Bi-thalamic stroke is rarely reported in the literature as a complication of an endonasal endoscopic procedure. It has been associated with presence of a Percheron artery variant, as well as with top of the basilar syndrome, both of which significantly increase patient surgical morbidity. Infundibulo-neurohypophysitis in turn, is an unusual inflammatory disorder affecting the infundibulum, the pituitary stalk, and the neurohypophysis. We present the case of a patient with visual impairment and an abnormal hormone profile compatible with infundibulo-neurohypophysitis, in whom tumor resection was conducted through an endoscopic endonasal approach (EEA). Patient developed postoperative bi-thalamic stroke due to Percheron artery infarct. A review of both conditions is included. This is the fourth case reported in the literature of a Percheron artery infarct, and to the best of our knowledge, the first linking it to endoscopic treatment of neurohypophysitis, itself an infrequent condition.