Rare liver tumor: symptomatic giant von Meyenburg complex

Singh, Yudhvir; So, Cawich; Ramjit, Chunilal; Naraynsingh,

doi:10.1093/jscr/rjw195

Cited by 6 publications

(3 citation statements)

References 9 publications

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“…On gross examination, biliary hamartoma appears as gray-white, cystic or solid, unique, or multiple nodules scattered throughout the liver. Generally, they measure less than 0.5 cm, although larger lesions up to 20 cm may be seen [ 8 ]. They are usually well-delineated and non-encapsulated, often portal or sub-capsular, making them visible to surgeons who may incidentally see them on the surface of the liver during abdominal surgery and submit a specimen for pathological examination to exclude metastatic disease [ 9 ], which was the case of our first patient.…”

Section: Discussionmentioning

confidence: 99%

Biliary hamartoma: a report of two cases

Haloui¹,

Kariche²,

Aissaoui³

et al. 2022

Pan Afr Med J

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Biliary hamartoma, also known as biliary micro hamartoma or Von Meyenburg complex, is a rare benign liver lesion, thought to be a ductal plate malformation rather than a true neoplasm. It is often seen incidentally on imagery or surgery as multiple small subcapsular nodules, scattered throughout the liver, making it likely to be mistaken for metastatic nodules. The histological presentation can also be deceptive, leading to the misdiagnosis of an adenocarcinoma of hepato-biliary differentiation or a metastasis. We hereby present two cases of biliary hamartoma, found incidentally on imagery and surgery, the first one in a 94-year-old woman, and the second in a 48-year-old man, which was initially misdiagnosed as an adenocarcinoma, along with a discussion of key clinical and pathological findings to help avoid this diagnostic pitfall.

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Section: Discussionmentioning

confidence: 99%

Biliary hamartoma: a report of two cases

Haloui¹,

Kariche²,

Aissaoui³

et al. 2022

Pan Afr Med J

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“…Biliary hamartomas are rare benign bile duct malformations consisting of multiple collections of localized ductlike structures that are lined by biliary epithelium and embedded in a fibrous stroma [1]. Hanns von Meyenburg, a Swiss pathologist, was the first to describe these lesions in 1918, and because of that are referred to as von Meyenberg complexes [2]. Interrupted remodeling of the ductal plates during late embryonic development of the intrahepatic bile ducts is thought to be responsible for the development of these malformations [3].…”

Section: Introductionmentioning

confidence: 99%

Multiple biliary hamartomas “von Meyenberg complexes” in a newborn: a unique observation

Hassan

2020

Egypt Liver Journal

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Background: Biliary hamartomas are rare benign bile duct malformations consisting of multiple collections of localized duct-like structures that are lined by biliary epithelium and embedded in a fibrous stroma. In general, these malformations are asymptomatic and are usually detected incidentally during imaging, surgical exploration, or autopsy. Previously, the definitive diagnosis of these lesions was possible only with liver biopsy. The use of advanced imaging modalities made it possible to establish the diagnosis when undoubtful imaging findings are evident. Case presentation: We present a 25-day-old male newborn who was referred to our institution with jaundice, abdominal distension, and abdominal U/S that revealed hepatomegaly and involvement of both lobes of the liver with diffuse cystic lesions of variable sizes and echogenicity without a diagnostic suggestion. We discuss the details of imaging findings that enabled to establish the diagnosis of multiple biliary hamartomas and brief the patient's status on follow-up. Conclusion: Up to our best knowledge, the diagnosis of multiple biliary hamartomas has not been previously reported among newborns, making this report an extremely rare if not a unique observation.

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“…Bile duct hamartomas (BDH), also referred to as von Meyenburg complex (VMC), were first described in 1918 (1), with a reported incidence of 5.6% in adults and 0.9% in children (2). An increasing number of patients with BDH may be diagnosed with the development of imaging modalities.…”

Section: Introductionmentioning

confidence: 99%