Ann Hepatobiliary Pancreat Surg.  2023 Feb;27(1):102-106. 10.14701/ahbps.22-024.

Not a neuroendocrine tumor: A case of hepatocellular carcinoma in ectopic liver tissue in the pancreas

• Correia AM
• Ribeiro C
• Videira F
• Gigliano D
• Cunha AL
• Afonso LP
• Peyroteo M
• Canotilho R
• Baía C
• Sousa F
• Sousa JAd

• KMID: 2539520
• DOI: http://doi.org/10.14701/ahbps.22-024

Abstract

Hepatocellular carcinoma (HCC) accounts for most of the hepatic neoplasms and can also occur in ectopic liver tissue. We present a case of a 55-year-old male complaining of weight loss. The imaging studies reported a 2.9 cm nodule in the pancreatic body, with a neuroendocrine tumor diagnosis by cytology. A corpo-caudal pancreatectomy was performed. Pathology showed a well-differentiated HCC developed in ectopic liver tissue with free margins and no lymph node metastases. HCC presenting in ectopic liver tissue is rare. In this case, the preoperative study did not establish the diagnosis, warranting the need for suspicion of this neoplasm.

Keyword

Hepatocellular carcinoma; Ectopic tissue; Surgical oncology; Differential diagnosis; Pancreatic neoplasms

Figure

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  Fig. 1 Thoracoabdominal and pelvic computed tomography (CT) showing a 2.9 cm × 2.5 cm × 2.8 cm solid well-circumscribed nodule inseparable from the superior border of the pancreatic body to the left of the celiac trunk, apparently irrigated by a branch of the splenic artery. The lesion is slightly hypervascular and does not have direct contact with the gastric wall. Due to its characteristics, it was considered more likely to be a neuroendocrine tumor of the pancreas, requiring characterization by biopsy, with the hypothesis of a gastrointestinal stromal tumor being unlikely. The remaining pancreatic gland presents a homogeneous texture of the parenchyma, without anomalous dilation of the Wirsung duct. The liver has a normal morphology and dimensions, presenting a homogeneous parenchymal texture. Along the retroperitoneum and less prominently in the mesentery, several lymph nodes may be observed, the largest an interaortocaval lymph node with a short-axis diameter measurement of 1 cm, of uncertain pathological significance. Coronal view (A) and axial view: arterial phase (B) and portal phase (C).

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  Fig. 2 Positron emission tomography (PET) 68Ga-DOTANOC scan. Discrete to moderate 68Ga-DOTANOC uptake (SUVmax = 5.8) in a lesion of uncertain etiology located in the upper surface of the body of the pancreas body (arrow): a tumor with low expression of somatostatin receptors? In the remaining study, no other foci of anomalous or significantly increased uptake were observed. Absence of 68Ga-DOTANOC uptake in the right upper lobe lung lesion.

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  Fig. 3 Gross view of the formalin-fixed surgical specimen. (A) Pancreatic tail with a solid and circumscribed tumor. (B) Detail of the limit of the pancreatic parenchyma and the tumor.

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  Fig. 4 Photomicrographs of the tumor. (A) Solid neoformation, predominantly wellcircumscribed and surrounded by a thin fibrous capsule (H&E, 40×). (B) Coexistence of two distinct microscopic patterns (pseudoglandular and trabecular): a common finding in hepatocellular carcinomas (H&E, 40×). (C) A positive reaction for HepPar1 confirms the hepatocellular nature of the lesion (DAB, 100×). (D) A positive reaction for CD34 in the hepatic sinusoids is a sign of “capillarization,” typical of hepatocellular carcinomas (DAB, 100×). (E) Positivity for polyclonal carcinoembryonic antigen (CEA) (canalicular pattern) further supports the hepatocellular differentiation of the neoplasm (DAB, 400×).

Reference

1. Forman D, Ferlay J. Stewart BW, Wild CP, editors. 2014. The global and regional burden of cancer. World cancer report 2014. International Agency for Research on Cancer;Lyon: p. 16–53.

2. Zhuo Y, Chen Q, Chhatwal J. Hoshida Y, editor. 2019. Changing epidemiology of hepatocellular carcinoma and role of surveillance. Hepatocellular carcinoma: translational precision medicine approaches. Humana Press;Cham: p. 53–67.

3. Ferlay J, Ervik M, Lam F, Colombet M, Mery L, Piñeros M, et al. Population Fact Sheets - Portugal [Internet]. Available from: https://gco.iarc.fr/today/fact-sheets-populations. Lyon: Cancer Today/IARC;2019. cited 2020 Oct 8.

4. European Association For The Study Of The Liver. European Organisation For Research And Treatment Of Cancer. 2012; EASL-EORTC clinical practice guidelines: management of hepatocellular carcinoma. J Hepatol. 56:908–943. Erratum in: J Hepatol 2012;56:1430. DOI: 10.1016/j.jhep.2012.03.006.

