Both proband 15, a boy affected by TS, OCD, behavioral problems, and mild dysmorphic features, and proband 19, a girl affected by TS, mild intellectual disability, and ODD, presented the recurrent 15q11.2 deletion of about 395 Kb, which includes TUBGCP5, CYFIP1, NIPA1, and NIPA2 genes. Here, TUBGCP5 is linked to Oppositional defiant disorder.