OFC的兄弟姐妹被诊断出患有任何精神疾病，智力障碍，语言障碍，ASD或ADHD的可能性更低。

结论

患有非综合征性left裂的儿童神经发育障碍的风险明显更高。这种风险不太可能由家族影响（例如遗传的遗传因素或共享的环境因素）来解释。