5. Hartke J, Johnson M, Ghabril M. 2017; The diagnosis and treatment of hepatocellular carcinoma. Semin Diagn Pathol. 34:153–159. DOI: 10.1053/j.semdp.2016.12.011. PMID: 28108047.

6. Lee HW, Cho KJ, Park JY. 2020; Current status and future direction of immunotherapy in hepatocellular carcinoma: what do the data suggest? Immune Netw. 20:e11. DOI: 10.4110/in.2020.20.e11. PMID: 32158599. PMCID: PMC7049588.

7. Lee JY, Kim KH, Kang MS, Kim KH. 2015; Ectopic hepatocellular carcinoma arising from the peritoneum in a patient with a history of oropharyngeal cancer: a case report. Case Rep Oncol. 8:456–460. DOI: 10.1159/000441020. PMID: 26600779. PMCID: PMC4649744.

8. Cardona D, Grobmyer S, Crawford JM, Liu C. 2007; Hepatocellular carcinoma arising from ectopic liver tissue in the pancreas. Virchows Arch. 450:225–229. DOI: 10.1007/s00428-006-0353-8. PMID: 17216188.

9. Kanzaki R, Yamada T, Gotoh K, Takahashi H, Ohigashi H, Ishikawa O. 2010; Ectopic hepatocellular carcinoma arising in the left triangular ligament of the liver. Case Rep Gastroenterol. 4:138–143. DOI: 10.1159/000314042. PMID: 20805935. PMCID: PMC2929406.

10. Adachi Y, Hayashi H, Yusa T, Takematsu T, Matsumura K, Higashi T, et al. 2020; Ectopic hepatocellular carcinoma mimicking a retroperitoneal tumor: a case report. World J Gastroenterol. 26:2268–2275. DOI: 10.3748/wjg.v26.i18.2268. PMID: 32476791. PMCID: PMC7235206.

11. Braun M, Kuncman W, Teresiński L, Kupnicki P, Jesionek-Kupnicka D, Kordek R. 2017; Pure hepatocellular carcinoma originates from an ectopic liver nodule located in the pancreas. Contemp Oncol (Pozn). 21:311–314. DOI: 10.5114/wo.2017.72403. PMID: 29416439. PMCID: PMC5799705.

12. Matsuyama M, Sugiura S, Kakita A, Sato Y, Kuroda M. 2011; Hepatocellular carcinoma arising from ectopic liver tissue in the spleen producing insulin-like growth factor II. Pathol Res Pract. 207:124–126. DOI: 10.1016/j.prp.2010.09.003. PMID: 20943327.

13. Caygill C, Gatenby P. 2004; Ectopic liver and hepatocarcinogenesis. Eur J Gastroenterol Hepatol. 16:727–729. DOI: 10.1097/01.meg.0000131037.92864.df. PMID: 15256972.

14. Mani VR, Farooq MS, Soni U, Kalabin A, Rajabalan AS, Ahmed L. 2016; Case report of ectopic liver on gallbladder serosa with a brief review of the literature. Case Rep Surg. 2016:7273801. DOI: 10.1155/2016/7273801. PMID: 27803835. PMCID: PMC5075603.

15. Li Z, Wu X, Wen T, Li C, Peng W. 2017; Multiple ectopic hepatocellular carcinomas in the pancreas: a case report. Medicine (Baltimore). 96:e6747. DOI: 10.1097/MD.0000000000006747. PMID: 28746170. PMCID: PMC5627796.

16. WHO Classification of Tumours Editorial Board. 2019. Digestive system tumours: WHO classification of tumours, Volume 1. 5th ed. International Agency for Research on Cancer;Lyon:

17. Zeng SX, Tan SW, Fong CTH, Liang Q, Zhao BL, Liu K, et al. 2020; Hepatoid carcinoma of the pancreas: a case report and review of the literature. World J Clin Cases. 8:1116–1128. DOI: 10.12998/wjcc.v8.i6.1116. PMID: 32258082. PMCID: PMC7103969.

18. Steen S, Wolin E, Geller SA, Colquhoun S. 2013; Primary hepatocellular carcinoma ("hepatoid" carcinoma) of the pancreas: a case report and review of the literature. Clin Case Rep. 1:66–71. DOI: 10.1002/ccr3.26. PMID: 25356215. PMCID: PMC4184752.

19. Kubota K, Kita J, Rokkaku K, Iwasaki Y, Sawada T, Imura J, et al. 2007; Ectopic hepatocellular carcinoma arising from pancreas: a case report and review of the literature. World J Gastroenterol. 13:4270–4273. DOI: 10.3748/wjg.v13.i31.4270. PMID: 17696261. PMCID: PMC4250631.

20. Kelly PJ, Spence R, Dasari BV, Burt AD, Taylor M, Loughrey MB. 2012; Primary hepatocellular carcinoma of the pancreas: a case report and review of the heterogeneous group of pancreatic hepatoid carcinomas. Histopathology. 60:1012–1015. DOI: 10.1111/j.1365-2559.2011.04129.x. PMID: 22320681.