Psammomatoid ossifying fibroma (PsOF) is a benign fibro-osseous neoplasm that predominantly affects frontal and ethmoid bones, with a preference for adolescents and young adults. The clinical and morphologic features of PsOF overlap with other fibro-osseous lesions, and additional molecular markers may help increase their diagnostic accuracy. Chromosomal breakpoints at bands Xq26 and 2q33 have been previously described. Our guest, Dr. Arjen Cleven, discusses his group’s recent identification of a SATB2 rearrangement in PsOF.