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经腹腔镜部分肝切除术治疗的长期逐渐增大的多囊性胆管错构瘤

Multicystic biliary hamartoma with long-term gradual enlargement treated by laparoscopic partial hepatectomy.

作者信息

Nishiwada Satoshi, Tanaka Tetsuya, Kirihataya Yuki, Takei Takeshi, Sadamitsu Tomomi, Takano Masato, Sawai Masayoshi, Yoshimura Atsushi

机构信息

Department of Surgery, Minami-Nara General Medical Center, 8 - 1 Fukugami Oyodo-cho, Yoshino, Nara, 638 - 8551, Japan.

Department of Pathology, Minami-Nara General Medical Center, 8 - 1 Fukugami Oyodo-cho, Yoshino, Nara, 638 - 8551, Japan.

出版信息

Clin J Gastroenterol. 2025 Jun;18(3):527-534. doi: 10.1007/s12328-025-02124-z. Epub 2025 Apr 5.

DOI:10.1007/s12328-025-02124-z
PMID:40186804
Abstract

Multicystic biliary hamartoma (MCBH) is an extremely rare liver tumor characterized by a well-circumscribed, multicystic honeycomb appearance on imaging. Herein, we report a case of long-term gradually enlarging MCBH that underwent laparoscopic hepatectomy. A 69-year-old man presented with mildly elevated carcinoembryonic antigen levels and underwent computed tomography (CT) 11 and 5 years ago, at which time the physicians did not note any particular abnormal findings. The current contrast CT for screening demonstrated a cystic lesion in segment 2 of the liver. A retrospective review of CT images showed that the cystic lesion had gradually increased over time. The patients successfully underwent laparoscopic partial hepatectomy and recovered without any complications. The surgically resected specimen grossly presented an aggregated nodule of small cysts. Pathological findings showed multiple cysts of various sizes covered with cuboidal cells without dysplasia resembling bile duct epithelium, with lumens containing a stone component consisting of brown bile. Around the cyst, there was vascular fibrous tissue, smooth muscle bundles, normal hepatocytes, and chronic inflammation. Based on these findings, we finally diagnosed MCBH. Although MCBH is histologically classified as a benign tumor, physicians may have to pay careful attention to the potential for enlargement in actual clinics.

摘要

多囊性胆管错构瘤(MCBH)是一种极其罕见的肝脏肿瘤,在影像学上表现为边界清晰的多囊性蜂窝状外观。在此,我们报告一例长期逐渐增大的MCBH病例,该病例接受了腹腔镜肝切除术。一名69岁男性癌胚抗原水平轻度升高,11年前和5年前接受了计算机断层扫描(CT)检查,当时医生未发现任何特殊异常发现。此次用于筛查的增强CT显示肝脏2段有一个囊性病变。对CT图像的回顾性分析显示,该囊性病变随时间逐渐增大。患者成功接受了腹腔镜部分肝切除术,术后恢复良好,无任何并发症。手术切除标本肉眼可见为小囊肿聚集形成的结节。病理结果显示多个大小不一的囊肿,被立方体细胞覆盖,无发育异常,类似胆管上皮,管腔内含有由棕色胆汁组成的结石成分。囊肿周围有血管纤维组织、平滑肌束、正常肝细胞和慢性炎症。基于这些发现,我们最终诊断为MCBH。尽管MCBH在组织学上被归类为良性肿瘤,但在实际临床中医生可能仍需密切关注其增大的可能性。

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本文引用的文献

1
Characteristics of multicystic biliary hamartoma: A case report.多囊性胆管错构瘤的特征:一例报告
Front Surg. 2023 Jan 6;9:1074899. doi: 10.3389/fsurg.2022.1074899. eCollection 2022.
2
A Case of Multicystic Biliary Hamartoma with a Marked Peribiliary Gland Component Successfully Treated by Purely Laparoscopic Anatomical Liver Resection.一份多房性胆管错构瘤病例报告,该肿瘤具有显著的周围胆管腺体成分,成功地通过纯腹腔镜解剖性肝切除术治疗。
J Gastrointest Cancer. 2023 Sep;54(3):996-999. doi: 10.1007/s12029-022-00893-1. Epub 2022 Nov 30.
3
Intrahepatic multicystic biliary hamartoma: A case report.
肝内多囊性胆管错构瘤:一例报告
World J Clin Cases. 2022 Sep 16;10(26):9361-9367. doi: 10.12998/wjcc.v10.i26.9361.
4
Multicystic Biliary Hamartoma With Xanthogranulomatous Inflammation on 18F-FDG PET/CT.18F-FDG PET/CT 上表现为多囊性胆管错构瘤伴黄色肉芽肿性炎症
Clin Nucl Med. 2022 Oct 1;47(10):882-884. doi: 10.1097/RLU.0000000000004204. Epub 2022 Apr 27.
5
Multicystic biliary hamartoma.多囊性胆管错构瘤
Cir Esp (Engl Ed). 2022 Dec;100(12):800-802. doi: 10.1016/j.cireng.2022.08.009. Epub 2022 Sep 1.
6
Case Report: Incidentally Discovered a Rare Cystic Lesion of Liver: Multicystic Biliary Hamartoma.病例报告:偶然发现的罕见肝脏囊性病变:多囊性胆管错构瘤。
Pathol Oncol Res. 2021 Mar 30;27:628323. doi: 10.3389/pore.2021.628323. eCollection 2021.
7
Intrahepatic cholangiocarcinoma and portal hypertension developing in a patient with multicystic biliary microhamartomas.患有多囊性胆管微错构瘤的患者发生肝内胆管癌和门静脉高压症。
Malays J Pathol. 2018 Dec;40(3):331-335.
8
A huge rapidly-enlarging multicystic biliary hamartoma.一个巨大的迅速增大的多囊性胆管错构瘤。
Dig Liver Dis. 2018 Jul;50(7):723. doi: 10.1016/j.dld.2018.01.136. Epub 2018 Feb 5.
9
Multicystic biliary hamartoma with extremely elevated CA19-9: a case report.伴有CA19-9极度升高的多囊性胆管错构瘤:病例报告
Scand J Gastroenterol. 2017 Aug;52(8):916-919. doi: 10.1080/00365521.2017.1322140. Epub 2017 May 9.
10
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Clin J Gastroenterol. 2014 Oct;7(5):418-21. doi: 10.1007/s12328-014-0513-3. Epub 2014 Jul 12.