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      A hepatic sclerosed hemangioma with significant morphological change over a period of 10 years: a case report

      case-report

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          Abstract

          Introduction

          Liver cavernous hemangioma is the most common noncystic hepatic lesion, and a hemangioma that undergoes degeneration and fibrous replacement is called a hepatic sclerosed hemangioma.

          Case presentation

          A 63-year-old Japanese man was admitted for detailed investigation of a liver tumor. Tumor markers carcinoembryonic antigen, alpha-fetoprotein, and CA19-9 levels in the peripheral blood were not elevated at any time. Plain computed tomography showed an approximately 1.5cm low density mass in the periphery of segment 8, which was marginally enhanced on contrast-enhanced dynamic computed tomography. On magnetic resonance imaging, the tumor was hypointense on T1-weighted image and hyperintense on T2-weighted image. The tumor was suspected to be an atypical hemangioma, metastatic, hepatocellular carcinoma, or cholangiocellular carcinoma. Segmental hepatectomy was performed. Histological examination of the resected tumor specimen revealed a sclerosed hemangioma with marked hyalinization and sparse stromal fibrosis. Immunochemically, the tumor cells were positive for CD34 and alpha smooth muscle actin. Electron microscopically, the residual hemangioma consisted of numerous caveolae and vesicles in endothelial cells in irregular shapes and sizes. Immunostaining for caveolin-1 showed decreased or no caveolin-1 reactivity in the hyalinized lesions of the sclerosed hemangioma, but abundant caveolin-1 reactivity in the residual cavernous hemangioma. Of interest, computed tomography images of the tumor obtained 10 years earlier at our hospital depicted a 3cm typical cavernous hemangioma.

          Conclusions

          Hepatic sclerosed hemangioma is a rare condition. Comparison of radiological findings of the lesion over a period of 10 years was valuable in providing insight for the evolutional process from liver cavernous hemangioma to hepatic sclerosed hemangioma.

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          Most cited references22

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          Caveolin-1 in tumor progression: the good, the bad and the ugly.

          Caveolin-1 (Cav1) is a multifunctional scaffolding protein with multiple binding partners that is associated with cell surface caveolae and the regulation of lipid raft domains. Cav1 regulates multiple cancer-associated processes including cellular transformation, tumor growth, cell migration and metastasis, cell death and survival, multidrug resistance and angiogenesis. However, Cav1 has been reported to impact both positively and negatively on various aspects of tumor progression and while reported to function as a tumor suppressor, it has also been identified as a poor prognostic factor in various human cancers. In this review, we survey the functional roles of Cav1 in cancer and argue that Cav1 function is interdependent on tumor stage and the expression of molecular effectors that impact on its role during tumor progression.
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            Direct evidence for the role of caveolin-1 and caveolae in mechanotransduction and remodeling of blood vessels.

            Caveolae in endothelial cells have been implicated as plasma membrane microdomains that sense or transduce hemodynamic changes into biochemical signals that regulate vascular function. Therefore we compared long- and short-term flow-mediated mechanotransduction in vessels from WT mice, caveolin-1 knockout (Cav-1 KO) mice, and Cav-1 KO mice reconstituted with a transgene expressing Cav-1 specifically in endothelial cells (Cav-1 RC mice). Arterial remodeling during chronic changes in flow and shear stress were initially examined in these mice. Ligation of the left external carotid for 14 days to lower blood flow in the common carotid artery reduced the lumen diameter of carotid arteries from WT and Cav-1 RC mice. In Cav-1 KO mice, the decrease in blood flow did not reduce the lumen diameter but paradoxically increased wall thickness and cellular proliferation. In addition, in isolated pressurized carotid arteries, flow-mediated dilation was markedly reduced in Cav-1 KO arteries compared with those of WT mice. This impairment in response to flow was rescued by reconstituting Cav-1 into the endothelium. In conclusion, these results showed that endothelial Cav-1 and caveolae are necessary for both rapid and long-term mechanotransduction in intact blood vessels.
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              Caveolae: mining little caves for new cancer targets.

              Caveolae exist at cell surfaces as caveolin-coated invaginations that perform transport and signalling functions influencing cell growth, apoptosis, angiogenesis and transvascular exchange. Caveolin could constitute a key switch in tumour development through its function as a tumour suppressor and as a promoter of metastasis, chemoresistance and survival. Targeting of drugs and gene vectors to tissue-specific proteins in caveolae allows selective delivery into vascular endothelial cells in vivo and might even improve direct access to solid-tumour cells. Therefore, caveolae seem to be rich in potential targets for cancer imaging and therapeutics.
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                Author and article information

                Contributors
                Journal
                J Med Case Rep
                J Med Case Rep
                Journal of Medical Case Reports
                BioMed Central
                1752-1947
                2013
                28 May 2013
                : 7
                : 139
                Affiliations
                [1 ]Department of Internal Medicine, Kitasato University Medical Center, 6-100 Arai, Kitamoto-shi, Saitama 364-8501, Japan
                [2 ]Department of Surgery, Kitasato University Medical Center, Saitama, Japan
                [3 ]Department of Radiology, Kitasato University Medical Center, Saitama, Japan
                [4 ]Department of Pathology, Kitasato University Medical Center, Saitama, Japan
                [5 ]Organized Center of Clinical Medicine, International University of Health and Welfare, Tokyo, Japan
                Article
                1752-1947-7-139
                10.1186/1752-1947-7-139
                3750244
                23714181
                528d24d7-0197-44a5-b1f0-c7bcde073ce7
                Copyright © 2013 Shimada 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 cited.

                History
                : 5 December 2012
                : 25 March 2013
                Categories
                Case Report

                Medicine
                caveolae,caveolin-1,electron microscopy,hepatic sclerosed hemangioma,immunohistochemistry,liver cavernous hemangioma

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