We report an unusual case of Parry-Romberg syndrome in which medically refractory focal epilepsy with ongoing epilepsia partialis continua (EPC) arose from the hemisphere contralateral to the side of facial atrophy. Unilateral cerebral involvement was confirmed by multi-modal brain imaging, as well as by electroencephalography (EEG) and magnetoencephalography (MEG). While in many cases of Parry-Romberg syndrome, the side of cerebral involvement is ipsilateral to that of the cutaneous lesion, these "discordant" exceptions imply that other yet undefined mechanisms may be responsible for the distribution of the cutaneous and cerebral pathologies.