Multiple Inflammatory and Serum Amyloid A Positive Telangiectatic Hepatic Adenomas with Glycogenated Nuclei Arising in a Background of Nonalcoholic Steatohepatitis

ABSTRACT

The authors describe multiple telangiectatic or inflammatory adenomas in a 53-year-old woman with steatohepatitis who presented with acute right upper quadrant abdominal pain. Magnetic resonance imaging revealed 6 lesions consistent with multiple hepatic adenomas, 2 of which showed hemorrhage. She underwent right lobectomy and nonanatomical segment 2 liver resections and seven nodules ranging in size from 1.0 to 5.0 cm were identified. All nodules contained portal-like structures and ductular reaction, features seen in focal nodular hyperplasia, as well as significant inflammation, telangiectatic sinusoids and immunoreactivity for serum amyloid A, placing them according to a recently described classification systems as telangiectatic or inflammatory adenomas. The diffuse positivity of the serum amyloid A staining results in this case suggests an important diagnostic role of this stain in smaller tissue samples, such as in core biopsy specimens.

KEYWORDS

Hepatic adenoma - nonalcoholic steatohepatitis - telangiectatic adenoma - inflammatory adenoma

REFERENCES

• 1 Wanless I R, Gryfe A. Nodular transformation of the liver in hereditary hemorrhagic telangiectasia.  Arch Pathol Lab Med. 1986;  110 331-335
• 2 Wanless I R, Albrecht S, Bilbao J et al.. Multiple focal nodular hyperplasia of the liver associated with vascular malformations of various organs and neoplasia of the brain: a new syndrome.  Mod Pathol. 1989;  2 456-462
• 3 Nguyen B N, Flejou J-F, Terris B et al.. Focal nodular hyperplasia of the liver: a comprehensive pathologic study of 305 lesions and recognition of new histologic forms.  Am J Surg Pathol. 1999;  23 1441-1463
• 4 Bioulac-Sage P, Reboiussou S, Cunha A S et al.. Clinical, morphologic and molecular features defining so-called telangiectatic focal nodular hyperplasia of the liver.  Gastroenterology. 2005;  128 1211-1218
• 5 Bioulac-Sage P, Rebouissou S, Thomas C et al.. Hepatocellular adenoma subtype classification using molecular markers and immunohistochemistry.  Hepatology. 2007;  46 740-748
• 6 Paradis V, Champault A, Ronot M et al.. Telangiectatic adenoma: an entity associated with increased body mass index and inflammation.  Hepatology. 2007;  46 140-146
• 7 Zucman-Rossi J, Benhamouche S, Godard C et al.. Differential effects of inactivated Axin 1 and activated beta-catenin mutations in human hepatocellular carcinomas.  Oncogene. 2007;  26 774-780
• 8 Attal P, Vilgrain V, Brancatelli G et al.. Telangiectatic focal nodular hyperplasia: US, CT, and MR imaging findings with histopathologic correlation in 13 cases.  Radiology. 2003;  228 465-472
• 9 Peterfy C G, Rosenthall L. Large telangiectatic focal nodular hyperplasia presenting with normal radionuclide studies: case report.  J Nucl Med. 1990;  31 2037-2039
• 10 Haber M, Rueben A, Oliveiro P et al.. Multiple focal nodular hyperplasia of the liver associated with hemihyperthrophy and vascular malformations.  Gastroenterology. 1995;  108 1256-1262
• 11 Guindi M. So called telangiectatic focal nodular hyperplasia: a newly recognized variant of hepatic adenoma.  Pathol Case Rev. 2007;  12 154-159
• 12 Bioulac-Sage P, Balabaud C, Bedossa P et al.. Pathological diagnosis of liver cell adenoma and focal nodular hyperplasia: Bordeaux update.  J Hepatol. 2007;  46 521-527
• 13 Kim H-S, Kim Y A, Kim C J et al.. Telangiectatic focal nodular hyperplasia of the liver: a case detected at birth.  J Korean Med Sci. 2003;  18 746-750
• 14 Zucman-Rossi J, Jeannot E, Nhieu J T et al.. Genotype-phenotype correlation in hepatocellular adenoma: a new classification and relationship with HCC.  Hepatology. 2006;  43 515-524
• 15 Yamagata K, Oda N, Kaisaki P J et al.. Mutations in the hepatocyte nuclear factor-1alpha gene in maturity-onset diabetes of the young (MODY3).  Nature. 1996;  384 458-460
• 16 Bacq Y, Jacquemin E, Balabaud C et al.. Familial liver adenomatosis associated with hepatocytes nuclear factor 1alpha inactivation.  Gastroenterology. 2003;  125 1470-1475
• 17 Reznik Y, Dao T, Coutant R et al.. Hepatocyte nuclear factor-1 alpha gene inactivation: cosegregation between liver adenomatosis and diabetes phenotypes in two maturity-onset diabetes of the young (MODY3) families.  J Clin Endocrinol Metab. 2004;  89 1476-1480