title: Ciliated hepatic foregut cyst with high intra-cystic carbohydrate antigen 19-9 level.
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.
authors.affiliation: 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.
authors.forename: Ziv
authors.initials: Z
authors.lastname: Ben Ari
authors.affiliation: 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.
authors.forename: Oranit
authors.initials: O
authors.lastname: Cohen-Ezra
authors.affiliation: 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.
authors.forename: Jonathan
authors.initials: J
authors.lastname: Weidenfeld
authors.affiliation: 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.
authors.forename: Tania
authors.initials: T
authors.lastname: Bradichevsky
authors.affiliation: 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.
authors.forename: Ella
authors.initials: E
authors.lastname: Weitzman
authors.affiliation: 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.
authors.forename: Uri
authors.initials: U
authors.lastname: Rimon
authors.affiliation: 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.
authors.forename: Yael
authors.initials: Y
authors.lastname: Inbar
authors.affiliation: 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.
authors.forename: Michal
authors.initials: M
authors.lastname: Amitai
authors.affiliation: 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.
authors.forename: Barak
authors.initials: B
authors.lastname: Bar-Zachai
authors.affiliation: 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.
authors.forename: Roni
authors.initials: R
authors.lastname: Eshkenazy
authors.affiliation: 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.
authors.forename: Arie
authors.initials: A
authors.lastname: Ariche
authors.affiliation: 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.
authors.forename: Daniel
authors.initials: D
authors.lastname: Azoulay
mesh_terms: D015415:Biomarkers / Q000032:analysis; D044963:Biopsy, Fine-Needle; D018395:CA-19-9 Antigen / Q000032:analysis*; D002272:Carcinoembryonic Antigen / Q000032:analysis; D002923:Cilia / Q000473:pathology; D003560:Cysts / Q000151:congenital / Q000175:diagnosis / Q000276:immunology* / Q000601:surgery; D003937:Diagnosis, Differential; D004847:Epithelial Cells / Q000276:immunology* / Q000473:pathology; D006801:Humans; D061705:Image-Guided Biopsy / Q000379:methods; D007150:Immunohistochemistry; D008107:Liver Diseases / Q000151:congenital / Q000175:diagnosis / Q000276:immunology* / Q000601:surgery; D008279:Magnetic Resonance Imaging; D008297:Male; D008875:Middle Aged; D011237:Predictive Value of Tests; D014057:Tomography, X-Ray Computed; D016896:Treatment Outcome; D015854:Up-Regulation
chemical_list: D015415:Biomarkers; D018395:CA-19-9 Antigen; D002272:Carcinoembryonic Antigen
keywords: Carbohydrate antigen 19-9; Carcinoembryonic antigen; Ciliated hepatic foregut cyst; Computed tomography-guided fine-needle aspiration; Magnetic resonance imaging; Squamous cell carcinoma