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Mixed neuroendocrine–non-neuroendocrine neoplasm with mucinous adenocarcinoma and amphicrine carcinoma components in the bile duct: an autopsy case

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Abstract

We report the first case of bile duct mixed neuroendocrine–non-neuroendocrine neoplasm (MiNEN) that had a mucinous carcinoma component. An 88-year-old man with biliary obstruction was diagnosed as having distal bile duct cancer using imaging examinations and endoscopic biopsy. The patient received the best supportive care without surgical resection for 13 months until death. An autopsy revealed a bulky mass involving the distal bile duct and multiple metastases in intra-abdominal lymph nodes, the liver, and the lungs. The primary cancer was microscopically diagnosed as a MiNEN, which consisted of mucinous adenocarcinoma and large cell-type neuroendocrine carcinoma (NEC) components. Metastatic lesions in the liver and lungs were composed of only NEC with rich extracellular mucin without adenocarcinoma cells. Using electron microscopy and immunohistochemistry, it was proved that all NEC cells in both primary and metastatic lesions had amphicrine features. On the basis of pathological findings, we thought that the MiNEN was initially derived from a mucinous adenocarcinoma that dedifferentiated to amphicrine NEC cells with mucin production.

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References

  1. WHO Classification of Tumors Editorial Board. WHO Classification of Tumours. Digestive System Tumours. Fifth Edition, IARC Press, Lyon, 2019

  2. Bosman F, Carneiro F, Hruban R, et al. WHO classification of tumours of the digestive system. 4th ed. Lyon: IARC Press; 2010.

    Google Scholar 

  3. La Rosa S, Sessa F, Uccella S. Mixed neuroendocrine-nonneuroendocrine neoplasms (MiNENs): unifying the concept of a heterogeneous group of neoplasms. Endocr Pathol. 2016;27:284–311.

    Article  PubMed  Google Scholar 

  4. de Mestier L, Cros J, Neuzillet C, et al. Digestive system mixed neuroendocrine-non-neuroendocrine neoplasms. Neuroendocrinology. 2017;105:412–25.

    Article  PubMed  Google Scholar 

  5. Hanamatsu Y, Saigo C, Asano N, et al. A case of gastric amphicrine signet-ring cell carcinoma. Clin Pathol. 2019;12:2632010X19880535.

  6. Yu R, Jih L, Zhai J, et al. Mixed acinar-endocrine carcinoma of the pancreas: new clinical and pathological features in a contemporary series. Pancreas. 2013;42:429–35.

    Article  CAS  PubMed  Google Scholar 

  7. Fujita Y, Uesugi N, Sugimoto R, et al. Gastric mixed neuroendocrine-non-neuroendocrine neoplasm (MiNEN) with pancreatic acinar differentiation: a case report. Diagn Pathol. 2019;14:38.

    Article  PubMed  PubMed Central  Google Scholar 

  8. Russo S, Russo F, Maiello FM, et al. Biphasic large cell neuroendocrine carcinoma—pure mucinous carcinoma of the gallbladder (MANEC): a unique combination. Pathologica. 2012;104:185–9.

    CAS  PubMed  Google Scholar 

  9. Harada K, Sato Y, Ikeda H, et al. Clinicopathologic study of mixed adenoneuroendocrine carcinomas of hepatobiliary organs. Virchows Arch. 2012;460:281–9.

    Article  PubMed  Google Scholar 

  10. Acosta AM, Wiley EL. Primary biliary mixed adenoneuroendocrine carcinoma (MANEC): a short review. Arch Pathol Lab Med. 2016;140:1157–62.

    Article  CAS  PubMed  Google Scholar 

  11. Ludmir EB, McCall SJ, Cardona DM, et al. Mixed adenoneuroendocrine carcinoma, amphicrine type, of the small bowel. Am J Clin Pathol. 2016;145:703–9.

    Article  PubMed  Google Scholar 

  12. de Mestier L, Cros J. Digestive system mixed neuroendocrine-non-neuroendocrine neoplasms (MiNEN). Ann Endocrinol. 2019;80:172–3.

    Article  Google Scholar 

  13. Ganesan K, Achmad E, Sirlin CB, et al. Amphicrine carcinoma of the liver. Ann Diagn Pathol. 2011;15:355–7.

    Article  PubMed  Google Scholar 

  14. Carriaga MT, Henson DE. Liver, gallbladder, extrahepatic bile ducts, and pancreas. Cancer. 1995;75:171–90.

    Article  CAS  PubMed  Google Scholar 

  15. Egawa S, Toma H, Ohigashi H, et al. Japan Pancreatic cancer registry; 30th year anniversary: Japan Pancreas Society. Pancreas. 2012;41:985–92.

    Article  PubMed  Google Scholar 

  16. Chun YK. Neuroendocrine tumors of the female reproductive tract: a literature review. J Pathol Transl Med. 2015;49:450–61.

    Article  PubMed  PubMed Central  Google Scholar 

Download references

Acknowledgements

We would like to express our appreciation to Mr. Kinji Ishida from Technical Support Center for Life Science Research, Iwate Medical University for his contributions toward the use of electronic microscopy and diagnosis.

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Correspondence to Toji Murabayashi.

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Toji Murabayashi, Yoshihide Kanno, Takashi Odaira, Shinsuke Koshita, Takahisa Ogawa, Hiroaki Kusunose, Toshitaka Sakai, Keisuke Yonamine, Kazuaki Miyamoto, Fumisato Kozakai, Kazuki Endo, Yutaka Noda, Takashi Sawai and Kei Ito declare that they have no conflict of interest.

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Murabayashi, T., Kanno, Y., Odaira, T. et al. Mixed neuroendocrine–non-neuroendocrine neoplasm with mucinous adenocarcinoma and amphicrine carcinoma components in the bile duct: an autopsy case. Clin J Gastroenterol 16, 310–316 (2023). https://doi.org/10.1007/s12328-023-01763-4

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