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Adrenal Hemorrhage: a Single Center Experience and Literature Review Original papers Adrenal hemorrhage: A single center experience and literature review Izabela M. KarwackaA–F, Łukasz ObołończykC–E, Krzysztof SworczakA,C–F Department of Endocrinology and Internal Diseases, Medical University of Gdańsk, Poland A – research concept and design; B – collection and/or assembly of data; C – data analysis and interpretation; D – writing the article; E – critical revision of the article; F – final approval of the article Advances in Clinical and Experimental Medicine, ISSN 1899-5276 (print), ISSN 2451-2680 (online) Adv Clin Exp Med. 2018;27(5):681–687 Address for correspondence Abstract Izabela M. Karwacka E-mail: [email protected] Background. Adrenal hemorrhage (AH) is a rare condition that can lead to acute adrenal insufficiency and may be fatal. The risk factors of AH include focal adrenal lesion, abdominal trauma and anticoagulation therapy. Funding sources None declared The clinical manifestation of AH varies widely; the symptoms may be related to adrenal insufficiency or may reflect multiple organ failure. However, in many cases, the course of AH is asymptomatic. Conflict of interest Objectives. The study is a retrospective analysis of 23 cases of AH, whose aim is to discuss the etiology and None declared the management of selected patients, as well as a literature review. Material and methods. The paper presents a retrospective analysis of 23 patients with AH confirmed Received on October 27, 2016 by radiological and/or pathological examination. Epidemiological data, the results of laboratory tests, and Reviewed on November 18, 2016 Accepted on February 9, 2017 radiological and pathological examinations were included in the analysis. Results. The risk factors of AH were not established in 13 patients, 5 patients had experienced a trauma prior to AH diagnosis, 1 patient was diagnosed with sepsis, 2 patients had concomitant neoplastic disease, and in 2 patients, 2 risk factors were present. Among patients who required emergency admission, 5 patients were hospitalized due to acute abdominal pain, 1 patient due to sepsis and 1 patient due to symptoms of active endocrinopathy. In the remaining patients, diagnostic procedures were prompted by the detection of adrenal incidentaloma (AI). A total of 40% of patients underwent surgical treatment due to the magnitude of AH or clinical and laboratory evidence of overt endocrinopathy. In the remaining patients, conservative treatment and further observation was recommended. In 34.8% of these patients, follow-up examinations revealed a gradual regression. Conclusions. It seems that there is a need to distinguish patients with AH who do not require surgical intervention. Follow-up radiological examination is necessary to reassess the lesion. The patients in whom shrinkage of the tumor can be observed are likely not to require surgical treatment. Key words: adrenal glands, hemorrhage, pseudocyst, primary adrenal insufficiency, adrenal incidentaloma DOI 10.17219/acem/68897 Copyright © 2018 by Wroclaw Medical University This is an article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc-nd/4.0/) 682 I. Karwacka, Ł. Obołończyk, K. Sworczak. Adrenal hemorrhage: A single center experience Introduction and in 2 patients (8.7%), 2 risk factors were present: anti- coagulant drugs and lung cancer, and trauma and chronic Adrenal hemorrhage (AH) is a rare condition that can lead oral anticoagulation. to acute adrenal insufficiency and may be fatal. It is poten- Among patients who required emergency admission, tially life-threatening when the adrenal glands are involved 5 patients (21.7%) were hospitalized due to acute abdomi- bilaterally, although at least 90% of each adrenal cortex must nal pain, 1 patient (4.3%) due to sepsis and 1 patient (4.3%) be compromised before this is clinically evident. The inci- due to symptoms of active endocrinopathy. In the re- dence of spontaneous AH based on data from autopsy stud- maining 16 patients (69.7%), diagnostic procedures were ies is 0.14–1.1%. The risk factors of AH include focal adrenal prompted by AI. The symptoms of adrenal insufficiency lesion, abdominal trauma, anticoagulation therapy, congeni- (in the course of sepsis) were confirmed in 1 patient, and tal or acquired bleeding disorders, sepsis, and pregnancy.1,2 1 patient had treatment-resistant hypertension. The rest Bleeding of an adrenal gland tumor is most frequently ob- of the patients were asymptomatic. served in pheochromocytoma and adrenal metastases.1,3–6 The results of biochemical tests and hormone assays Larger lesions of adrenal myelolipoma (>5 cm) rarely present in serum and urine – e.g., cortisol, adrenocorticotropic with acute retroperitoneal AH.6 The clinical