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A 64-year-old Korean woman presented with a 3-week history of severe headache and ocular pain. Her brain MRI showed a cystic pituitary mass compressing the optic chiasm. A hormonal study revealed anterior pituitary insufficiency and a slightly increased prolactin level. We performed a transsphenoidal resection of the pituitary mass. A pathological examination revealed the presence of a heavy inflammatory infiltrate, composed of lymphocytes and plasma cells, and destruction of the adenohypophysial structures. Five months after surgery, her hormonal levels had nearly normalized, without hormone replacement therapy. A follow-up MRI showed no recurrence. We conclude that lymphocytic hypophysitis should be included in the differential diagnosis of pituitary mass at any age. We discuss the features that can help to make a preoperative differential diagnosis, and selection of the appropriate treatment.