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A variant of multicystic biliary hamartoma presenting as an intrahepatic cystic neoplasm

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Abstract

A rare case of an intrahepatic multicystic tumor is described. A 26-year-old man visited our hospital because of abdominal discomfort. Contrast-enhanced computed tomography and magnetic resonance cholangiopancreatography revealed a 10 × 7 cm multicystic tumor of the bile duct in the right side of the liver. The gross appearance of the tumor resembled an intraductal papillary neoplasm of the bile duct, and right hepatectomy with regional lymphadenectomy was performed. Histologically, these cystic lesions were composed of variably and irregularly dilated duct structures lined by columnar epithelium resembling bile duct lining. There were no atypical cells and no papillary growth of the epithelial cells. Interestingly, the dilated ducts contained inspissated bile, and the inter-cystic parenchyma contained variable but irregularly distributed and hamartomatous hepatic parenchyma with an abnormal lobular pattern. Though it had atypical features of a hamartoma in some aspects (age, smooth muscle), this case could finally be regarded as a variant of multicystic biliary hamartoma.

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Abbreviations

IPNB:

Intraductal papillary neoplasm

ICGR15:

Indocyanine green retention rate at 15 min

LHL15:

Liver uptake ratio

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Disclosures

Conflict of Interest: The authors declare that they have no conflict of interest.

Human/Animal Rights: All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation.

Informed Consent: Informed consent was obtained from all patients to be included in the study.

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Authors and Affiliations

  1. Division of Surgical Oncology, Department of Translational Medical Sciences, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki, 852-8501, Japan

    Tetsuro Tominaga, Takafumi Abo, Takeshi Nagayasu & Atsushi Nanashima

  2. Department of Pathology, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan

    Naoe Kinoshita

  3. Department of Radiology, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan

    Tomonori Murakami

  4. Department of Human Pathology, Kanazawa University Graduate School of Medicine, Kanazawa, Japan

    Yasunori Sato, Yasuni Nakanuma & Kenich Harada

  5. Department of Gastroenterology and Hepatology, Nijigaoka Hospital, Nagasaki, Japan

    Junichi Masuda

  6. Division of Surgical Oncology and Regulation of Organ Function, Miyazaki University Graduate School of Medicine, Miyazaki, Japan

    Atsushi Nanashima

Authors
  1. Tetsuro Tominaga
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  2. Takafumi Abo
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  3. Naoe Kinoshita
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  4. Tomonori Murakami
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  5. Yasunori Sato
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  6. Yasuni Nakanuma
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  7. Kenich Harada
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  8. Junichi Masuda
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  9. Takeshi Nagayasu
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  10. Atsushi Nanashima
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Corresponding author

Correspondence to Tetsuro Tominaga.

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Tominaga, T., Abo, T., Kinoshita, N. et al. A variant of multicystic biliary hamartoma presenting as an intrahepatic cystic neoplasm. Clin J Gastroenterol 8, 162–166 (2015). https://doi.org/10.1007/s12328-015-0574-y

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  • DOI: https://doi.org/10.1007/s12328-015-0574-y

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