This individual harbored a de novo variant in PTEN with a consistent phenotype of macrocephaly and focal epilepsy but also had the episignature for KDM2B. Further analysis identified a paternally inherited missense variant in KDM2B. We performed methylation array analysis for the unaffected father and found that he, too, harbored the KDM2B episignature. This evidence concerns the gene PTEN and focal epilepsy.