Context: Lymphocytic anterior hypophysitis in association with a pituitary adenoma was reported previously. In rare instance, inflammatory infiltration was confined to adenoma tissue itself, excluding anterior pituitary. Case: The patient a 27-year-old male presented with visual field defect. Further examination revealed a pituitary mass with suprasellar extension. Hormonal evaluation indicated mild hyperprolactinemia (42 ng/ml, normal < 19). After transsphenoidal resection, a pituitary adenoma showing cytoplasmic immunoreactivity to prolactin was identified. Dense and diffuse lymphocytic infiltration was seen within the tumor. At 15th month, a second transsphenoidal operation was necessary because of rapid development of visual compromise and headache. Excised surgical specimen consistent with previously resected adenoma showed diffuse lymphocytic infiltration composed of B and T cells within the adenoma tissue again. Conclusion: Presence of dense, hypophysitis-like lymphocytic infiltration within pituitary adenoma tissue obtained by two consecutive operations may reflect an host-mediated immune reaction to tumor. This rare finding could be challenging in terms of differential diagnosis and follow-up course.