Introduction: Hypophysitis is a rare, inflammatory disorder of the pituitary that mimics neoplastic lesions resulting in anterior and posterior pituitary dysfunction. Presentation of case:We present a 36-year-old woman who presented with chronic headaches and was found to have a pituitary mass on MRI, suggested of craniopharyngioma, and the histological inspection showed a piloide gliosis, one year ago. She continuing with visual disturbance and recurrence was suspected and she was operated to craniopharyngioma. After that, she showed clinical and radiologically symptoms of recurrence and was reopeated which biopsied was reported as xantogranulomatosous hypophysitis.Discussion: This woman illustrates a rare pituitary pathology presented with a literature review of published patients describing xanthomatous hypophysitis. In this case we describe a patients with xanthogranulomatous hypophysitis (XGH) demonstrating post-surgical procedure of craniopharyngioma resection. Conclusion:Xantomagranulomatous reaction was due to rupture of cyst walls of craniopharyngioma or natural immunological and inflammatory mechanism of reduction of tumor growth and progression of disease caused by extravased cyst content into the adjacent gland and favoring the reduction or regression of tumor.

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