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      A case of multiple hepatic angiomyolipomas with high 18 F-fluorodeoxyglucose uptake

      case-report

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          Abstract

          Background

          Hepatic angiomyolipoma is a rare benign mesenchymal tumor. We report an unusual case of a patient with multiple hepatic angiomyolipomas exhibiting high 18 F-fluorodeoxyglucose (FDG) uptake.

          Case presentation

          A 29-year-old man with a medical history of tuberous sclerosis was admitted to our hospital for fever, vomiting, and weight loss. Abdominal dynamic computed tomography revealed faint hypervascular hepatic tumors in segments 5 (67 mm) and 6 (10 mm), with rapid washout and clear borders; however, the tumors exhibited no definite fatty density. Abdominal magnetic resonance imaging revealed that the hepatic lesions were slightly hypointense on T1-weighted imaging, slightly hyperintense on T2-weighted imaging, and hyperintense with no apparent fat component on diffusion-weighted imaging. FDG-positron emission tomography (PET) imaging revealed high maximum standardized uptake values (SUVmax) of 6.27 (Segment 5) and 3.22 (Segment 6) in the hepatic tumors. A right hepatic lobectomy was performed, and part of the middle hepatic vein was also excised. Histological examination revealed that these tumors were characterized by the background infiltration of numerous inflammatory cells, including spindle-shaped cells, and a resemblance to an inflammatory pseudotumor. Immunohistochemical evaluation revealed that the tumor stained positively for human melanoma black-45. The tumor was therefore considered an inflammatory pseudotumor-like angiomyolipoma. Although several case reports of hepatic angiomyolipoma have been described or reviewed in the literature, only 3 have exhibited high 18 F-FDG uptake on PET imaging with SUVmax ranging from 3.3–4.0. In this case, increased 18 F-FDG uptake is more likely to appear, particularly if the inflammation is predominant.

          Conclusion

          Although literature regarding the role of 18 F-FDG-PET in hepatic angiomyolipoma diagnosis is limited and the diagnostic value of 18 F-FDG-PET has not yet been clearly defined, the possibility that hepatic angiomyolipoma might exhibit high 18 F-FDG uptake should be considered.

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          Most cited references19

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          Fat-containing lesions of the liver: radiologic-pathologic correlation.

          Fat-containing tumors of the liver are a heterogeneous group of tumors with characteristic histologic features, variable biologic profiles, and variable imaging findings. Benign liver lesions that contain fat include focal or geographic fatty change (steatosis), pseudolesions due to postoperative packing material (omentum), adenoma, focal nodular hyperplasia, lipoma, angiomyolipoma, cystic teratoma, hepatic adrenal rest tumor, pseudolipoma of the Glisson capsule, and xanthomatous lesions in Langerhans cell histiocytosis. Malignant liver lesions that can contain fat include hepatocellular carcinoma, primary and metastatic liposarcoma, and hepatic metastases. Identification of fat within a liver lesion can be critical in characterization of the lesion. The imaging characteristics of a lesion coupled with the pattern of intratumoral fatty change are helpful in narrowing the differential diagnosis. Although the presence of fat can be demonstrated with computed tomography or ultrasound, magnetic resonance imaging is the most specific imaging technique for demonstration of both microscopic and macroscopic fat. (c) RSNA, 2005.
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            Renal epithelioid angiomyolipoma with atypia: a series of 40 cases with emphasis on clinicopathologic prognostic indicators of malignancy.

