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ABSTRACT Description of a patient with Fuchs endothelial dystrophy submitted to a corneal transplant, performed by Descemet membrane endothelial keratoplasty, which evolved with sudden, paroxysmal pain in the frontotemporal region, postoperatively. Due to the ophthalmologic picture of the patient, the attending physician believed in possible rejection of the graft, neglecting the complaint of pain. Even after a successful second transplant, performed due to primary failure, disabling pain persisted and the physician did not manage it. After years of investigation, consulting with several specialists, it was concluded the patient presented trigeminal neuralgia that had not been treated since the surgical procedure. In addition, it led to several psychosocial consequences. Therefore, it is essential to be aware trigeminal neuralgia is a possible outcome of corneal transplantation, and its symptoms should not be neglected by the attending physician, thus contributing to better management for transplanted patients.