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      Intrahepatic adrenocortical adenoma arising from adrenohepatic fusion mimicking hepatic malignancy : Two case reports

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          Abstract

          Rationale:

          Intrahepatic adrenocortical adenoma (IAA) arising from adrenohepatic fusion (AHF) is rare and its imaging findings are not well established. Moreover, it is easily misdiagnosed as malignant hepatic tumor in patients at risk of malignancy. Its key finding is the connection between the tumor and adrenal gland. When IAA from AHF is suspected, biopsy should be considered to avoid unnecessary surgery. Herein, we report 2 cases of IAA from AHF.

          Patient concerns:

          A 59-year-old woman was admitted due to a 1.5-cm hypoechoic nodule in the right hepatic lobe detected on ultrasound for hepatocellular carcinoma (HCC) surveillance due to chronic hepatitis B. Contrast-enhanced computed tomography (CT) and gadoxetic acid-enhanced magnetic resonance imaging (MRI) were performed to evaluate the hepatic mass. Another 75-year-old woman was admitted due to rectal adenocarcinoma detected on colonoscopy. Contrast-enhanced CT depicted a 2.5-cm mass in the right hepatic lobe.

          Diagnosis:

          In case 1, CT and MRI showed a 1.5-cm subcapsular mass in the right hepatic lobe with typical findings of HCC in a patient with chronic hepatitis B. The mass was confirmed as IAA from AHF after the laparoscopic surgery. In case 2, CT showed advanced rectal malignancy and a 2.5-cm poorly enhancing mass in the right hepatic lobe. The tentative diagnosis was hepatic metastasis. However, based on the connection between the tumor and adrenal gland during preoperative review, the presumed diagnosis was changed to IAA from AHF, which was confirmed on biopsy.

          Interventions:

          The hepatic mass connected with the right adrenal gland was laparoscopically resected in case 1. Laparoscopic lower anterior resection for rectal malignancy and percutaneous biopsy for the hepatic mass were performed in case 2.

          Outcomes:

          The first patient had an uneventful recovery, without recurrence on the 3-year follow-up CT. The second patient had an uneventful postoperative course and has been alive for 12 months postoperatively without pathologically proven IAA changes on follow-up CT.

          Lessons:

          When hepatic mass is found adjacent to the right adrenal gland on imaging, the connection between the tumor and adrenal gland should be investigated. When IAA arising from AHF is suspected, biopsy should be considered to avoid unnecessary surgery.

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          Most cited references12

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          Adrenal masses: characterization with combined unenhanced and delayed enhanced CT.

          To assess the accuracy of a dedicated adrenal computed tomographic (CT) protocol. One hundred sixty-six adrenal masses were evaluated with a protocol consisting of unenhanced CT, and, for those with attenuation values greater than 10 HU, contrast material-enhanced and delayed enhanced CT. Attenuation values and enhancement washout calculations were obtained. An adenoma was diagnosed if a mass had an attenuation value of 10 HU or less at unenhanced CT or a percentage enhancement washout value of 60% or higher. The final diagnosis was adenoma in 127 masses and non-adenoma in 39. Masses measuring more than 10 HU on unenhanced CT scans were confirmed at biopsy (n = 28) or were examined for stability or change in size at follow-up CT performed at a minimum interval of 6 months (n = 33). Thirty-six (92%) of 39 non-adenomas and 124 (98%) of 127 adenomas were correctly characterized. The sensitivity and specificity of this protocol were 98% and 92%, respectively. This protocol correctly characterized 160 (96%) of 166 masses. With a combination of unenhanced and delayed enhanced CT, nearly all adrenal masses can be correctly categorized as adenomas or non-adenomas.
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            Characterization of adrenal masses using unenhanced CT: an analysis of the CT literature.

