Background: Bi-thalamic stroke following an endoscopic endonasal approach (EEA) is a rare complication scarcely reported in the literature. Its occurrence has been associated both with the anatomical Percheron artery variant as well as with the top of the basilar syndrome, and it results in significant morbidity for the patient. Infundibulo-neurohypophysitis is an unusual inflammatory entity that affects the infundibulum, the pituitary stalk, and the neurohypophysis. Methods and results: We present a case of a patient with Infundibulo-neurohypophysitis who underwent an EEA for diagnosis and tumor resection due to visual and hormonal profile impairment. After the surgery, the patient developed a bi-thalamic stroke due to a Percheron artery infarct. We also conduct a review of both pathologies. Conclusion: This corresponds to the fifth reported case of this complication and the first linking Percheron artery infarct with the endoscopic treatment of neurohypophysitis, an infrequent disease itself.