Cherubism is a rare hereditary benign lesion of the mandible that appears in childhood as bilateral painless swellings that progress until puberty, and then spontaneously regress. It was first described in 1933 and referred to as familial multilocular cystic disease of the mandible. These lesions are rarely seen and the diagnosis of the lesion requires the integration of the histopathology with history, clinical course of the disease and the radiographic appearance. Plain radiographs and computed tomography (CT) scan are sufficient for radiological diagnosis. Orbital involvement secondary to cherubism is rare. In this article, we describe CT and three-dimensional (3D) imaging features in two cases with extensive orbital involvement of cherubism.