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Primary hypophysitis (PH) is an unusual disorder characterized by inflammatory infiltration of the pituitary gland with various degree of pituitary dysfunction. Glucocorticoids are the treatment of choice in the majority of patients. Still, in patients with poor response in glucocorticoids or when their administration is accompanied with serious side effects, the use of alternative agents should be considered; up to now, data on other therapeutic approaches remains scant mainly due to the rarity of the disease. Among them, the immunosuppressant azathioprine could represent an effective and safe alternative. In this article, we present our clinical experience of two cases with PH successfully treated with azathioprine following serious side effects after initial treatment with glucocorticoids and provide a brief review of the existing literature. [ABSTRACT FROM AUTHOR]