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      Lymphocytic Hypophysitis: Differential Diagnosis and Effects of High-Dose Pulse Steroids, Followed by Azathioprine, on the Pituitary Mass and Endocrine Abnormalities — Report of a Case and Literature Review

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          We report on a man with a progressively increasing pituitary mass, as demonstrated by MRI. It produced neurological and ophthalmological symptoms, and, ultimately, hypopituitarism. MRI also showed enlargement of the pituitary stalk and a dural tail phenomenon. An increased titer of antipituitary antibodies (1:16) was detected in the serum. Pituitary biopsy showed autoimmune hypophysitis (AH). Neither methylprednisolone pulse therapy nor a subsequent treatment with azathioprine were successful in recovering pituitary function, or in inducing a significant reduction of the pituitary mass after an initial, transient clinical and neuroradiological improvement. Anterior pituitary function evaluation revealed persistent hypopituitarism. AH is a relatively rare condition, particularly in males, but it represents an emerging entity in the diagnostic management of pituitary masses. This case shows that response to appropriate therapy for hypophysitis may not be very favorable and confirms that diagnostic management of nonsecreting pituitary masses can be a challenge. Clinical, imaging, and laboratory findings are useful for suggesting the diagnosis, but pituitary biopsy may be necessary to confirm it.

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          Most cited references 26

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          Autoimmune hypophysitis.

          Autoimmune (lymphocytic) hypophysitis is a rare disease that should be considered in the differential diagnosis of any nonsecreting pituitary mass, especially when occurring during pregnancy or postpartum. We have analyzed 370 articles published from January 1962 to October 2004 and identified a total of 379 patients with primary lymphocytic hypophysitis. The present review synthesizes the clinical and research data reported in this body of scientific literature.
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            Ipilimumab: controversies in its development, utility and autoimmune adverse events.

            A promising new class of anti-cancer drugs includes antibodies that mediate immune regulatory effects. It has become very clear over the last decade that different types of immune cells and different pathways serve to suppress anti-cancer immunity, particularly in the microenvironment of the tumor. The first examples of immune modulating antibodies are those directed against cytotoxic T lymphocyte antigen-4 (CTLA-4), a molecule present on activated T cells. Human antibodies that abrogate the function of CTLA-4 have been tested in the clinic and found to have clinical activity against melanoma. In this review, we discuss some of the controversies surrounding the potential clinical utility of one of those antibodies, ipilimumab, formerly MDX-010, from Medarex and Bristol Myers Squibb. The optimal dose and schedule of ipilimumab was derived in multiple clinical trials whose latest results are described below. Favorable survival in patients with stage IV melanoma were observed that appear to be associated with unique side effects of the drug called "immune-related adverse events". The management of these side effects is described, and the unusual kinetics of anti-tumor response with ipilimumab as well as a newly proposed schema for assessing anti-tumor responses in patients receiving biologic compounds like ipilimumab, which may supercede RECIST or WHO criteria, are addressed.
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              Differential diagnosis of sellar masses.

               P. Freda,  K Post (1999)
              The differential diagnosis of nonpituitary sellar masses is broad; differentiating among potential etiologies may not always be straightforward because many of these lesions, tumorous and nontumorous, may mimic the clinical, endocrinologic, and radiologic presentations of pituitary adenomas. This article provides an overview of the clinical and radiographic characteristics of both pituitary tumors and the nonpituitary lesions found in the sellar/parasellar region and discusses, in detail, the specific nonpituitary origins of the sellar masses.

                Author and article information

                The Scientific World Journal
                21 January 2010
                : 10
                : 126-134
                1 Department of Medicine and Pharmacology, Section of Endocrinology, University of Messina, Italy
                2 Department of Neurosurgery, San Raffaele Scientific Institute, University Vita-Salute, Milan, Italy
                3 Department of Pathology, San Raffaele Scientific Institute, University Vita-Salute, Milan, Italy
                4 Translational Research Unit, Department of Oncology, Hospital of Prato, Istituto Toscana Tumori, Florence, Italy
                Author notes
                *Corresponding author.

                Academic Editor: Lawrence A. Frohman

                Copyright © 2010 Lorenzo Curtò et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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