Surgery is the preferred treatment for bile duct hamartomas

Yang,Xiao‑Yu; Zhang,Hai‑Bo; Wu,Bin; Li,Ai‑Jun; Fu,Xiao‑Hui

doi:10.3892/mco.2017.1354

Surgery is the preferred treatment for bile duct hamartomas

Authors:
- Xiao‑Yu Yang
- Hai‑Bo Zhang
- Bin Wu
- Ai‑Jun Li
- Xiao‑Hui Fu
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Affiliations: Division of Special Treatment II, Eastern Hepatobiliary Surgery Hospital, Second Military Medical University, Shanghai 200438, P.R. China, The First Department of General Surgery, Cixian People's Hospital, Handan, Hebei 056500, P.R. China
Published online on: July 31, 2017 https://doi.org/10.3892/mco.2017.1354
Pages: 649-653

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Abstract

Bile duct hamartoma (BDH) is a rare liver disease with a rising incidence in recent years. A retrospective study on BDH patients between June 2007 and December 2015 was conducted. All the medical records, including clinical, imaging and pathological characteristics, were retrieved. Follow‑up data were obtained by telephone communication. The majority of the patients had no symptoms and the laboratory tests were normal. The appearance on ultrasound examination was variable. The lesion exhibited low density on plain computed tomography and no enhancement in 4 cases, and inhomogeneous enhancement in 1 case. Magnetic resonance imaging examination revealed hypointensity on T1-weighted imaging (WI), hyperintensity on T2WI, no enhancement in 1 case, and enhancement in 4 cases. Two patients underwent magnetic resonance cholangiopancreatography due to jaundice and intrahepatic bile duct dilation. The preoperative diagnosis was liver cancer in 4 cases, hepatic hilar biliary obstruction in 1, chronic calculous cholecystitis in 1, suspected hepatic abscess in 1, and benign liver tumor in 2 cases. All the patients underwent surgery. Nest‑like cell arrangements were observed in 2 patients, and in 1 patient the lesion transformed to intrahepatic cholangiocarcinoma, resulting in the patient's death 25 months after surgery. Thus, BDH may transform into a life‑threatening cholangiocarcinoma and it should be surgically removed. Furthermore, the mechanism underlying carcinogenesis in BDH requires further investigation.

