The clinical profiles, management, and prognostic factors of biliary mixed neuroendocrine nonneuroendocrine neoplasms

Wen, Lijia; Chen, Junhong; Xu, Hong‐Ji; Yu, Qiong; Deng, Yu

doi:10.1097/md.0000000000023271

Cited by 10 publications

(15 citation statements)

References 50 publications

(3 reference statements)

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“…Due to the few numbers of cases and their heterogeneity, the standard treatment for MINEN, especially involving adjuvant therapy, is still controversial. In their systematic review of biliary MINEN, Wen et al reported that incomplete resection, Ki-67 index (≥ 50), tumor stage, NEC grade, and non-NEC grade were prognostic factors for poor overall survival, and that adjuvant chemoradiotherapy for those patients may contribute to better overall survival [ 8 ]. Yoshimachi et al also reported in their literature review of ampullary MANEC that multidisciplinary treatment including chemotherapy may improve the prognosis of patients with MANEC [ 9 ].…”

Section: Discussionmentioning

confidence: 99%

“…According to the literature review reported by Yoshimachi et al, seven out of 15 cases of ampullary MINEN received adjuvant chemotherapy (oxaliplatin-based combination chemotherapy; n = 5, S-1; n = 2) [ 9 ]. In addition, Wen et al reported that 22 out of 51 patients with biliary MINEN received adjuvant chemotherapy, and 21 of 22 received platinum-based combination therapy [ 8 ]. Thus, platinum-based combination therapy is likely to be the mainstream adjuvant chemotherapy for MINEN following radical resection.…”

Section: Discussionmentioning

confidence: 99%

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Surgical resection of mixed neuroendocrine-non-neuroendocrine neoplasm in the biliary system: a report of two cases

et al. 2022

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Background Mixed neuroendocrine-non-neuroendocrine neoplasm (MINEN) is a rare disease and there is scarce literature on its diagnosis, treatment, and prognosis. We encountered two unusual cases of MINEN in the biliary tract, one in the ampulla of Vater and the other in the distal bile duct. In this report, we describe the clinical course of these two cases in detail. Case presentation Case 1: A 69-year-old woman presented with a chief complaint of epigastric pain. When endoscopic sphincterotomy and retrograde biliary drainage were performed for gallstone pancreatitis, an ulcerated lesion was found in the ampulla of the Vater. Based on the biopsy results, the lesion was diagnosed as the ampulla of Vater carcinoma and subtotal stomach-preserving pancreatoduodenectomy (SSPPD) was performed. Postoperative histopathological examination revealed the coexistence of adenocarcinoma and neuroendocrine carcinoma components, consistent with the diagnosis of MINEN. In addition, lymph node metastasis was found on the dorsal side of the pancreas and the metastatic component was adenocarcinoma. Adjuvant chemotherapy with etoposide and cisplatin was administered for 6 months, and presently the patient is alive without recurrence 64 months after surgery. Case 2: A 79-year-old man presented with a chief complaint of anorexia. Cholangiography showed severe stenosis of the distal bile duct. A biopsy was conducted from the stenotic lesion and it revealed the lesion to be adenocarcinoma. A diagnosis of distal bile duct carcinoma was made, and SSPPD was performed. Histopathological examination revealed the coexistence of adenocarcinoma and neuroendocrine carcinoma components, and the tumor was confirmed as MINEN of the distal bile duct. No adjuvant chemotherapy was administered due to the poor performance status. 7 months later, the patient was found to have a liver metastasis. Conclusion We experienced two valuable cases of biliary MINEN. To identify better treatments, it is important to consider the diversity of individual cases and to continue sharing a variety of cases with different presentations.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Surgical resection of mixed neuroendocrine-non-neuroendocrine neoplasm in the biliary system: a report of two cases

et al. 2022

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“…Furthermore, MiNEN with predominant NEC components are more likely to metastasize to LNs [ 3 ]. Several authors reported that patients with resectable cases of MiNEN who underwent complete surgical resection had overall better prognosis [ 2 , 8 , 9 ]. Chen et al [ 1 ] described the aggressive nature of the gallbladder NEC, and reported that patients with NEC had significantly higher rate of LN metastases and poorer prognosis, when compared to patients with gallbladder adenocarcinoma.…”

Section: Discussionmentioning

confidence: 99%

“…Although each component of MiNEN must account for at least 30% of the whole neoplasm to meet the criteria for MiNEN, several authors suggested that the prognosis was influenced by the most aggressive histological components, regardless of the percentage of those components [ 8 , 9 , 15 ]. Our patient had less than 30% of NEC components, who did not meet the criteria for MiNEN, but presented with diffusely metastatic LNs with NEC components.…”

