Lymphocytic hypophysitis is a rare inflammatory disease of the pituitary gland that is one of the cause of hypopituitarism. The majority of cases occur in women during the peripartum period, and it is rare in men. Testicular feminization syndrome is a genetic disorder presenting a 46XY karyotype, but a normal female phenotype. We report a case of lymphocytic hypophysitis in a 23-year-old woman with testicular feminization syndrome.