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1	von Meyenburg H: Uber die Cystenleber. Beitr Pathol Anat. 64:477–532. 1918.(In German).
2	Singh Y, Cawich SO, Ramjit C and Naraynsingh V: Rare liver tumor: Symptomatic giant von Meyenburg complex. J Surg Case Rep. 2016:pii: rjw1952017. View Article : Google Scholar
3	Kim HK and Jin SY: Cholangiocarcinoma arising in von Meyenburg complexes. Korean J Hepatol. 17:161–164. 2011. View Article : Google Scholar : PubMed/NCBI
4	Parekh V and Peker D: Malignant transformation in von-meyenburg complexes: Histologic and immunohistochemical clues with illustrative cases. Appl Immunohistochem Molecul Morphol: AIMM. 23:607–614. 2015. View Article : Google Scholar
5	Gupta A, Pattnaik B, Das A and Kaman L: Von Meyenburg complex and complete ductal plate malformation along with Klatskin tumour: A rare association. BMJ Case Reports. 2016:10.1136/bcr-2016-215220, 2016.
6	Chandramouleeswari K, Anita S and Shivali B: Mesenchymal hamartoma of the liver: A case report. J Clin Diagn Res. 6:1552–1554. 2012.PubMed/NCBI
7	Hashimoto M, Ouchi M, Norose J, Suda FS, Suzuki K, Matsumura N, Lgari Y, Suzuki T, Nakano H, Mizuse M, et al: Bile duct hamartomas (von Meyenburg complexes) associated with a bacterial infection: Case report of elderly diabetic patient. Geriatr Geront Int. 11:534–536. 2011. View Article : Google Scholar
8	Sinakos E, Papalavrentios L, Chourmouzi D, Dimopoulou D, Drevelegas A and Akriviadis E: The clinical presentation of Von Meyenburg complexes. Hippokratia. 15:170–173. 2011.PubMed/NCBI
9	Cnossen WR and Drenth JP: Polycystic liver disease: An overview of pathogenesis, clinical manifestations and management. Orphanet J Rare Dis. 9:692014. View Article : Google Scholar : PubMed/NCBI
10	Xu AM, Xian ZH, Zhang SH and Chen XF: Intrahepatic cholangiocarcinoma arising in multiple bile duct hamartomas: Report of two cases and review of the literature. Eur J Gastroenterol Hepatol. 21:580–584. 2009. View Article : Google Scholar : PubMed/NCBI
11	Lung PF, Jaffer OS, Akbar N, Sidhu PS and Ryan SM: Appearances of von meyenburg complex on cross sectional imaging. J Clin Imaging Sci. 3:222013. View Article : Google Scholar : PubMed/NCBI
12	Shin YM: Biliary hamartoma presented as a single mass. Korean J Hepatol. 17:331–334. 2011. View Article : Google Scholar : PubMed/NCBI
13	Shi QS, Xing LX, Jin LF, Wang H, Lv XH and Du LF: Imaging findings of bile duct hamartomas: A case report and literature review. Int J Clin Exp Med. 8:13145–13153. 2015.PubMed/NCBI
14	Wohlgemuth WA, Böttger J and Bohndorf K: MRI, CT, US and ERCP in the evaluation of bile duct hamartomas (von Meyenburg complex): A case report. Eur Radiol. 8:1623–1626. 1998. View Article : Google Scholar : PubMed/NCBI
15	Luo TY, Itai Y, Eguchi N, Kurosaki Y, Onaya H, Ahmadi Y, Niitsu M and Tsunoda HS: Von Meyenburg complexes of the liver: imaging findings. J Comput Assist Tomogr. 22:372–378. 1998. View Article : Google Scholar : PubMed/NCBI
16	Zheng RQ, Zhang B, Kudo M, Onda H and Inoue T: Imaging findings of biliary hamartomas. World J Gastroenterol. 11:6354–6359. 2005. View Article : Google Scholar : PubMed/NCBI
17	Suzuki H, Tsurita G, Ishihara S, Akahane M, Kitayama J and Nagawa H: Resovist-enhanced MRI for preoperative assessment of colorectal hepatic metastases: A case of multiple bile duct hamartomas associated with colon cancer. Case Rep Gastroenterol. 2:509–516. 2008. View Article : Google Scholar : PubMed/NCBI
18	Patel S, Rajalakshmi BR and Manjunath GV: Histopathologic findings in autopsies with emphasis on interesting and incidental findings-a pathologist's perspective. J Clin Diagn Res. 10:EC08–EC12. 2016.PubMed/NCBI
19	Gong J, Kang W and Xu J: MR imaging and MR Cholangiopancreatography of multiple biliary hamartomas. Quant Imaging Med Surg. 2:133–134. 2012.PubMed/NCBI
20	Lin S, Weng Z, Xu J, Wang MF, Zhu YY and Jiang JJ: A study of multiple biliary hamartomas based on 1697 liver biopsies. Eur J Gastroenterol Hepatol. 25:948–952. 2013. View Article : Google Scholar : PubMed/NCBI
21	Saidi RF, Yoon V, Jabbour N, Shah SA and Bozorgzadeh A: Liver transplantation from a donor with multiple biliary hamartomata. Int J Organ Transplant Med. 4:35–37. 2013.PubMed/NCBI
22	Barboi OB, Moisii LG, Albu-Soda A, Ciortescu I and Drug V: Biliary hamartoma. Clujul Med. 86:383–384. 2013.PubMed/NCBI
23	Yamada N, Sanada Y, Katano T, Tashiro M, Hirata Y, Okada N, Ihara Y, Miki A, Sasanuma H, Urahashi T, et al: Pediatric living donor liver transplantation for congenital hepatic fibrosis using a mother's graft with von Meyenburg complex: A case report. World J Gastroenterol. 22:9865–9870. 2016. View Article : Google Scholar : PubMed/NCBI
24	van Vlerken LG, van Leeuwen MS, Schipper ME and van Erpecum KJ: The ‘Von Meyenburg complex’: An unusual cause of cholangitis? Clin Res Hepatol Gastroenterol. 35:762–764. 2011. View Article : Google Scholar : PubMed/NCBI
25	Ioannidis O, Iordanidis F, Paraskevas G, Ntoumpara M, Tsigkriki L, Chatzopoulos S, Kotronis A, Papadimitriou N, Konstantara A, Makrantonakis A, et al: Incidentally discovered white subcupsular liver nodules during laparoscopic surgery: Biliary hamartoma and peribiliary gland hamartoma. Klin Onkol. 25:468–470. 2012.PubMed/NCBI
26	Patel SR, Misra V, Verma K, Gupta P and Dhingra V: Benign hepatic mesenchymal hamartoma (HMH) - A case report. J Clin Diagn Res. 8:119–120. 2014.PubMed/NCBI
27	Ettel M, Eze O and Xu R: Clinical and biological significance of precursor lesions of intrahepatic cholangiocarcinoma. World J Hepatol. 7:2563–2570. 2015. View Article : Google Scholar : PubMed/NCBI
28	Liu S, Zhao B, Ma J, Li J and Li X: Lesions of biliary hamartoms can be diagnosed by ultrasonography, computed tomography and magnetic resonance imaging. Int J Clin Exp Med. 7:3370–3377. 2014.PubMed/NCBI
29	Lee KB: Histopathology of a benign bile duct lesion in the liver: Morphologic mimicker or precursor of intrahepatic cholangiocarcinoma. Clin Mol Hepatol. 22:400–405. 2016. View Article : Google Scholar : PubMed/NCBI