            Unenhanced CT scanning can reliably characterize incidentally detected adrenal masses when observers use density measurements of the adrenal gland. However, controversy exists as to the optimal density threshold required to differentiate benign from malignant lesions. This study attempts to establish a consensus by performing a pooled analysis of data found in the CT literature. Ten CT reports were analyzed, from which individual adrenal lesion density measurements were obtained for 495 adrenal lesions (272 benign lesions and 223 malignant lesions). Threshold analysis generated a range of sensitivities and specificities for lesion characterization at different density thresholds. Sensitivity for characterizing a lesion as benign ranged from 47% at a threshold of 2 H to 88% at a threshold of 20 H. Similarly, specificity varied from 100% at a threshold of 2 H to 84% at a threshold of 20 H. The attempt to be absolutely certain that an adrenal lesion is benign may lead to an unacceptably low sensitivity for lesion characterization. The threshold chosen will depend on the patient population and the cost-benefit approach to patient care.
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              Clinical utility of noncontrast computed tomography attenuation value (hounsfield units) to differentiate adrenal adenomas/hyperplasias from nonadenomas: Cleveland Clinic experience.

              Radiological characterization of an adrenal tumor as adenoma may decrease the need for follow-up imaging studies, biopsies, and unnecessary adrenalectomies. We retrospectively reviewed 299 adrenalectomies in 290 patients at Cleveland Clinic Foundation over a recent 5-yr period to assess the value of noncontrast Hounsfield units (HU) in characterizing whether an adrenal mass is adenoma or nonadenoma. The mean (+/- SD) HU value for the adrenocortical adenoma/hyperplasia group was 16.2 +/- 13.6 and significantly lower (P < 0.0001) than primary adrenocortical cancers (36.9 +/- 4.1), metastases (39.2 +/- 15.2), and pheochromocytomas (38.6 +/- 8.2). The sensitivity and specificity for 10- and 20-HU cutoff values to differentiate adenomas/hyperplasias from nonadenomas were 40.5 and 100% and 58.2 and 96.9%, respectively. The size of the adrenal tumor had less value with only 40.7 and 81.3% sensitivity and 94.7 and 61.4% specificity for 2- and 4-cm cutoff values. A combination of less than or equal to 4-cm adrenal mass size and noncontrast computed tomography HU less than or equal to 20 had 42.1% sensitivity and 100% specificity. Our study, the largest with surgical histopathology as the gold standard for diagnosis, supports a noncontrast computed tomography attenuation value of 10 HU as a safe cutoff value to differentiate adrenal adenomas/hyperplasias from nonadenomas.
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                Author and article information

                Journal
                Medicine (Baltimore)
                Medicine (Baltimore)
                MEDI
                Medicine
                Wolters Kluwer Health
                0025-7974
                1536-5964
                June 2019
                07 June 2019
                : 98
                : 23
                : e15901
                Affiliations
                [a ]Department of Radiology, Chosun University Hospital, Chosun University College of Medicine, Gwangju, Republic of Korea
                [b ]Department of Internal Medicine, Chosun University Hospital, Chosun University College of Medicine, Gwangju, Republic of Korea
                [c ]Department of Otolaryngology-Head and Neck Surgery, Chosun University Hospital, Chosun University College of Medicine, Gwangju, Republic of Korea
                [d ]Impedance Imaging Research Center, Kyung Hee University, Seoul, Republic of Korea
                [e ]Department of Pathology, Chonnam National University Hwasun Hospital, Chonnam National University Medical School, Jeollanam-do, Republic of Korea
                [f ]Department of Hepato-Biliary-Pancreas Surgery, Chonnam National University Hwasun Hospital, Chonnam National University Medical School, Jeollanam-do, Republic of Korea.
                Author notes
                []Correspondence: Young Hoe Hur, Department of Hepato-Biliary-Pancreas Surgery, Chonnam National University Hwasun Hospital, Chonnam National University Medical School, #322 Seoyang-ro, Hwasun-eup, Hwasun-gun, Jeollanam-do 58128, Republic of Korea (e-mail: surgihur@ 123456naver.com ).
                Article
                MD-D-19-00086 15901
                10.1097/MD.0000000000015901
                6571242
                31169702
                a9a32162-c862-4f02-b8b7-19f2e008197b
                Copyright © 2019 the Author(s). Published by Wolters Kluwer Health, Inc.

                This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0

                History
                : 8 January 2019
                : 19 April 2019
                : 9 May 2019
                Categories
                4500
                Research Article
                Clinical Case Report
                Custom metadata
                TRUE

                adrenohepatic fusion,biopsy,computed tomography,intrahepatic adrenocortical adenoma,magnetic resonance imaging

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