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      ISCHEMIC PITUITARY APOPLEXY, HYPOPITUITARISM AND DIABETES INSIPIDUS: A TRIAD UNIQUE TO NECROTIZING HYPOPHYSITIS

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          Abstract

          SUMMARY – A rare case of necrotizing hypophysitis (NH) in a 52-year-old man presenting with pituitary apoplexy and sterile meningitis is described. This case indicates that the diagnosis of NH could be made without biopsy, based on concomitant presence of diabetes insipidus, hypopituitarism and radiologic features of ischemic pituitary apoplexy. Conservative management of pituitary apoplexy should be advised in NH. Additionally, this is the first report of a case of sterile meningitis caused by ischemic pituitary apoplexy.

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          Most cited references9

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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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            A radiologic score to distinguish autoimmune hypophysitis from nonsecreting pituitary adenoma preoperatively.

            Autoimmune hypophysitis (AH) mimics the more common nonsecreting pituitary adenomas and can be diagnosed with certainty only histologically. Approximately 40% of patients with AH are still misdiagnosed as having pituitary macroadenoma and undergo unnecessary surgery. MR imaging is currently the best noninvasive diagnostic tool to differentiate AH from nonsecreting adenomas, though no single radiologic sign is diagnostically accurate. The purpose of this study was to develop a scoring system that summarizes numerous MR imaging signs to increase the probability of diagnosing AH before surgery. This was a case-control study of 402 patients, which compared the presurgical pituitary MR imaging features of patients with nonsecreting pituitary adenoma and controls with AH. MR images were compared on the basis of 16 morphologic features besides sex, age, and relation to pregnancy. Only 2 of the 19 proposed features tested lacked prognostic value. When the other 17 predictors were analyzed jointly in a multiple logistic regression model, 8 (relation to pregnancy, pituitary mass volume and symmetry, signal intensity and signal intensity homogeneity after gadolinium administration, posterior pituitary bright spot presence, stalk size, and mucosal swelling) remained significant predictors of a correct classification. The diagnostic score had a global performance of 0.9917 and correctly classified 97% of the patients, with a sensitivity of 92%, a specificity of 99%, a positive predictive value of 97%, and a negative predictive value of 97% for the diagnosis of AH. This new radiologic score could be integrated into the management of patients with AH, who derive greater benefit from medical as opposed to surgical treatment.
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              Acute management of pituitary apoplexy--surgery or conservative management?

              The rarity of pituitary apoplexy renders it a difficult subject for audit; hence there are no evidence-based standards of optimum care for such patients. The key controversy in management relates to the role of acute neurosurgical intervention. In recent years we have adopted a relatively conservative approach towards patients presenting with pituitary apoplexy. Against this background, we aimed to determine whether our less-interventional approach affected long-term clinical outcome in these patients. A retrospective analysis was performed to evaluate clinical presentation, management and clinical outcomes in a cohort of patients who presented acutely with pituitary apoplexy during the period 1994-2004. Data from 33 patients (13 female) were included, with a mean age of 52 (range 27-79) years and mean follow-up duration of 3.7 (0.4-10.1) years. The most common presenting symptoms were headache (97%), visual deficits (82%) and nausea/vomiting (78%). Fifteen patients (46%) underwent transsphenoidal surgery while 18 were managed conservatively. Indications for surgery were deteriorating visual deficit (n = 13), hemiparesis (n = 1) and altered conscious level (n = 1). Eight patients in the surgical group had ocular paresis that resolved in 63% following surgery, and seven had visual field defects with recovery in 57% postsurgery. Conservative management was reserved for patients with absent, or evidence of resolving, visual deficits at presentation. In this group, seven presented initially with ocular paresis and six with visual field defects but all made full recoveries. Of the patients managed neurosurgically, 87% required long-term glucocorticoid replacement and 60% required long-term thyroid hormone replacement. Conservatively managed patients required glucocorticoid replacement in 72% and thyroid hormone replacement in 72% of cases (P = NS between the two groups). Sex steroid replacement was required in 67% and 83% of patients managed neurosurgically and conservatively respectively (P = NS). At latest follow-up one patient in the conservatively managed group had required surgery and one in the surgically managed group had received pituitary radiotherapy, in both instances due to evidence of tumour regrowth on magnetic resonance imaging (MRI). Our findings suggest that patients presenting with pituitary apoplexy in whom visual deficits are stable or improving may be managed expectantly as there is no identifiable deleterious effect on visual or endocrine outcome. One patient from each group experienced tumour regrowth that necessitated further treatment intervention, highlighting the importance of long-term follow-up in patients with pituitary apoplexy.
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                Author and article information

                Journal
                Acta Clin Croat
                Acta Clin Croat
                ACC
                Acta Clinica Croatica
                Sestre Milosrdnice University Hospital and Institute of Clinical Medical Research, Vinogradska cesta c. 29 Zagreb
                0353-9466
                1333-9451
                December 2018
                December 2018
                : 57
                : 4
                : 768-771
                Affiliations
                Mladen Sekso Department of Endocrinology, Diabetes and Metabolic Diseases, Sestre milosrdnice University Hospital Center, School of Medicine, University of Zagreb , Zagreb, Croatia; Department of Neurosurgery, Sestre milosrdnice University Hospital Center , Zagreb, Croatia; Ljudevit Jurak Department of Pathology, Sestre milosrdnice University Hospital Center , Zagreb, Croatia; Department of Radiology, Sestre milosrdnice University Hospital Center , Zagreb, Croatia
                Author notes
                Correspondence to: Ivan Kruljac, MD, Mladen Sekso Department of Endocrinology, Diabetes and Metabolic Diseases, Sestre milosrdnice University Hospital Center, Vinogradska c. 29, HR-10000 Zagreb, Croatia
E-mail: ivkruljac@ 123456gmail.com
                Article
                acc-57-768
                10.20471/acc.2018.57.04.20
                6544104
                31168215
                d621f6ff-c557-4fd1-bba9-24bd22fe2393
                Copyright @ 2018

                This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial No Derivatives (CC BY-NC-ND) 4.0 License.

                History
                : 12 April 2016
                : 13 June 2016
                Categories
                Case Reports

                stroke,diabetes insipidus,hypopituitarism,hypophysitis,pituitary apoplexy,meningitis

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