Cherubism, a rare hereditary fibro-osseous lesion characterized by painless expansion of jaws, starts early in life manifesting itself fully in the second decade of life and is almost regressed in the third decade. Here, we report a sporadic case of Cherubism with clinico-radiological and scintigraphic presentation of its classical features for its disease rarity and single-photon emission computed tomography/computed tomography findings with review the literature.