Conversion surgery after extensive chemotherapy for stage IV mixed adenoneuroendocrine carcinoma (MANEC) of the gallbladder: clinical implications from the patterns of response and recurrence

Kamei, Koichi; Shindoh, Junichi; Kiya, Yoshitaka; Matsumoto, Ippei; Hashimoto, Masaji; Takeyama, Yoshifumi

doi:10.1007/s12328-019-01053-y

Cited by 7 publications

(5 citation statements)

References 19 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…[ 31 , 32 ] In fact, there have been 4 reports of MiNEN in patients with PMJ (Table 1 ). [ 11 – 14 ] These cases support the mechanism of MiNEN development in patients with PMJ outlined above. Five cases with Japanese patients, including our case, were reported.…”

Section: Discussionsupporting

confidence: 63%

See 1 more Smart Citation

Mixed neuroendocrine non-neuroendocrine neoplasm of the gallbladder complicated by a pancreaticobiliary maljunction of a non-dilated biliary duct

et al. 2021

View full text Add to dashboard Cite

Rationale: Mixed neuroendocrine non-neuroendocrine neoplasm (MiNEN) is a rare tumor. MiNEN of the gallbladder (GB) with pancreaticobiliary maljunction (PMJ) is extremely rare. The origin of MiNEN of the GB remains unknown; the biliary tract normally lacks neuroendocrine cells. MiNEN of the GB has a poor prognosis; because of its rarity, no treatment or management guidelines have been established yet. Patient concerns: A 47-year-old male presenting with right hypochondrial pain and malaise for 3 months was referred to our hospital for further management. Diagnosis: The neuron-specific enolase level was increased. Contrast-enhanced computed tomography revealed a mass of 70 mm in size with unclear boundaries in the liver. The GB was surrounded by this mass, narrowing the lumen of the GB. Many swollen lymph nodes were observed in the hepatoduodenal ligament. Endoscopic retrograde cholangiopancreatography revealed a PMJ with a non-dilated biliary duct. A percutaneous biopsy was performed on the liver mass, and the pathological findings were neuroendocrine carcinoma (NEC) (small cell type). We diagnosed a NEC of the GB, T3N1M0, stage IIIB (Union for International Cancer Control, 7th edition). Interventions: Because of advanced lymph node metastasis, we considered this tumor difficult to cure solely by surgical intervention. After initial chemotherapy consisting of cisplatin and irinotecan, a marked reduction in both tumor and lymph node sizes enabled conversion surgery. The pathological diagnosis of the resected tumor was MiNEN consisting of NEC and adenocarcinoma. The primary lesion was the adenocarcinoma occupying the luminal side of the GB. As a postsurgical treatment, the patient received additional irradiation therapy to the common hepatic duct and liver stump because of positive surgical margins. Outcomes: At 13 months postoperatively, computed tomography findings revealed the appearance of a hypervascular liver tumor, and laboratory data showed increased serum neuron-specific enolase levels. Chemotherapy was unsuccessful, leading to the death of the patient 36 months from the date of diagnosis. Lessons: There are several reports on the development of MiNEN of the GB. In our case, a PMJ-related adenocarcinoma of the GB transdifferentiated into NEC. Further accumulation of cases is necessary to establish a treatment strategy for MiNEN of the GB.

show abstract

Section: Discussionsupporting

confidence: 63%

“…MiNEN of the GB with pancreaticobiliary maljunction (PMJ) is extremely rare. [11][12][13][14] PMJ is a rare congenital anomaly and mostly prevalent in Asians, especially in the Japanese population. The prevalence rate of PMJ is 0.03% in Japan.…”

Section: Introductionmentioning

confidence: 99%

Mixed neuroendocrine non-neuroendocrine neoplasm of the gallbladder complicated by a pancreaticobiliary maljunction of a non-dilated biliary duct

et al. 2021

View full text Add to dashboard Cite

show abstract

“…There is currently no consensus on the postoperative adjuvant chemotherapy, but it is usually determined based on the most aggressive components of MiNEN [ 8 , 7 , 12 ]. The latest National Comprehensive Cancer Network (NCCN) guideline recommends several therapy options for resectable gallbladder cancer, including; (a) observation without any additional therapy, (b) neoadjuvant/adjuvant systemic chemotherapy, and (c) fluoropyrimidine + radiotherapy (RT) [ 13 ].…”

Section: Discussionmentioning

confidence: 99%

“…If the predominant component of the MiNEN is NEC, the tumor tends to behave more aggressively, presenting with frequent LN metastases [ 3 , 7 ]. Gallbladder NEC is notorious for high recurrence rate, poor morbidity, and mortality [ 1 , 5 , 11 , 12 ]. The patient will continue to be closely monitored.…”

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

View full text Add to dashboard Cite

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.

show abstract

“…Herein, we report a case of primary gMANEC, the first reported in Latin America and the 24 th worldwide [ 5 – 22 ]. This case has been reported in line with the SCARE criteria [ 23 ].…”

Section: Introductionmentioning

confidence: 99%

Rare Mixed Adenoneuroendocrine Carcinoma of the Gallbladder: Case Report and Review of Literature

et al. 2021

View full text Add to dashboard Cite

Patient: Female, 58-year-old Final Diagnosis: Mixed adenoneuroendocrine carcinoma of the gallbladder Symptoms: Abdominal pain • asthenia Medication: — Clinical Procedure: — Specialty: Surgery Objective: Rare disease Background: Mixed adenoneuroendocrine carcinoma of the gallbladder (gMANEC) is an extremely rare cancer. Most of the cases are reported in Asia, North America, and Europe, with no cases reported in Latin America; this is the first report for this region, and the 24 th case reported worldwide. Case Report: A 68-year-old woman was referred to our department due to asthenia and moderate abdominal pain in the right upper quadrant for 6 months, with imaging examinations showing a solid heterogeneous expansive lesion in gallbladder topography and segment IV of the liver. The MRI displayed an expansive and heterogeneous lesion with inaccurate limits in the gallbladder affecting segment IVb of the liver, in addition to lymphadenopathy in the hepatic hilum. A cholecystectomy with resection of segments IV-B and V of the liver (radical cholecystectomy) and hepatic hilar lymphadenectomy were performed. Anatomopathological examination and immunohisto-chemistry confirmed a primary mixed adenoneuroendocrine carcinoma of the gallbladder. The patient received adjuvant chemotherapy and radiotherapy; however, after the patient reported experiencing low back pain, a CT was performed, revealing retroperitoneal metastasis, and the radiotherapy was interrupted. Currently, the patient has a stable disease, following a protocol of 5-Fluorouracil and somatostatin, and she reports having low back pain of low intensity. Conclusions: This is the 24 th gMANEC case reported in the literature. The tumor was successfully resected; however, the patient presented retroperitoneal metastasis 6 months after surgery, despite combined adjuvant therapy.

show abstract

Conversion surgery after extensive chemotherapy for stage IV mixed adenoneuroendocrine carcinoma (MANEC) of the gallbladder: clinical implications from the patterns of response and recurrence

Cited by 7 publications

References 19 publications

Mixed neuroendocrine non-neuroendocrine neoplasm of the gallbladder complicated by a pancreaticobiliary maljunction of a non-dilated biliary duct

Mixed neuroendocrine non-neuroendocrine neoplasm of the gallbladder complicated by a pancreaticobiliary maljunction of a non-dilated biliary duct

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

Rare Mixed Adenoneuroendocrine Carcinoma of the Gallbladder: Case Report and Review of Literature

Contact Info

Product

Resources

About