Section: Discussionmentioning

confidence: 99%

Focal neuroendocrine carcinoma mixed with adenocarcinoma of the gallbladder with aggressive lymph node metastasis in a patient who did not meet the mixed neuroendocrine–non-neuroendocrine neoplasm criteria

Kadota

Funakoshi

Hirose

et al. 2021

Clin J Gastroenterol

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A 70-year-old Japanese woman who was treated for interstitial pneumonia (IP) with steroid therapy developed cholecystitis. A serial computed-tomography (CT) imaging showed irregular thickness of the fundus wall of the gallbladder and two rapidly enlarged lymph nodes (LNs): number (no.) 12 and no. 8a. Positron-emission tomography-computed tomography (PET-CT) scan showed an abnormal uptake at the site of the gallbladder tumor and those LNs. We subsequently performed open radical cholecystectomy and LN dissection of the no. 12 and 8a LNs, following complete remission of IP. The histology showed gallbladder adenocarcinoma, with a single focus of neuroendocrine carcinoma (NEC) component of less than 30%; Ki-67 index > 80%, synaptophysin (Syn) (+), chromogranin A (CgA) (+), and clusters of differentiation (CD) 56 (+) (T2bN1M0, Stage IIIB). LN no. 8a was diffusely metastatic with NEC components. LN no. 12c, which was adjacent to the cystic duct, revealed necrosis without apparent tumor cells, but was highly suspicious for tumor necrosis. The final diagnosis was adenocarcinoma of the gallbladder with focal NEC (< 30%), which did not meet the criteria for mixed neuroendocrine–non-neuroendocrine neoplasm (MiNEN). Postoperatively, she completed 4 cycles of adjuvant chemotherapy for NEC (Cisplatin plus Etoposide), and no recurrence was observed after 12 months.

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“…A recent systemic review based on 53 studies to predict the clinicopathological features and prognosis of biliary MiNENs, including gallbladder MiNENs, showed a median overall survival time of 21 mo. In addition, radical resection and small morphological subtype were independent prognostic factors associated with higher overall survival, and radical resection (R0) and younger age (< 65 years) were associated with higher recurrence-free survival time[ 115 ].…”

Section: Organ-specific Clinicopathological Findingsmentioning

confidence: 99%

Mixed neuroendocrine–nonneuroendocrine neoplasms of the gastrointestinal system: An update

Elpek¹

2022

WJG

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Mixed neuroendocrine-nonneuroendocrine neoplasms (MiNENs) of the digestive tract are a rare heterogeneous group of tumors that present many challenges in terms of diagnosis and treatment. Over the years, the diagnostic criteria, classification, and clinical behavior of these tumors have been the subjects of ongoing debate, and the various changes in their nomenclature have strengthened the challenges associated with MiNENs. This review is performed to provide an understanding of the key factors involved in the evolution of the designation of these tumors as MiNEN, highlight the current diagnostic criteria, summarize the latest data on pathogenesis and provide information on available treatments. Moreover, this work seeks to increase the awareness about these rare neoplasms by presenting the clinicopathological features and prognostic factors that play important roles in their behavior and discussing their different regions of origin in the gastrointestinal system (GIS). Currently, the MiNEN category also includes tumors in the GIS with a nonneuroendocrine component and epithelial tumors other than adenocarcinoma, depending on the organ of origin. Diagnosis is based on the presence of both morphological components in more than 30% of the tumor. However, this value needs to be reconfirmed with further studies and may be a limiting factor in the diagnosis of MiNEN by biopsy. Furthermore, available clinicopathological data suggest that the inclusion of amphicrine tumors in the definition of MiNEN is not supportive and warrants further investigation. The diagnosis of these tumors is not solely based on immunohistochemical findings. They are not hybrid tumors and both components can act independently; thus, careful grading of each component separately is required. In addition to parameters such as the metastatic state of the tumor at the time of diagnosis and the feasibility of surgical resection, the aggressive potential of both components has paramount importance in the choice of treatment. Regardless of the organ of origin within the GIS, almost MiNENs are tumors with poor prognosis and are frequently encountered in the elderly and men. They are most frequently reported in the colorectum, where data from molecular studies indicate a monoclonal origin; however, further studies are required to provide additional support for this origin.

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The clinical profiles, management, and prognostic factors of biliary mixed neuroendocrine nonneuroendocrine neoplasms

Cited by 10 publications

References 50 publications

Surgical resection of mixed neuroendocrine-non-neuroendocrine neoplasm in the biliary system: a report of two cases

Surgical resection of mixed neuroendocrine-non-neuroendocrine neoplasm in the biliary system: a report of two cases

Focal neuroendocrine carcinoma mixed with adenocarcinoma of the gallbladder with aggressive lymph node metastasis in a patient who did not meet the mixed neuroendocrine–non-neuroendocrine neoplasm criteria

Mixed neuroendocrine–nonneuroendocrine neoplasms of the gastrointestinal system: An update

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