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DOI: 10.1055/s-2005-871202
Copyright © 2005 by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA.
Benign Hepatocellular Tumors (Adenomatosis) in Nonalcoholic Steatohepatitis: A Case Report
Publication History
Publication Date:
25 May 2005 (online)
Hepatic adenomatosis is a very rare condition of multiple benign hepatocellular tumors involving a noncirrhotic liver.[1] [2] The definition is exclusive of glycogen storage disease or androgen use, as these conditions are known for their potential to give rise to multiple hepatocellular tumors, some of which may be adenomas or hepatocellular carcinomas (HCCs).[3] [4] [5] A review of the literature from 1963 to 2003[1] [2] [6] [7] [8] [9] [10] [11] [12] [13] [14] [15] [16] [17] [18] [19] [20] [21] [22] [23] [24] [25] shows that adenomatosis is more common in women but, unlike adenomas, has a variable relationship to oral contraceptive use (Table [1]). Nonalcoholic steatohepatitis (NASH) is considered the hepatic manifestation of the metabolic syndrome (obesity, insulin resistance, hyperlipidemia, and hypertension); NASH may progress to cirrhosis, and cases of HCC in cirrhotic NASH have been reported.[26] [27] [28] [29] [30] [31] To our knowledge, benign hepatocellular neoplasms have not been reported in association with NASH. In this report, we document a case of hepatic adenomatosis arising in a noncirrhotic liver with steatohepatitis.
Table 1 Review of the Literature on Adenomatosis Author, Year, Cases (n), Ages (yrs) M:F Oral Contraceptive Use and/or Para Diabetes/Familial Steatosis, HCC, and Other Liver Lesions Monaco, 19646 21 F N/A Para: 0 No/ N/A Resection: multiple tumors 0.8-3.5 cm, some steatotic; no steatosis in uninvolved parenchyma Brander, 19767 24 F High dose for DUB, 6 years Para: 0 No/N/A Rupture of liver; multiple tumors; no steatosis Edmondson, 19778 n = 2 (32, 35) 0:2 100% (4, 5 years) Para: 0, 6 N/A Tissue dx: no mention of steatosis Kalra, 198710 n = 2 (23, 33) 0:2 DUB, renal tx N/A Tissue dx: no mention of steatosis Foster, 19789 n = 3 (15,17,43) 1:2 Not stated 43 years/o was mother 3/3 Steatosis not mentioned Liu, 198011 39 F No Para: 4 No/ N/A Steatosis not mentioned Sinha, 198212 37 F No Para: 3 N/A Steatotic nodules but not in nontumor liver Chen, 198313 28 F No Para: 0 N/A Presented at age 12: liver tumors (adenomas) bxd; no steatosis. Died 15 years later, congenital heart disease; 10 lesions 1-12 cm; one FNH Le Bail, 199214 39, 52 2F 100% (12, 18 years) Para: 3, 2 N/A Mild zone 1 steatosis in one case Propst, 199515 30 F N/A N/A Steatosis not mentioned Gokhale, 199616 17 F No Para: 0 N/A Tissue bx: steatosis in tumors Arsenault, 199617 41 F No N/A N/A Tissue bx: mild steatosis in lesion Ye, 199918 42 F 25 years N/A One tumor with central HCC. Steatosis not mentioned Suarez, 200119 35 F 3 years; 21 years prior; Norplant, 2 years No/No Steatosis in adenomas; none in nontumor liver Flejou, 198520 n = 5 (13-75) Review of lit: 8 cases 3:2 No N/A No steatosis mentioned in nodules or in nontumor liver Leese, 198821 n = 2 (16, 45) 1:1 No 0 and N/A Steatosis not mentioned. Multiple HCCs in explant Ribeiro, 19981 n = 8 (37-48) Review of lit: 22 cases 0:8 88% N/A and 0 Steatosis not mentioned Yoshidome, 199922 n = 2 (29, 53) Review of lit: 30 cases 0:2 1, remote past N/A “Mild steatosis” in nontumor parenchyma in both cases Chiche, 20002 n = 8 (14-45) Review of lit: 30 cases 1:7 43% (1, 6, 15 years) 4/4 Steatosis in all adenomas; not mentioned in nontumor liver. FNH (2), portal vein agenesis (1) Grazioli, 200023 n = 15 (19-66) 1:14 36% (2-15 years) N/A Mild steatosis in a minority of tumors; not mentioned in nontumor liver. FNH (2); portal vein thrombosis (1); portal vein absence (2), p-h shunt (2); HCC in explant (1) and in allograft (1) Bluteau, 200224 n = 7 (27-49) 1:6 66% females Para: not known Not specified; some family members of adenomatosis cases Steatosis not mentioned Bacq, 200325 n = 5 (14-42) 2:3 66% females Para: one female, 1 Two families with affected members Steatosis in adenomas in 3; diffuse steatosis in one relative without tumor Total cases n = 71 10:61 OC use:42% See above Steatosis in nontumor liver: 4 HCC: 4 bx, biopsy; DUB, dysfunctional uterine bleeding; dx, diagnosis; FNH, focal nodular hyperplasia; HCC, hepatocellular carcinoma; OC, oral contraceptive; p-h, portal-hepatic; tx, treatment; N/A, not assessed.