Individuals with orofacial clefts (OFCs) may be at an increased risk of developing autism spectrum disorder (ASD) and attention deficit hyperactivity disorder (ADHD). This systematic review provides a summary of the most recent data regarding the prevalence of ASD and ADHD in the OFC population and compares this to the general paediatric population. Multiple databases were searched including PubMed/Medline and Embase in July 2024, following the Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines and was registered in PROSPERO (CRD42024565219). 1025 papers were identified for screening, of which nine were included in the report. Percentage prevalence was calculated and compared to global prevalence or control populations where available. Overall, ASD prevalence among children with a cleft ranged from 0 to 50% (Mean = 2.87%; SD = 5.40) compared to ~ 1% globally (WHO) and ADHD prevalence ranged from 2.34 to 31.71% (Mean = 3.63%; SD = 4.30) compared to ~ 5% globally (NICE). Matched control populations showed larger differences. Isolated cleft palate was associated with higher rates than isolated cleft lip or combined cleft lip and palate. Prevalence in individuals with syndromic clefts appeared greater still (Mean = 14.80%; SD = 16.58) although populations were small. Children with OFCs demonstrate increased prevalence of ASD compared to the average paediatric population. Evidence for increased prevalence of ADHD is less clear, with varying rates across studies. Children with isolated cleft palate or cleft associated with genetic syndromes appear most at risk, although further research is needed.
Literature has demonstrated a potentially elevated risk of certain neurodevelopmental disorders in individuals with orofacial clefts (OFCs). This study found an increased prevalence of ASD in children with orofacial clefts compared to matched controls and global prevalence. ADHD may also be increased in these populations, although data is less clear. Both ASD and ADHD prevalence appeared even greater in individuals with syndrome-associated OFCs, as well as those with cleft palate only (CPO). Clinical assessments should involve greater vigilance for ASD and ADHD amongst both clinicians and parents/guardians through patient education. Future research is required to clarify the nature of the association between ASD/ADHD and OFCs and identify any causative factors that may lead to this increased risk.
Keywords: 22q11 deletion syndrome; Attention deficit hyperactivity disorder; Autism spectrum disorder; Cleft lip and palate; Orofacial clefts.
© 2024. The Author(s).