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Original research
von Meyenburg complexes are more frequently associated with cholangiocarcinoma
  1. Dhanpat Jain1,
  2. Binny Khandakar2,
  3. Pu Ni3,
  4. Barton Kenney4,
  5. Lihui Qin5,
  6. Vikram Deshpande6,
  7. Maria Isabel Fiel3
  1. 1Department of Pathology, Yale School of Medicine, New Haven, Connecticut, USA
  2. 2Department of Pathology and Laboratory Medicine, Donald Barbara Zucker School of Medicine at Hofstra/Northwell, Manhasset, New York, USA
  3. 3Department of Pathology, Molecular and Cell-Based Medicine, Icahn School of Medicine at Mount Sinai, New York City, New York, USA
  4. 4Department of Pathology, Middlesex Health, Middletown, Connecticut, USA
  5. 5Department of Pathology, Weill Cornell Medical College, New York City, New York, USA
  6. 6Department of Pathology, Harvard Medical School, Boston, Massachusetts, USA
  1. Correspondence to Dr Dhanpat Jain, Pathology, Yale School of Medicine, New Haven, CT 06510, USA; dhanpat.jain{at}yale.edu

Abstract

Aim There is some evidence that von Meyenburg complexes (VMCs) can progress to cholangiocarcinoma (CC). This study aimed to evaluate the prevalence of VMCs in CC cases.

Methods All hepatic resections and explants with intra-hepatic CC (I-CC) and hilar-CC (H-CC) from 1985 to 2020 were studied. Hepatic resections (n=68) for benign lesions or metastatic colonic carcinoma and 15 cases with cirrhosis without any cancer were used as controls.

Results A total of 118 cases of CC (88 I-CC, 30 H-CC) were identified. Of these, 61 (52%) patients had no known background liver disease, and 20 (17%) had cirrhosis. Associated liver disorders included metabolic dysfunction-associated steatohepatitis (23), chronic viral hepatitis B or C (13), biliary disease (primary or secondary sclerosing cholangitis) (8), polycystic kidney disease (6), cryptogenic cirrhosis (5) and others miscellaneous disorders (7). VMCs were present in 34 (39%) of 88 I-CC cases and 7 (23%) of 30 H-CC cases. VMCs were present within the tumour (20 cases), outside the cancer (21 cases) or at both locations (10 cases). VMCs with dysplasia/carcinoma in situ were seen in 19 of 41 (46%) cases with CC and VMCs. In addition, bile duct adenomas were identified in 6 (5%) of CC. 7% of controls showed the presence of VMCs compared with 35% of CC cases (p<0.05).

Conclusions VMCs are seen far more frequently in patients with CC than in the control group. The findings support the hypothesis that VMCs could represent a precursor of CC or a marker for a higher risk of developing CC.

  • CHOLANGIOCARCINOMA
  • Liver Neoplasms
  • HAMARTOMA

Data availability statement

Data sharing not applicable as no datasets generated and/or analysed for this study.

https://doi.org/10.1136/jcp-2024-209572

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    Data availability statement

    Data sharing not applicable as no datasets generated and/or analysed for this study.

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    Footnotes

    • Handling editor Yoh Zen.

    • Contributors DJ: planning, conduct, reporting, conception and design of the study, acquisition of data, analysis and interpretation of data, writing and editing of manuscript. MIF and VD: planning, conduct, reporting, conception and design of the study, editing of manuscript. BK, PN, BK and LQ: acquisition of data and analysis, writing and editing of manuscript draft. DJ and MIF: guarantor

    • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

    • Competing interests None declared.

    • Provenance and peer review Not commissioned; internally peer reviewed.