Here we find strong evidence implicating rare de novo CNVs in genetic risk for bipolar disorder. We also confirm previous findings that de novo CNVs occur at a significantly increased rate in individuals with schizophrenia (Xu et al., 2008). Based on our study, a contemporaneous study (Kirov et al., 2011), and an earlier study by our coauthor (Xu et al., 2008), we estimate that the overall frequency of de novo CNVs > 10 kb is approximately 4% in BD and 5%–10% in SCZ.