Abstract

Background: Individuals with orofacial cleft (OFC) may be at higher risk of developing psychiatric disorders(PD). We determined the risk of psychiatric diagnoses in children with OFC in Canada. Methods: This population-based retrospective cohort study used health administrative data from the province of Ontario, Canada. Children with OFC who were born from April 1, 1994 to March 31, 2017 in Ontario were each matched to five non-OFC children based on sex, date of birth and mother’s age. We determined rate of events and time-to-event for first diagnosis of PD in children aged ≥3 years(y), and for intellectual developmental delay (IDD) from birth. Risk factors for PD and IDD were assessed using 1-way ANOVA for means, Kruskal-Wallis for medians, χ 2 test for categorical variables. Outcomes: There were 3,051 children with OFC (matched to 15,255 controls), of whom 2,515 OFC patients had complete follow-up to the third birthday (matched to 12,575 controls). The mean age to first PD diagnosis was 8.6±4.2y for OFC patients; the cleft palate group had a higher risk (HR 1.33, 95% CI 1.18-1.49). The OFC patients had a higher risk of IDD diagnosis (HR 8.08, 95% CI 7.28-8.97, p<001), especially those with other congenital malformations (HR 4.57, 95% CI 3.95-5.28 p<0.001). Geography (place of living) was a significant risk factor for PD and IDD. Interpretation: Children born with OFC in Ontario had a higher risk of psychiatric diagnosis and IDD compared to controls. Further research is required to better understand the cause of geographical variation and identify potential areas for intervention. Funding: Claudia Malic was supported by the ICES Faculty Scholars Program. Eric Benchimol was supported by a New Investigator Award from the Canadian Institutes of Health Research, Canadian Association of Gastroenterology, and Crohn’s and Colitis Canada. He was also supported by the Career Enhancement Program of the Canadian Child Health Clinician Scientist Program. Conflict of Interest: Simone Vigod reports royalties from UpToDate Inc for authorship of chapters related to depression and pregnancy. The remaining authors have no conflicts of interest to disclose. Ethical Approval: The Research Ethics Board of the Children’s Hospital of Eastern Ontario approved this study and informed consent was not required as per Ontario privacy law.

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