A Case of Inflammatory Hepatocellular Adenoma Complicated by Steatohepatitis

Abstract

A 54-year-old man with hypertension, dyslipidemia, diabetes mellitus and fatty liver was referred to our institution for accurate diagnosis of a 4-cm space-occupying lesion in subsegment 6 of the liver, identified on ultrasonography as an iso-to hypointense lesion with clear margins. Dynamic computed tomography disclosed light accumulation of contrast medium at the margins of the space-occupying lesion from the arterial phase through to the venous phase. Sonazoid-enhanced ultrasonography showed staining of the tumor margins in the vascular phase, disappearing in the post-vascular phase. Hepatic arteriography identified the space-occupying lesion as hypervascular. Since accurate diagnosis by imaging was difficult, tumor resection was performed to exclude hepatocellular carcinoma, after obtaining consent from the patient. Histological examination revealed steatohepatitis and sinusoidal dilatation, representing socalled peliosis hepatis, in the surroundings, while the tumor showed mild hepatocellular atypia. On immunohistochemistry, tumor cells were positive for liver fatty acid-binding protein, glutamine synthetase and serum amyloid A, and negative for β-catenin and glypican 3. Glutamine synthetase, in particular, showed strong diffuse staining. Inflammatory hepatocellular adenoma was thus considered the most likely diagnosis. The pathogenesis and outcome of inflammatory hepatocellular adenoma are not fully understood, and this case with concomitant steatohepatitis was considered worth reporting.

Share and Cite:

Harima, Y. , Urayama, N. , Hisano, F. , Matsuzaki, Y. , Sanuki, K. , Kuwahara, T. , Kaneko, T. , Harada, T. , Kawaoka, T. , Hiraki, S. , Fukuda, S. , Okita, K. and Nakashima, O. (2014) A Case of Inflammatory Hepatocellular Adenoma Complicated by Steatohepatitis. Open Journal of Gastroenterology, 4, 121-129. doi: 10.4236/ojgas.2014.43019.

Conflicts of Interest

The authors declare no conflicts of interest.