            As epithelioid cellular morphology can be seen in clinically benign usual angiomyolipomas (AMLs), we divide epithelioid AMLs into those without and with atypia, the latter category associated in the literature with malignant potential. We herein report the histologic spectrum and biologic behavior of 40 consecutive cases of epithelioid AML with atypia and assess whether cases can be stratified prognostically based on clinical and pathologic features. Atypical epithelioid cells were defined as atypical polygonal cells with abundant cytoplasm, vesicular nuclei, prominent nucleoli, and nuclear size that exceeds x2 the size of adjacent nuclei. The degree of atypia was divided to moderate and severe. Cases with bland epithelioid cells with minimal variation in nuclear size were not included. Mean age was 50.5 years (range 17 to 81), and the female to male ratio was 1.6:1. Average tumor size was 7.2 cm (range 1.0 to 17.7). The percentage of epithelioid component ranged from 5%-100% (mean 68%). Of the epithelioid component, the percentage of cells exhibiting nuclear atypia ranged in individual cases from 5% to 100% (mean of 58.4% atypical cells); 26/40 (65%) cases showed severe nuclear atypia. Cells displaying severe nuclear atypia were typically of large size with abundant cytoplasm, compared with those with moderate atypia being of small to intermediate in size with scant to moderate amount of cytoplasm. Neoplastic multinucleated giant cells and necrosis was present in 22 cases (55%) and 15 cases (37.5%), respectively. Mitoses were identified in 72.5% (29/40) of cases and ranged from 1 to 6 per 10 hpf with 7 cases showing atypical mitotic figures. Lymphovascular invasion or renal vein invasion was present in 3 cases each. Hilar and perinephric fat involvement was present in 5 and 6 cases, respectively. Clinical follow-up was available in 34 out of the 40 cases. Of the 34 cases, 9 (26%) were malignant and showed local recurrence or distant metastases. Of the 9 patients with malignant tumors, 4 died of the disease at 6, 12, 15, and 34 months after the original diagnosis was rendered, and 4 were alive with disease (mean follow-up period of 52 mo, range 24 to 72 mo). Twenty-four patients showed no evidence of recurrence and/or metastases with a mean follow-up period of 34 months (range 1 to 156 mo). We compared the 21 cases of atypical epithelioid AMLs that exhibited a benign clinical course with a minimum follow-up period of 6 months postsurgery to the 9 cases with malignant behavior. All of these were more frequently observed in clinically malignant cases: older age, larger tumor size, higher percentage of epithelioid component, severe atypia, higher percentage of atypical cells, higher mitotic count, atypical mitotic figures, necrosis, lymphovascular invasion, and renal vein invasion. Using these features, we developed a predictive model of 4 atypical features that included: (1) > or =70% atypical epithelioid cells, (2) > or =2 mitotic figures per 10 hpf, (3) atypical mitotic figures, and (4) necrosis; the presence of 3 or all of the features was highly predictive of malignant behavior. This model accurately categorized 78% of clinically malignant and 100% of the clinically benign epithelioid AMLs with atypia.
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              Malignant angiomyolipoma of the liver: a hitherto unreported variant.

              After their original recognition in the kidney, angiomyolipomas (AMLs) have been reported in the liver for more than 20 years. In the kidney, five cases of malignant AML have been reported. We report the first case of malignant hepatic AML. A 70-year-old female patient presented with abdominal discomfort. Clinical examination revealed a palpable liver. CT scan showed a polymorphous hypervascular lesion in the right liver lobe. A biopsy was taken and resulted initially in a differential diagnosis between a hepatocellular carcinoma, a metastatic tumour (possibly of renal origin) and angiomyolipoma (AML). After immunohistochemistry, a hepatic AML was suggested, given the immunoreactivity for HMB45/NKIC-3. The mass was resected 5 years later because of relapsing abscess formation. Gross examination of the resection specimen showed a focally encapsulated brown mass with focal necrosis. Microscopic examination showed a tumour growing in sheets, separated by sinusoidal-like vessels. Most of the tumour cells had a large, polygonal, clear cytoplasm, often with eosinophilic condensation around the nucleus. There was prominent vascular invasion. Immunohistochemistry (reactivity for HMB-45, NKIC-3, S100 and alpha smooth muscle actin, negativity for cytokeratin and vimentin) and electron microscopy confirmed the diagnosis of monomorphic epithelioid AML with prominent vascular invasion. Seven months after tumour resection, the patient died of recurrent disease. This case highlights the importance of immunohistochemistry and electron microscopy in diagnosing this type of tumour. Possibly, in the past, malignant AML of the liver has been misdiagnosed as HCC.
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                Author and article information

                Contributors
                Journal
                BMC Med Imaging
                BMC Med Imaging
                BMC Medical Imaging
                BioMed Central
                1471-2342
                2014
                20 May 2014
                : 14
                : 17
                Affiliations
                [1 ]Department of Diagnostic Radiology and Nuclear Medicine, Gunma University Graduate School of Medicine, Showa-machi, Maebashi 3-39-22 Gunma, Japan
                Article
                1471-2342-14-17
                10.1186/1471-2342-14-17
                4036299
                24885757
                0b0f89a4-4294-46fe-8de0-d6689ab750e6
                Copyright © 2014 Kumasaka et al.; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 20 January 2014
                : 14 May 2014
                Categories
                Case Report

                Radiology & Imaging
                hepatic angiomyolipoma,fdg,pet,hmb-45
                Radiology & Imaging
                hepatic angiomyolipoma, fdg, pet, hmb-45

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