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      Spontaneous adrenal hemorrhage. Report of two cases and review of pathogenesis, diagnosis and management

      case-report

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          Abstract

          Adrenal hemorrhage represents a relatively rare condition, usually associated with meningococcal septicemia. It is an underestimated cause of acute decompensation, multiorgan failure and death, usually diagnosed post-mortem. Depending on its etiology adrenal hemorrhage is categorized as traumatic and non-traumatic. The technical advancement of imaging modalities, has made diagnosis and management more efficient. Assessment of hemodynamic stability, exclusion of a hormonal active adrenal tumor and assessment for adrenal insuffi¬ciency, are of cardinal importance. Angiographic embolization has contributed significantly in better outcomes as emergency laparotomy is associated with high morbidity and mortality rates. Hereby we present two cases of spontaneous adrenal hemorrhage associated with extensive retroperitoneal bleeding and hemodynamic instability. Both underwent angiography with one requiring embolization with favorable outcome. Investigation for exclusion of underlying adrenal tumor, adrenal insufficiency and follow-up imaging are presented in detail for both cases.

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          Most cited references10

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          Diagnosis of adrenal insufficiency.

          The cosyntropin stimulation test is the initial endocrine evaluation of suspected primary or secondary adrenal insufficiency. To critically review the utility of the cosyntropin stimulation test for evaluating adrenal insufficiency. The MEDLINE database was searched from 1966 to 2002 for all English-language papers related to the diagnosis of adrenal insufficiency. Studies with fewer than 5 persons with primary or secondary adrenal insufficiency or with fewer than 10 persons as normal controls were excluded. For secondary adrenal insufficiency, only studies that stratified participants by integrated tests of adrenal function were included. Summary receiver-operating characteristic (ROC) curves were generated from all studies that provided sensitivity and specificity data for 250-microg and 1-microg cosyntropin tests; these curves were then compared by using area under the curve (AUC) methods. All estimated values are given with 95% CIs. At a specificity of 95%, sensitivities were 97%, 57%, and 61% for summary ROC curves in tests for primary adrenal insufficiency (250-microg cosyntropin test), secondary adrenal insufficiency (250-microg cosyntropin test), and secondary adrenal insufficiency (1-microg cosyntropin test), respectively. The area under the curve for primary adrenal insufficiency was significantly greater than the AUC for secondary adrenal insufficiency for the high-dose cosyntropin test (P 0.5) for secondary adrenal insufficiency. At a specificity of 95%, summary ROC analysis for the 250-microg cosyntropin test yielded a positive likelihood ratio of 11.5 (95% CI, 8.7 to 14.2) and a negative likelihood ratio of 0.45 (CI, 0.30 to 0.60) for the diagnosis of secondary adrenal insufficiency. Cortisol response to cosyntropin varies considerably among healthy persons. The cosyntropin test performs well in patients with primary adrenal insufficiency, but the lower sensitivity in patients with secondary adrenal insufficiency necessitates use of tests involving stimulation of the hypothalamus if the pretest probability is sufficiently high. The operating characteristics of the 250-microg and 1-microg cosyntropin tests are similar.
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            Imaging of nontraumatic hemorrhage of the adrenal gland.

            Nontraumatic hemorrhage of the adrenal gland is uncommon. The causes of such hemorrhage can be classified into five categories: (a) stress, (b) hemorrhagic diathesis or coagulopathy, (c) neonatal stress, (d) underlying adrenal tumors, and (e) idiopathic disease. Computed tomography (CT), ultrasonography (US), and magnetic resonance (MR) imaging play an important role in diagnosis and management. CT is the modality of choice for evaluation of adrenal hemorrhage in a patient with a history of stress or a hemorrhagic diathesis or coagulopathy (anticoagulant therapy). CT may yield the first clue to the diagnosis of adrenal insufficiency secondary to bilateral massive adrenal hemorrhage; such insufficiency is rare but life threatening. US is the modality of choice for evaluation of neonatal hematoma, and MR imaging is helpful for further characterization. MR imaging is also useful in the diagnosis of coexistent renal vein thrombosis. When an adrenal abscess is suspected, percutaneous aspiration and drainage under imaging guidance should be performed. Hemorrhage into an adrenal cyst or tumor can cause acute onset of symptoms and signs in a patient without discernible risk factors for adrenal hemorrhage. A hemorrhagic adrenal tumor should be suspected when CT or MR imaging reveals a hemorrhagic adrenal mass of heterogeneous attenuation or signal intensity that demonstrates enhancement.
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              Adrenal hemorrhage: a 25-year experience at the Mayo Clinic.

              To characterize the clinical course of adrenal hemorrhage (AH) by using a systematic review of the presentation, associated conditions, and outcomes in patients with AH seen at our institution between 1972 and 1997 (a 25-year period). A computer search of recorded dismissal diagnoses identified 204 patients with a diagnosis of AH, but only 141 fulfilled our study criteria. Their records were analyzed systematically by presentation, bilateral or unilateral hemorrhage, corticosteroid treatment, and survival. AH is a heterogeneous entity that occurs in the postoperative period, in the antiphospholipid-antibody syndrome, in heparin-associated thrombocytopenia, or in the setting of severe physical stress and multiorgan failure. Standard laboratory evaluation is not helpful in establishing the diagnosis. Of the 141 cases of AH, 78 were bilateral, and 63 were unilateral. Corticosteroid treatment in situations of severe stress or sepsis had little effect on outcome (9% vs. 6% survival with and without corticosteroid treatment, respectively). This is in sharp contrast to AH occurring postoperatively (100% vs. 17% survival with or without treatment, respectively) or in the antiphospholipid-antibody syndrome (73% vs. 0% survival, respectively). A high index of suspicion is required to make a timely diagnosis of AH. Fever and hypotension in the appropriate clinical setting necessitate further investigation. Although the diagnosis of AH is infrequently made while the patient is alive, appropriate imaging techniques are useful for establishing a timely diagnosis. In severe physical stress or sepsis, AH may be a marker of severe, preterminal physiologic stress and poor outcome.
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                Author and article information

                Journal
                J Surg Case Rep
                J Surg Case Rep
                jscr
                Journal of Surgical Case Reports
                Oxford University Press
                2042-8812
                June 2018
                11 June 2018
                11 June 2018
                : 2018
                : 6
                : rjy129
                Affiliations
                [1 ]Department of General Surgery, South Warwickshire NHS Foundation Trust, Warwick, UK
                [2 ]Department of Surgical Oncology, Heraklion University Hospital, Crete, Greece
                [3 ]Department of General Surgery, Heart of England NHS Foundation Trust, Birmingham, UK
                [4 ]Department of General Surgery, Venizeleion General Hospital, Crete, Greece
                Author notes
                Correspondence address. South Warwickshire NHS Foundation Trust, Lakin Road, CV34 5BW Warwick, UK. Tel: +44-744-525-8227; Email: vcharalambakis@ 123456gmail.com
                Article
                rjy129
                10.1093/jscr/rjy129
                6007599
                4f3f2bcc-06a4-401a-9371-fd7d5908f313
                Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@ 123456oup.com

                History
                : 15 April 2018
                : 22 May 2018
                Page count
                Pages: 4
                Categories
                Case Report

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