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      Hepatic Angiomyolipoma with Minimal Intratumoral Fat Content

      case-report

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          Abstract

          We report a rare case of hepatic angiomyolipoma with minimal fat content. The low fat content led to an incorrect preoperative diagnosis. A 38-year-old man who was a carrier of hepatitis B virus infection incidentally presented with a hepatic tumor. His serum alpha-fetoprotein level was normal. Ultrasonography revealed a well-circumscribed, heterogeneous hypoechoic nonencapsulated liver tumor measuring 34 × 24 mm. Precontrast computed tomography (CT) did not reveal fatty attenuation in the lesion. Contrast-enhanced CT revealed a hypervascular nonencapsulated tumor in the arterial phase and moderate washing out of the contrast medium in the portal phase. A hypervascular tumor was observed on CT hepatic arteriography, and complete washing out of the contrast medium on CT during arterial portography. These findings are compatible with hepatocellular carcinoma. The tumor exhibited low signal intensity on T1-weighted images and high signal intensity on T2-weighted images; no hypointensity was observed on fat suppression images. The patient underwent left hemihepatectomy because of a preoperative diagnosis of hepatocellular carcinoma. The histopathological diagnosis was a hepatic angiomyolipoma with 5% fat content. Low fat content makes the diagnosis of this condition difficult. The absence of serum tumor markers and the presence of a nonencapsulated hypervascular tumor may facilitate the accurate preoperative diagnosis of hepatic angiomyolipomas that have a low fat content and mimic hepatocellular carcinoma.

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          Most cited references16

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          Hepatic angiomyolipoma: a clinicopathologic study of 30 cases and delineation of unusual morphologic variants.

          Hepatic angiomyolipoma (AML) is frequently misdiagnosed. HMB-45 is a promising immunomarker for this tumor that leads to recognition of some AMLs with unusual morphology. The purpose of this collaborative study is to better define the morphologic variations of AML. Thirty AMLs were examined, including four biopsy specimens and two fine-needle aspirates. The diagnosis was confirmed by the presence of HMB-45-positive myoid cells. Almost half the cases were originally misdiagnosed as carcinomas or sarcomas. There was marked female predominance (25:5), and the mean age was 48.7 years (range 29-68). Three patients (10%) had evidence of tuberous sclerosis and all had renal AML. According to the line of differentiation and predominance of tissue components, the tumors was subcategorized into mixed, lipomatous (> or = 70% fat), myomatous (< or = 10% fat), and angiomatous type. The mixed type was the most common (11 resected cases), comprising sheets of epithelioid muscle cells admixed with islands of adipocytes, abnormal vessels, and frequently, hematopoietic cells. Six tumors (including three from biopsy specimens) were heavily fatty and showed predominantly adipocytes with epithelioid and short spindle myoid cells webbed between fat cells. Of 10 myomatous AMLs, five tumors showed a pure sinusoidal trabecular pattern and comprised mainly epithelioid cells. Typically, mature adipocytes were absent or scanty, but fat was seen as fine droplets within cytoplasm or as occasional large globules in sinusoids. Pelioid and inflammatory pseudotumor-like patterns were identified focally. Regarding cellular features of the myoid cells, most of the epithelioid cells were either eosinophilic or clear with spiderweb cell morphology. Three AMLs showed an almost purely oncocytic appearance with scanty fat. Large pleomorphic epithelioid cells existed as small foci. Spindle cells arranged in long fascicles were uncommon. D-PAS-positive globules were common around pelioid areas. Brown pigments with staining characteristics of hemosiderin and/or melanin were noted. In conclusion, we propose HMB-45-positive myoid cells as the defining criterion of hepatic AML, which is a tumor capable of dual myomatous and lipomatous differentiation and melanogenesis. Because of its protean morphologic appearance, recognition of the various variant patterns and cell types is important for a correct diagnosis, assisted by immunohistochemical confirmation with HMB-45. Trabecular and oncocytic cell tumors appear to stand out as distinctive subtypes.
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            Management of Hepatic Angiomyolipoma