manifestation hormone (ACTH), androstenedione, dehydroepiandros- of AH varies widely; the symptoms may be related to adrenal terone sulfate, and metoxycatecholamine (MT) – and ra- insufficiency or may reflect multiple organ failure. However, diological images of the study participants were analyzed. in many cases, the course of AH is asymptomatic.7 In 13 patients (56.2%), laboratory test results were within Until recently, AH diagnosis was often made at post- the range of normal values. The remaining 10 patients had mortem examination.8 Currently, due to the increased laboratory abnormalities, including biochemical markers availability of modern imaging techniques, AH is more of adrenal insufficiency (4.3%), hypercortisolemia (8.7%), frequently diagnosed intravitally and in many cases lesions elevated urinary MTs (mainly normetanephrine, 21.8%), and in the adrenal glands are detected unexpectedly (adrenal disorders of both the corticotropic axis and MT secretion incidentaloma – AI). The features of AH on radiological (8.7%). In all patients, computed tomography (CT) examina- imaging are specific; therefore, its diagnosis based on im- tion was performed. A lesion in the right adrenal gland was aging studies is relatively simple.8,9 found in 12 patients (52.2%), a lesion in the left adrenal gland Data on AH in the literature are scarce and management was found in 10 patients (43.5%), and 1 patient had bilateral standards have not been precisely established. It seems lesions. The diameter of the AH ranged from 17 to 150 mm that there is a need to redefine AH risk factors and to estab- (mean diameter: 60.6 mm) (Table 2). lish guidelines for the management of high-risk patients, Nine patients underwent surgical treatment (40%) due particularly considering that some AH cases may be as- to the magnitude of AH or to clinical and laboratory evi- sociated with metastases or pheochromocytoma. dence of overt endocrinopathy. The patient in whom both This paper summarizes data on 23 cases of AH, and dis- an elevated MT level and hypercortisolemia were found was cusses the etiology and the management of selected patients. disqualified from surgery due to the burden of concomitant diseases. It should be stressed that the results of radiological examinations did not raise any oncological concerns. More- Material and methods over, pathological assessment unequivocally indicated AH; only in 1 specimen was AH with the presence of neoplastic This paper presents a retrospective analysis of 23 pa- cells (a metastasis from lung cancer) found. In the remaining tients with AH confirmed by radiological and/or patho- group of patients (n = 14; 60%) conservative treatment and logical examination. The study group included patients further observation was recommended. In this group, specif- treated at the Department of Endocrinology and Internal ic risk factors were identified (sepsis, trauma and anticoagu- Diseases and the Outpatient Clinic of the University Clini- lation) and no clinical or laboratory evidence of endocrinopa- cal Center in Gdańsk from 2002 to 2016. Epidemiological thy was observed, while the adrenal lesions were explicitly data, the results of laboratory tests, and radiological and described in CT reports as AH. Follow-up examinations pathological examinations were included in the analysis. performed in 8 patients (34.8%) revealed a gradual regression of AH, and in the patient with bilateral lesions, a complete absorption of hematomas was found. Table 2 summarizes Results the clinical course of AH in the study population. The study group included 23 patients; 60.8% women and 39.2% men. The mean age was 60.6 years (Table 1). Discussion The risk factors of AH were not established in 13 patients (56.6%), 5 patients (21.8%) had experienced a trauma prior The pathophysiological mechanism of AH remains un- to AH diagnosis, 1 patient (4.3%) was diagnosed with sep- clear. The adrenal glands have unique vasculature provid- sis, 2 patients (8.7%) had concomitant neoplastic disease, ing abundant blood supply, which significantly increases Adv Clin Exp Med. 2018;27(5):681–687 683 Table 1. Study group characteristics Adrenal Size on CT No. Age Gender Risk factor Laboratory tests Intervention Additional information gland [mm] 1 38 female trauma right 30 × 17 normal observation none anticoagulant decreased size of mass to 25 mm 2 70 male drugs, lung right 55 × 40 elevated normetanephrine observation at 18 months cancer 3 60 female urosepsis bilateral 52 × 32 adrenal insufficiency observation resolution of mass at 12 months decreased size of mass to 60 mm 4 87 male unknown right 115 × 70 normal observation at 3 months hypercortisolemia, elevated decreased size of mass to 25 mm 5 67 female unknown right 32 × 28 observation normetanephrine
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