Case Report
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World J Gastroenterol. Nov 21, 2014; 20(43): 16355-16358
Published online Nov 21, 2014. doi: 10.3748/wjg.v20.i43.16355
Ciliated hepatic foregut cyst with high intra-cystic carbohydrate antigen 19-9 level
Ziv Ben Ari, Oranit Cohen-Ezra, Jonathan Weidenfeld, Tania Bradichevsky, Ella Weitzman, Uri Rimon, Yael Inbar, Michal Amitai, Barak Bar-Zachai, Roni Eshkenazy, Arie Ariche, Daniel Azoulay
Ziv Ben Ari, Oranit Cohen-Ezra, Tania Bradichevsky, Ella Weitzman, Liver Disease Center, Sheba Medical Center and Sackler School of Medicine, Tel Aviv University, 52621 Tel Aviv, Israel
Jonathan Weidenfeld, Department of Pathology, Sheba Medical Center and Sackler School of Medicine, Tel Aviv University, 52621 Tel Aviv, Israel
Uri Rimon, Yael Inbar, Michal Amitai, Department of Radiology, Sheba Medical Center and Sackler School of Medicine, Tel Aviv University, 52621 Tel Aviv, Israel
Barak Bar-Zachai, Roni Eshkenazy, Arie Ariche, Daniel Azoulay, Depatrment of Hepatobiliary Surgery, Sheba Medical Center and Sackler School of Medicine, Tel Aviv University, 52621 Tel Aviv, Israel
Author contributions: Ben Ari Z and Azoulay D designed the study; Ben Ari Z, Cohen-Ezra O, Bradichevsky T, Weitzman E, Bar-Zachai B and Eshkenazy R collected the patient’s clinical data and performed the research; Rimon U, Amitai M and Inbar Y contributed the radiological images and their interpretation; Weidenfeld J contributed the histological studies and their interpretation; Ariche A and Azoulay D analyzed the data; Ben Ari Z wrote the paper.
Correspondence to: Ziv Ben Ari, MD, Professor of Internal Medicine, Liver Disease Center, Sheba Medical Center and Sackler School of Medicine, Tel Aviv University, Ramat Gan, 52621 Tel Aviv, Israel. ziv.ben-ari@sheba.health.gov.il
Telephone: +972-3-5307180 Fax: +972-3-5307190
Received: March 28, 2014
Revised: May 25, 2014
Accepted: July 16, 2014
Published online: November 21, 2014
Abstract

A ciliated hepatic foregut cyst (CHFC) is a rare foregut developmental malformation usually diagnosed in adulthood. Five percent of reported cases of CHFC transform into squamous cell carcinoma. We report the presentation, evaluation, and surgical management of a symptomatic 45-year-old male found to have a 6.2 cm CHFC. Contrast tomography-guided fine-needle aspiration demonstrated columnar, ciliated epithelium consistent with the histologic diagnosis of CHFC. The intracystic levels of carbohydrate antigen (CA) 19-9 and carcinoembryonic antigen (CEA) were extremely high (978118 U/mL and 973 μg/L, respectively). Histologically, the wall of the cyst showed characteristic pseudopapillae lined with a ciliated stratified columnar epithelium, underlying smooth muscle, an outer fibrous layer and no atypia. Immunohistochemistry for CA19-9 and CEA was positive. This is the first case report of a CHFC in which levels of CA 19-9 and CEA were measured. Our findings suggest that a large sized multilocular cyst and elevated cyst CA19-9 and CEA levels do not exclude a CHFC from consideration in the diagnosis. CHFCs should be included in the differential diagnosis of hepatic lesions. Accurate diagnosis of a CHFC is necessary given its potential for malignant transformation, and surgical excision is recommended.

Keywords: Ciliated hepatic foregut cyst, Carbohydrate antigen 19-9, Carcinoembryonic antigen, Computed tomography-guided fine-needle aspiration, Magnetic resonance imaging, Squamous cell carcinoma

Core tip: A ciliated hepatic foregut cyst (CHFC) is a rare foregut developmental malformation usually diagnosed in adulthood. Five percent of reported CHFC cases transform into squamous cell carcinoma. We report the evaluation and surgical management of a symptomatic 45-year-old male found to have a 6.2 cm CHFC diagnosed by an intracystic columnar, ciliated epithelium. We report for the first time extremely high intracystic level of carbohydrate antigen (CA) 19-9 and no atypia. A large sized multilocular cyst and elevated cyst CA19-9 do not exclude CHFCs from diagnostic consideration. Given its potential for malignant transformation, surgical excision is recommended.