References

[1] Rooks, J.B., Ory, H.W., Ishak, K.G., et al. (1979) Epidemiology of Hepatocellular Adenoma. The Role of Oral Contraceptive Use. The Journal of the American Medical Association, 242, 644-648.
http://dx.doi.org/10.1001/jama.1979.03300070040020
[2] Bioulac-Sage, P., Balabaud, C. and Wanless, I. (2010) Focal Nodular Hyperplasia and Hepatocellular Adenoma. In: Bosman, F., Ed., WHO Classification of Tumours of the Digestive System, 4th Edition, IRAC, Lyon, 198-204.
[3] Bioulac-Sage, P., Laumonier, H., Couchy, G., et al. (2009) Hepatocellular Adenoma Management and Phenotypic Classification: The Bordeaux Experience. Hepatology, 50, 481-489.
http://dx.doi.org/10.1002/hep.22995
[4] Farges, O., Ferreira, N., Dokmak, S., et al. (2011) Changing Trends in Malignant Transformation of Hepatocellular Adenoma. Gut, 60, 85-89.
http://dx.doi.org/10.1136/gut.2010.222109
[5] Sasaki, M., Yoneda, N., Kitamura, S., et al. (2011) Characterization of Hepatocellular Adenoma Based on the Phenotypic Classification: The Kanazawa Experience. Hepatology Research, 41, 982-988.
http://dx.doi.org/10.1111/j.1872-034X.2011.00851.x
[6] Konishi, M., Ryu, M., Kinoshita, T., et al. (1995) A Case Report of Liver Cell Adenoma. Acta Hepatologica Japonica, 36, 223-229.
http://dx.doi.org/10.2957/kanzo.36.223
[7] Lin, H., van den Esschert, J., Liu, C., et al. (2011) Systematic Review of Hepatocellular Adenoma in China and Other Regions. Journal of Gastroenterology and Hepatology, 26, 28-35.
http://dx.doi.org/10.1111/j.1440-1746.2010.06502.x
[8] Paradis, V., Champault, A., Ronot, M., et al. (2007) Telangiectatic Adenoma: An Entity Associated with Increased Body Mass Index and Inflammation. Hepatology, 46, 140-146.
http://dx.doi.org/10.1002/hep.21684
[9] Ascha, M.S., Hanouneh, I.A., Lopez, R., et al. (2010) The Incidence and Risk Factors of Hepatocellular Carcinoma in Patients with Nonalcoholic Steatohepatitis. Hepatology, 51, 1972-1978.
http://dx.doi.org/10.1002/hep.23527
[10] Sanyal, A.J., Banas, C., Sargeant, C., et al. (2006) Similarities and Differences in Outcomes of Cirrhosis Due to Nonalcoholic Steatohepatitis and Hepatitis C. Hepatology, 43, 682-689.
http://dx.doi.org/10.1002/hep.21103
[11] Hashimoto, E., Yatsuji, S., Tobari, M., et al. (2009) Hepatocellular Carcinoma in Patients with Non-Alcoholic Steatohepatitis. Journal of Gastroenterology, 44, 89-95.
http://dx.doi.org/10.1007/s00535-008-2262-x
[12] Adams, L.A., Lymp, J.F., St Sauver, J., et al. (2005) The Natural History of Nonalcoholic Fatty Liver Disease: A Population-Based Cohort Study. Gastroenterology, 129, 113-121.
http://dx.doi.org/10.1053/j.gastro.2005.04.014
[13] Ong, J.P., Pitts, A. and Younossi, Z.M. (2008) Increased Overall Mortality and Liver-Related Mortality in Nonalcoholic Fatty Liver Disease. Journal of Hepatology, 49, 608-612.
http://dx.doi.org/10.1016/j.jhep.2008.06.018
[14] Rafiq, N., Bai, C., Fang, Y., et al. (2009) Long-Term Follow-Up of Patients with Nonalcoholic Fatty Liver. Clinical Gastroenterology and Hepatology, 7, 234-238.
http://dx.doi.org/10.1016/j.cgh.2008.11.005
[15] Kawamura, Y., Arase, Y., Ikeda, K., et al. (2012) Large-Scale Long-Term Follow-Up Study of Japanese Patients with Non-Alcoholic Fatty Liver Disease for the Onset of Hepato-cellular Carcinoma. The American Journal of Gastroenterology, 107, 253-261.
http://dx.doi.org/10.1038/ajg.2011.327
[16] Van Aalten, S.M., Verheij, J., Terkivatan, T., et al. (2011) Validation of a Liver Adenoma Classification System in a Tertiary Referral Centre: Implications for Clinical Practice. Journal of Hepatology, 55, 120-125.
http://dx.doi.org/10.1016/j.jhep.2010.10.030
[17] Zucman-Rossi, J., Jeannot, E., Nhieu, J.T., et al. (2006) Genotype-Phenotype Correlation in Hepatocellular Adenoma: New Classification and Relationship with HCC. Hepatology, 43, 515-524.
http://dx.doi.org/10.1002/hep.21068
[18] Bioulac-Sage, P., Rebouissou, S., Thomas, C., et al. (2007) Hepatocellular Adenoma Subtype Classification Using Molecular Markers and Immunohistochemistry. Hepatology, 46, 740-748.
http://dx.doi.org/10.1002/hep.21743
[19] de La Coste, A., Romagnolo, B., Billuart, P., et al. (1998) Somatic Mutations of the Beta-Catenin Gene Are Frequent in Mouse and Human Hepatocellular Carcinomas. Proceedings of the National Academy of Sciences of the United States of America, 95, 8847-8851.
http://dx.doi.org/10.1073/pnas.95.15.8847
[20] Miyoshi, Y., Iwao, K., Nagasawa, Y., et al. (1998) Activation of the Beta-Catenin Gene in Primary Hepatocellular Carcinomas by Somatic Alterations Involving Exon 3. Cancer Research, 95, 2524-2527.
[21] Nhieu, J.T., Renard, C.A., Wei, Y., et al. (1999) Nuclear Accumulation of Mutated Beta-Catenin in Hepatocellular Carcinoma Is Associated with Increased Cell Proliferation. The American Journal of Pathology, 155, 703-710.
http://dx.doi.org/10.1016/S0002-9440(10)65168-1
[22] Fulcher, A.S. and Sterling, R.K. (2002) Hepatic Neoplasms: Computed Tomography and Magnetic Resonance Features. Journal of Clinical Gastroenterol, 34, 463-471.
http://dx.doi.org/10.1097/00004836-200204000-00019
[23] Hussain, S.M., van den Bos, I.C., Dwarkasing, R.S., et al. (2006) Hepatocellular Adenoma: Findings at State-of-the-Art Magnetic Resonance Imaging, Ultrasound, Computed Tomography and Pathologic Analysis. European Radiology, 16, 1873-1886.
http://dx.doi.org/10.1007/s00330-006-0292-4
[24] Ronot, M., Bahrami, S., Calderaro, J., et al. (2011) Hepatocellular Adenomas: Accuracy of Magnetic Resonance Imaging and Liver Biopsy in Subtype Classification. Hepatology, 53, 1182-1191.
http://dx.doi.org/10.1002/hep.24147
[25] Katabathina, V.S., Menias, C.O., Shanbhogue A.K.P., et al. (2011) Genetics and Imaging of Hepatocellular Adenomas: 2011 Update. Radiographics, 31, 1529-1543.
[26] Watkins, J., Balabaud, C., Bioulac-Sage, P., et al. (2009) Hepatocellular Adenoma in Advanced-Stage Fatty Liver Disease. European Journal of Gastroenterology and Hepatology, 21, 932-936.
http://dx.doi.org/10.1097/MEG.0b013e328311a67c
[27] Brunt, E.M., Wolverson, M.K. and Di Bisceglie, A.M. (2007) Benign Hepatocellular Tumors (Adenomatosis) in Nonalcoholic Steatohepatitis: A Case Report. Seminars in Liver Disease, 25, 230-236.
http://dx.doi.org/10.1055/s-2005-871202
[28] Vega, G.I., Chandalia, M., Szczepaniak, L.S., et al. (2007) Metabolic Correlates of Nonalcoholic Fatty Liver in Women and Men. Hepatology, 46, 716-722.
http://dx.doi.org/10.1002/hep.21727
[29] van der Poorten, D., Milner, K.L., Hui, J., et al. (2008) Visceral Fat a Key Mediator of Steatohepatitis in Metabolic Liver Disease. Hepatology, 48, 449-457.
http://dx.doi.org/10.1002/hep.22350
[30] Fan, J.G., Li, F., Cai, X.B., et al. (2007) Effects of Nonalcoholic Fatty Liver Disease on the Development of Metabolic Disorders. Journal of Gastroenterology and Hepatology, 22, 1086-1091.
http://dx.doi.org/10.1111/j.1440-1746.2006.04781.x

Copyright © 2023 by authors and Scientific Research Publishing Inc.

Creative Commons License

This work and the related PDF file are licensed under a Creative Commons Attribution 4.0 International License.