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DOI:10.2214/AJR.05.0830
AJR 2007; 188:1615-1617
© American Roentgen Ray Society

Case Report

Primary Hepatic Angiosarcoma on Coregistered FDG PET and CT Images

Tetsuo Maeda1, Ukihide Tateishi1, Tadashi Hasegawa2, Hidenori Ojima3, Yasuaki Arai1 and Kazuro Sugimura4

1 Division of Diagnostic Radiology, National Cancer Center Hospital, 5-1-1, Tsukiji, Chuo-Ku, Tokyo 104-0045, Japan.
2 Department of Clinical Pathology, Sapporo Medical University, School of Medicine, Sapporo, Japan.
3 Division of Pathology, National Cancer Center Research Institute, Tokyo, Japan.
4 Department of Radiology, Kobe University Graduate School of Medicine, Kobe, Japan.

Received May 16, 2005; accepted after revision July 12, 2005.

 
Address correspondence to U. Tateishi.

Keywords: angiosarcoma • CT • FDG PET • liver cancer • oncologic imaging • PET/CT


Introduction
Top
 Introduction
Case Report
 Discussion
 References
 
Angiosarcoma is an uncommon mesenchymal malignant neoplasm of the vascular or lymphatic endothelium, accounting for 2% of all soft-tissue sarcomas [1–4], that can affect any organ but seems to have a predilection for the skin in the head and neck regions [3, 4]. Although primary hepatic angiosarcoma is rare and accounts for only 2% of primary hepatic tumor, it is the most common malignant mesenchymal tumor of the liver [5].

Various appearances of primary hepatic angiosarcoma on CT and on MRI have been noted [3, 5]. However, to the best of our knowledge, the FDG PET findings have not been described yet. Here, we present a case of primary hepatic angiosarcoma and the coregistered FDG PET and CT images that show the affected sites.


Case Report
Top
 Introduction
 Case Report
Discussion
 References
 
A 65-year-old woman with primary biliary cirrhosis underwent a medical checkup, including abdominal CT that revealed multiple hepatic masses, at a local hospital. The results of an initial biopsy led to suspicion of hepatic angiosarcoma. She was referred to our hospital for further examination and treatment.

At presentation to our institution, the patient had no symptoms, and the physical examination findings were unremarkable. There was no prior exposure to vinyl chloride monomer, thorium dioxide (thorotrast), or arsenic. The results of blood chemistry analyses, including {alpha}-fetoprotein, carcinoembryonic antigen, and carbohydrate antigen 19-9, were normal. Autoantibodies and viral hepatitis test results were negative.

Unenhanced CT showed multiple hypoattenuating lesions in the liver. After IV administration of contrast material, arterial phase CT showed those lesions with faint peripheral enhancement (Fig. 1A). On delayed phase images, the hepatic masses appeared to be heterogeneous and hypoattenuating relative to the normal liver parenchyma.


Figure 1
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  		Fig. 1A —Primary hepatic angiosarcoma in 65-year-old woman. Arterial phase CT scan obtained after IV administration of contrast material shows multiple hypoattenuating liver lesions with faint peripheral enhancement (arrows). Liver masses appeared to show heterogeneous enhancement and to be hypoattenuating compared with liver parenchyma on delayed images (not shown).

 
Histologic examination of a biopsy specimen revealed proliferation of dilated cavernous vascular channels with neoplastic cells (Fig. 1B). Tumor cells showed immunoreactivity for vimentin and for vascular markers (i.e., CD31 and factor VIII). On the basis of these histologic findings, the diagnosis of primary hepatic angiosarcoma was confirmed. Additional immunohistochemical staining of MIB-1 revealed marked activity of proliferation in the tumor cells (Fig. 1C). Moreover, expression of glucose transporter-1 (GLUT-1), which is generally present on the membranes of almost all cells and is the main pathway by which glucose enters the cell body, was also observed in the tumor in this case (Fig. 1D).


Figure 2
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  		Fig. 1B —Primary hepatic angiosarcoma in 65-year-old woman. Photomicrograph of resected specimen shows proliferation of dilated cavernous vascular channels with neoplastic cells. (H and E stain, original magnification, x100)

 

Figure 3
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  		Fig. 1C —Primary hepatic angiosarcoma in 65-year-old woman. Photomicrograph of resected specimen shows positivity of tumor cells, suggestive of highly proliferative activity. (MIB-1 immunohistochemical staining, original magnification, x100)

 

Figure 4
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  		Fig. 1D —Primary hepatic angiosarcoma in 65-year-old woman. At immunohistochemical analysis, photomicrograph of resected specimen shows overexpression of glucose transporter-1 in tumor cells. (original magnification, x100)

 
Whole-body PET/CT fusion scanning was performed after administration of 10 mCi (370 MBq) of 18F-FDG for staging purposes to identify metastatic sites other than the hepatic masses identified on CT. Multiple focal areas of increased uptake were seen in the liver, which corresponded to the hepatic masses seen on CT (Fig. 1E). The maximum standardized uptake value (SUV) of the tumor was 4.65. Furthermore, multiple areas of increased uptake were detected in the mesenterium and the left pubis—findings that are suggestive of intraperitoneal dissemination (Fig. 1F) and bone metastasis (Fig. 1G), respectively.


Figure 5
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  		Fig. 1E —Primary hepatic angiosarcoma in 65-year-old woman. Coregistered FDG PET and CT image shows marked FDG accumulation within hepatic tumors.

