A 41-year-old woman with central diabetes insipidus underwent trans-sphenoidal surgery for a magnetic resonance imaging (MRI) abnormality thought to be a pituitary adenoma. Upon exposure of the sella, neurosurgeons did not discover an adenoma but rather lymphocytic material per laboratory analysis leading to a diagnosis of lymphocytic hypophysitis. Upon follow-up care with an endocrinologist, this patient was also diagnosed with secondary adrenal insufficiency.Postoperatively, the patient reported a new onset of 2 headache syndromes. The first syndrome was described as bouts of right-sided periorbital severe pain associated with right-sided facial edema, lacrimation with conjunctival injection, and nasal congestion. These particular headaches occurred at least twice daily and were less than 30 minutes duration. Although bouts of periorbital headache were mostly right-sided, bilateral pain and facial edema on occasion were reported.The second pattern of headache was described as brief bouts of neuralgiform pain described as ice pick jabs and jolts in various areas of the scalp on either side that were of sudden onset and were often, but not always, associated with previously described periorbital headache, but were not always ispilateral to these.Sumatriptan helped to alleviate the periorbital headaches but a trial of indomethacin for the bouts of neuralgiform pain was not helpful. Prophylaxis for bouts of periorbital headache was changed to verapamil as patient could not tolerate topiramate due to cognitive slowing as a side effect. Ultimately, patient agreed that carbamazepine significantly reduced the number of neuralgiform pain bouts to a tolerable level and was still experiencing 2 bouts of periorbital headaches per day with verapamil as a prophylaxis, but did not find these episodes to be as severe. Although the patient found sumatriptan effective for relief of periorbital headaches, she restricted its usage to no more than 2 days in a given week for fear of transformed migraine with more frequent usage. Thus, a trial of high flow oxygen per face mask was offered as an additional abortive and this was found to be effective.