A long-term recurrence-free survival of a patient with the mixed adeno-neuroendocrine bile duct carcinoma: A case report and review of the literature

Izumo, Wataru; Higuchi, Ryota; Yazawa, Takehisa; Uemura, Shuichiro; Matsunaga, Yutaro; Shiihara, Masahiro; Furukawa, Toru; Yamamoto, Masakazu

doi:10.1016/j.ijscr.2017.07.052

Cited by 8 publications

(11 citation statements)

References 27 publications

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“…Interestingly, in 43 studies (n = 61) where more than one diagnostic sample was available [11,[13][14][15][16][17][18][19][20][21][22][23][24][25][26][27][28]30,[32][33][34][36][37][38][39][41][42][43][44][45][46][47]49,50,53,55,[59][60][61][62]64,65,96], the initial diagnosis from the first sample collected (either cytological or histological) was in keeping with MiNEN or suspicion of MiNEN in 36.1% (n = 22) of cases, adenocarcinoma in 36.1% (n = 22), poorly differentiated neuroendocrine carcinoma in 21.3% (n = 13), and well differentiated neuroendocrine tumour in 6.6% (n = 4).…”

Section: Clinical-pathological Characteristics Treatment Modalitiesmentioning

confidence: 99%

Mixed Neuroendocrine Non-Neuroendocrine Neoplasms: A Systematic Review of a Controversial and Underestimated Diagnosis

Frizziero

Chakrabarty

Nagy

et al. 2020

JCM

115

140

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Mixed neuroendocrine non-neuroendocrine neoplasms (MiNENs) represent a rare diagnosis of the gastro-entero-pancreatic tract. Evidence from the current literature regarding their epidemiology, biology, and management is of variable quality and conflicting. Based on available data, the MiNEN has an aggressive biological behaviour, mostly driven by its (often high-grade) neuroendocrine component, and a dismal prognosis. In most cases, the non-neuroendocrine component is of adenocarcinoma histology. Due to limitations in diagnostic methods and poor awareness within the scientific community, the incidence of MiNENs may be underestimated. In the absence of data from clinical trials, MiNENs are commonly treated according to the standard of care for pure neuroendocrine carcinomas or adenocarcinomas from the same sites of origin, based on the assumption of a biological similarity to their pure counterparts. However, little is known about the molecular aberrations of MiNENs, and their pathogenesis remains controversial; molecular/genetic studies conducted so far point towards a common monoclonal origin of the two components. In addition, mutations in tumour-associated genes, including TP53, BRAF, and KRAS, and microsatellite instability have emerged as potential drivers of MiNENs. This systematic review (91 full manuscripts or abstracts in English language) summarises the current reported literature on clinical, pathological, survival, and molecular/genetic data on MiNENs.

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Section: Clinical-pathological Characteristics Treatment Modalitiesmentioning

confidence: 99%

Mixed Neuroendocrine Non-Neuroendocrine Neoplasms: A Systematic Review of a Controversial and Underestimated Diagnosis

Frizziero

Chakrabarty

Nagy

et al. 2020

JCM

115

140

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“…By searching the literature, we found that biliary MANEC is rare, and only one of the retrospective studies mentioned two cases of hepatic MANEC. But it lacked corresponding analysis [10] 0.10 cases of biliary MANEC patients were summarized [10][11][12][13][14][15][16][17][18]. Including 4 cases of distal bile duct, 2 cases of hilar bile duct, 1 case of common bile duct, 1 case of intrahepatic bile duct, 1 case of cystic duct, and 1 case of extrahepatic bile duct.…”

Section: Discussionmentioning

confidence: 99%

Mixed adenoneuroendocrine carcinoma of the hepatic bile duct: a case report and review of the literature

et al. 2020

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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.

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“…MANECs predominately occur in the colon, appendix and stomach, where neuroendocrine cells are diffusely distributed (4). MANECs arising from the EHBT are extremely rare, with a total of eight cases reported in the medical literature since the category was introduced in 2010 (13–20). The histogenesis of biliary MANEC remains under debate due to the scarcity of enterochromaffin (Kultchisky) cells in the normal bile duct (21).…”

Section: Discussionmentioning

confidence: 99%

“…The search identified 37 patients with NEN in the EHBT since 2010. Eventually, a total of 38 cases, including the present case, were analyzed (Table I) (9,10,13–20,28–53). Clinical characteristics and survival outcomes among different pathological types were compared, and they are summarized in Table II.…”

Section: Discussionmentioning

confidence: 99%

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Mixed adenoendocrine carcinoma in the extrahepatic biliary tract: A case report and literature review

et al. 2019

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Neuroendocrine neoplasm (NEN) comprises a group of tumors that exhibit neuroendocrine phenotypes. NEN is subclassified into neuroendocrine tumor (NET), neuroendocrine carcinoma (NEC) and mixed adenoendocrine carcinoma (MANEC), based on histopathological parameters. NEN in the extrahepatic biliary tract (EHBT) is uncommon. Little is known about its clinicopathological features and prognostic indicators. The present study presented a case of MANEC in the distal common bile duct (CBD) and reviewed previous cases of NENs in the EHBT to characterize the clinical settings of this disease entity and to identify influencing factors of survival outcomes. A 64-year-old Chinese woman presented with abdominal pain and jaundice. Imaging studies demonstrated malignant stenosis in the distal CBD. Bile duct brush cytology revealed small clusters of atypical cells. Following an initial diagnosis of distal cholangiocarcinoma (CCA), the patient underwent pancreaticoduodenectomy. Histological analysis combined with immunohistochemical investigation of the resected specimen revealed a collision tumor that was composed of poorly differentiated adenocarcinoma and NEC. Each histological component accounted for >30% of the tumor. The definitive diagnosis was a MANEC in the distal CBD. Multiple intrahepatic and pulmonary metastases were observed postoperatively over 8 months. The patient succumbed to the disease 12 months after surgery. In conclusion, NEN occurs infrequently in the EHBT, with NET being the predominant type. NEN in the EHBT is extremely challenging to diagnose preoperatively due to its tendency to mimic CCA. Patients with NEN in the EHBT exhibited extremely distinct oncology outcomes according to pathological types. Additionally, old age (>60 years) and the presence of tumor recurrence were associated with decreased survival of patients with NEN.

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A long-term recurrence-free survival of a patient with the mixed adeno-neuroendocrine bile duct carcinoma: A case report and review of the literature

Cited by 8 publications

References 27 publications

Mixed Neuroendocrine Non-Neuroendocrine Neoplasms: A Systematic Review of a Controversial and Underestimated Diagnosis

Mixed Neuroendocrine Non-Neuroendocrine Neoplasms: A Systematic Review of a Controversial and Underestimated Diagnosis

Mixed adenoneuroendocrine carcinoma of the hepatic bile duct: a case report and review of the literature

Mixed adenoendocrine carcinoma in the extrahepatic biliary tract: A case report and literature review

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