A significantly higher prevalence of de novo damaging variants (33.9%) was found among probands. These variants were estimated to be present in 22.2% of overall OCD cases.SCUBE1 and CHD8 were identified as high-confidence risk genes for OCD. Genes carrying these variants significantly overlap among ASD and TS probands and were associated with immune system-related processes. Here, SCUBE1 is linked to Timothy syndrome.