 

Figure 6
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  		Fig. 1F —Primary hepatic angiosarcoma in 65-year-old woman. Coregistered FDG PET and CT image shows marked FDG accumulation within mesenteric lesion (arrow).

 

Figure 7
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  		Fig. 1G —Primary hepatic angiosarcoma in 65-year-old woman. Coregistered FDG PET and CT image shows marked FDG accumulation within left pubis (arrow).

 
Because of the extent of the disease, surgery was not selected for treatment, and systemic chemotherapy using paclitaxel was performed but led to no regression of the primary and metastatic lesions. The patient is still alive after 2 years.


Discussion
Top
 Introduction
 Case Report
 Discussion
References
 
Angiosarcoma, which usually affects adults and can develop from the endothelium of the lymphatics or blood vessels, is a rare tumor among soft-tissue sarcomas. Hepatic angiosarcoma is the most common primary sarcoma in the liver and can be induced by exposure to vinyl chloride monomer, ingestion of thorium dioxide (thorotrast), or ingestion of arsenic. Strategies for treating this tumor have not been confirmed, and the prognosis of patients with this tumor is poor [5].

In our patient, the hepatic tumors appeared as hypoattenuating masses with heterogeneous enhancement on contrast-enhanced CT. Although these findings are considered to be nonspecific and do not aid in distinguishing the tumors seen on CT from other metastatic hepatic tumors, these radiologic findings were consistent with previous descriptions of primary or metastatic angiosarcoma of the liver [3, 5]. In fact, it is difficult to make a diagnosis of hepatic angiosarcoma preoperatively using only imaging techniques such as CT and MRI. A percutaneous needle biopsy might be a reasonable option, as was performed in our patient.

In some instances, even if liver biopsy results are suspicious for angiosarcoma, determining whether the liver or the other organ is the site of the primary lesion is difficult. In our patient, a diagnosis of primary hepatic angiosarcoma was made because there were no affected organs with findings suggestive of primary soft-tissue angiosarcoma.

Although FDG PET is well known as a functional imaging technique in the evaluation of various malignant tumors, to our knowledge, only one report has previously described PET features of angiosarcoma [6]. To our knowledge, the current case report is the first description of coregistered FDG PET and CT images of primary hepatic angiosarcoma. In our patient, the PET scan showed multiple focal intense accumulations of FDG that were consistent with the hepatic masses detected on CT. In addition, areas of increased uptake of FDG were seen in the left pubis and mesenterium that were considered to be metastases. Most patients who have primary hepatic angiosarcoma have metastatic lesions at the time of presentation [5]. In patients with known extrahepatic lesions, therefore, PET may contribute to confirm the presence or absence of distant metastases at other sites and may assist in the selection of an appropriate treatment.

The reason FDG accumulates in hepatic angiosarcoma is not known. However, in general, the degree of FDG accumulation in various tumors is considered to depend on the activity of glucose transporters, especially GLUT-1, and of hexokinases in the cells based on the findings of experimental and clinical studies [7]. In our patient, overexpression of GLUT-1 was seen. Thus, it can be the reason for the marked FDG accumulations in the hepatic tumors in our patient.

Highly proliferative activity in the resected specimens from our patient was also confirmed immunohistochemically. A positive correlation between the degree of FDG uptake and the proportion of MIB-1–positive cells has been described in previous studies [8]. Active proliferation of tumor cells may be associated with increased accumulation of FDG even in angiosarcoma.

In summary, we herein described the coregistered FDG PET and CT images of primary hepatic angiosarcoma with multiple FDG accumulations in various lesions. Although the correlation between FDG uptake and the overexpression of GLUT-1 in various tumor cells is still under debate, we believe that both the overexpression of GLUT-1 and the active proliferation of tumor cells might be related to FDG uptake in the current tumors.


References
Top
 Introduction
 Case Report
 Discussion
 References
 

  1. Maddox JC, Evans HL. Angiosarcoma of the skin and soft tissue. Cancer 1981; 48:1907 –1921[CrossRef][Medline]
  2. Meis-Kindblom JM, Kindblom LG. Angiosarcoma of soft tissue. Am J Surg Pathol 1998;22 : 683–697[CrossRef][Medline]
  3. Tateishi U, Hasegawa T, Muramatsu Y, Moriyama N. Hepatic metastases of soft tissue angiosarcoma: CT and MR imaging findings. Abdom Imaging 2003; 28:660 –664[CrossRef][Medline]
  4. Naka N, Ohsawa M, Tomita Y, Kanno H, Uchida A, Aozasa K. Angiosarcoma in Japan: a review of 99 cases. Cancer1995; 75:989 –996[CrossRef][Medline]
  5. Koyama T, Fletcher JG, Johnson CD, Kuo MS, Notohara K, Burgart LJ. Primary hepatic angiosarcoma: findings at CT and MR imaging. Radiology 2002;222 : 667–673[Abstract/Free Full Text]
  6. Del Frate C, Mortele K, Zanardi R, et al. Pseudomesotheliomatous angiosarcoma of the chest wall and pleura. J Thorac Imaging 2003; 18:200 –203[CrossRef][Medline]
  7. Koga H, Matsuo Y, Sasaki M, et al. Differential FDG accumulation associated with GLUT-1 expression in a patient with lymphoma. Ann Nucl Med 2003; 17:327 –331[Medline]
  8. Avril N, Menzel M, Dose J, et al. Glucose metabolism of breast cancer assessed by 18F-FDG PET: histologic and immunohistochemical tissue analysis. J Nucl Med 2001;42 : 9–16[Abstract/Free Full Text]

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