            Preoperative diagnosis of hepatic angiomyolipoma is difficult, and the treatment for it remains controversial. The aim of this study is to review our experience in the treatment of hepatic angiomyolipoma and to propose a treatment strategy for this disease. We retrospectively collected the clinical, imaging, and pathological features of patients with hepatic angiomyolipoma. Immunohistochemical studies with antibodies for HMB-45, actin, S-100, cytokeratin, vimentin, and c-kit were performed. Treatment experience and long-term follow-up results are summarized. During a period of 9 years, 10 patients with hepatic angiomyolipoma were treated at our hospital. There was marked female predominance (nine patients). Nine patients received surgical resection without complications. One patient received nonoperative management with biopsy and follow-up. One patient died 11 months after surgery because of recurrent disease. We propose all symptomatic patients should receive surgical resection for hepatic angiomyolipoma. Conservative management with close follow-up is suggested in patients with asymptomatic tumors and meet the following criteria: (1) tumor size smaller than 5 cm, (2) angiomyolipoma proved through fine needle aspiration biopsy, (3) patients with good compliance, and (4) not a hepatitis virus carrier.
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              Angiomyolipoma of the liver: a collective review.

              This review summarizes the clinical and pathological findings of 52 cases of hepatic angiomyolipoma to discern and establish the most pertinent clinical and pathologic characteristics of the tumor. The disease was symptomatic in 60% of the patients. Abdominal pain or distress was the most common symptom, appearing in 37% of the patients, followed by malaise and upper abdominal mass or hepatomegaly. Of the 52 patients, only three (5.8%) showed associated tuberous sclerosis. Antemortem diagnosis of the tumor has been made with increasing frequency with the recent advent of computed tomography (CT) and ultrasound (US). The tumor was usually visualized as a hyperechoic mass by US imaging and as a low density mass less than -20 Housefield units by CT, and was hypervascular on angiography. The tumor was usually yellow to light tan, depending on the amount of fat tissue. Histologically, the tumor was characterized by an admixture of mature fat cells, blood vessels, and smooth muscle cells, with occasional foci of extramedullary hematopoiesis. The amount of smooth muscle component varied and often exhibited hypercellularity, pleomorphism with occasional bizarre giant cells, and moderate motitic activity. These features are considered conducive to an erroneous diagnosis of malignant tumor. However, since no malignant counterpart has been reported, it can easily be accurately differentiated histologically, if one is aware of the entity and can identify the three components of the tumor; blood vessels, smooth muscle cells, and fat. With regard to the histogenesis of angiomyolipoma, primitive mesenchymal cells around blood vessels may be the precursor cells.
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                Author and article information

                Journal
                Case Rep Gastroenterol
                CRG
                Case Reports in Gastroenterology
                S. Karger AG (Allschwilerstrasse 10, P.O. Box · Postfach · Case postale, CH–4009, Basel, Switzerland · Schweiz · Suisse, Phone: +41 61 306 11 11, Fax: +41 61 306 12 34, karger@karger.ch )
                1662-0631
                Sep-Dec 2009
                20 November 2009
                20 November 2009
                : 3
                : 3
                : 324-331
                Affiliations
                [1] aDepartment of Surgery, Division of Frontier Medical Science, Programs for Biomedical Research, Graduate School of Biomedical Sciences, Hiroshima University, Hiroshima, Japan
                [2] bDepartment of Anatomical Pathology, Hiroshima University, Hiroshima, Japan
                Author notes
                *Masataka Banshodani, MD, Department of Surgery, Division of Frontier Medical SciencePrograms for Biomedical Research, Graduate School of Biomedical Sciences Hiroshima University, 1-2-3 Kasumi, Minami-ku, Hiroshima 734-8551 (Japan), Tel. +81 82 257 5222, Fax +81 82 257 5224, E-Mail mban1127@ 123456yahoo.co.jp
                Article
                crg0003-0324
                10.1159/000255355
                2988925
                21103249
                4a65c8ff-9ed4-40a0-b739-d263c1a5a358
                Copyright © 2009 by S. Karger AG, Basel

                This is an Open Access article distributed under the terms of the Creative Commons Attribution-Noncommercial-No-Derivative-Works License ( http://creativecommons.org/licenses/by-nc-nd/3.0/). Users may download, print and share this work on the Internet for noncommercial purposes only, provided the original work is properly cited, and a link to the original work on http://www.karger.com and the terms of this license are included in any shared versions.

                History
                Page count
                Figures: 3, References: 15, Pages: 8
                Categories
                Published: November 2009

                Gastroenterology & Hepatology
                angiomyolipoma,liver tumor,homatropine methylbromide-45
                Gastroenterology & Hepatology
                angiomyolipoma, liver tumor, homatropine methylbromide-45

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