Mixed adenoneuroendocrine carcinoma of the hepatic bile duct: a case report and review of the literature
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CASE REPORT
Mixed adenoneuroendocrine carcinoma of the hepatic bile duct: a case report and review of the literature Sulai Liu1†, Zhendong Zhong1,2†, Meng Xiao3, Yinghui Song1, Youye Zhu4, Bo Hu1, Zengpeng Sun1, Weimin Yi1* and Chuang Peng1*
Abstract Background: The World Health Organization’s updated classification of digestive system neuroendocrine tumors in 2010 first proposed the classification of mixed adenoneuroendocrine carcinoma (MANEC). The incidence of biliary malignant tumors with neuroendocrine tumors accounts for less than 1% of all neuroendocrine tumors. Moreover, the incidence of hilar bile duct with MANEC is very rare. Case presentation: A 65-year-old female patient came to our hospital for repeated abdominal pain for more than 4 months and skin sclera yellow staining for 1 week. Contrast-enhanced computed tomography imaging and magnetic resonance results suggested a hilar tumor for Bismuth-Corlette Type II. The patient underwent radical surgery for hilar cholangiocarcinoma. Finally, the patient was diagnosed with hilar bile duct MANEC, staged 1 (pT1N0M0) based on the eighth edition of the AJCC. Histopathology showed that the tumor was a biliary tumor with both adenocarcinoma and neuroendocrine carcinoma. No evidence of recurrence and metastasis after 20 months of follow-up. Conclusions: We first reported a MANEC that originated in the hilar bile duct. As far as we known, there were few reports of biliary MANEC, and the overall prognosis was poor. We also found that the higher the Ki-67 index, the worse the prognosis of this type of patient. Radical surgery is the most effective treatment. Keywords: Neuroendocrine tumor, Mixed adenoneuroendocrine carcinoma, Hepatic bile duct Background The most common pathological type of hilar cholangiocarcinoma is adenocarcinoma, accounting for more than 90%. Others mainly include gland-squamous tumors, intraductal papillary tumors, mucinous gland tumors [1]. Mixed adenine neuroendocrine carcinoma (MANEC) is defined as a compound tumor of adenocarcinoma or squamous cell carcinoma mixed with neuroendocrine carcinoma (NEC) or neuroendocrine tumor (NET), with *Correspondence: [email protected]; [email protected] † Sulai Liu and Zhendong Zhong have contributed equally to this work 1 Department of Hepatobiliary Surgery, Hunan Provincial People’s Hospital/The First Affiliated Hospital of Hunan Normal University, Changsha, Hunan Province, People’s Republic of China Full list of author information is available at the end of the article
each tumor accounting for at least 30% of the tumor. Meanwhile, these two histological components must be malignant [2]. Neuroendocrine neoplasms (NENs) are distributed in the neuroendocrine system, mainly from the neuroendocrine cells of the digestive system and respiratory tract. The diagnosis depends on histopathological examination [3].NENs can form a mixed carcinoma with malignant-tumors in the primary site, and neuroendocrine tumors of biliary mixed adenocarcinoma are rare in clinical practice [4]. Since the conce
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