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      • Record: found
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      A case report of primary neuroendocrine carcinoma of the perihilar bile duct

      case-report

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          Abstract

          Background

          Although neuroendocrine tumors are most commonly found in the digestive system, neuroendocrine tumors originating from the bile duct are rare, and neuroendocrine carcinomas derived from the perihilar bile duct are extremely rare. This report presents the clinical course and clinicopathological features of neuroendocrine carcinomas arising from the extrahepatic bile duct.

          Case presentation

          A 70-year-old Japanese woman was preoperatively diagnosed with perihilar cholangiocarcinoma, and a radical resection with an extended left hepatic lobectomy and a choledochojejunostomy was performed. From the histopathological findings, we diagnosed the tumor as a neuroendocrine carcinoma of the bile duct (small cell type) with lymph node metastasis. The patient was treated with the same adjuvant chemotherapy as that used for small cell carcinoma of the lung. At 10 months after surgery, there was no recurrence of the disease.

          Conclusion

          Neuroendocrine carcinoma of the extrahepatic biliary tracts is a very rare and highly malignant disease with a poor prognosis. A multidisciplinary approach could improve the prognosis for this neoplasm.

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          Most cited references35

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          Extrapulmonary small-cell carcinoma: a single-institution experience.

          Kihyun Kim, Jinny Park, T. J. Kim … (2004)
          Extrapulmonary small-cell carcinoma (EPSCC) has been recognized as a clinicopathological entity distinct from small-cell carcinoma (SCC) of the lung. This study aimed to review the clinical features, therapy and natural course of patients with EPSCC in Oriental single-institution series. We retrospectively reviewed the medical records of patients with SCC between September 1995 and December 2002. Study eligibility required that patients had pathologically proven SCC in sites other than lung and normal radiological findings of the chest and normal sputum cytology or negative bronchoscopic findings. Twenty-four patients with EPSCC were identified and primary sites were various: uterine cervix in seven (29%), urinary bladder in five, colon or rectum in three, kidney in two and stomach, esophagus, pancreas, common bile duct, larynx, parotid gland, thymus in one each. Sixteen patients (66.7%) had limited disease (LD) and eight had extensive disease (ED). Patients with ED received mostly platinum-based chemotherapy, for which the response rate was 57%, but showed an aggressive natural history, with median overall survival (OS) of 9.2 months. Patients with LD were treated with a variety of therapeutic modalities. LD SCC of the cervix showed a favorable clinical course, with five patients being disease-free with a median follow-up of 28.4 months. Patients with LD SCC of sites other than cervix had an aggressive course with a median OS of 9.6 months. EPSCC was identified in various sites, with the most common primary site being the uterine cervix. Regardless of the primary site or disease stage, EPSCC of sites other than cervix was usually a fatal disease with a discouraging outcome for various treatment modalities.
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            Small cell carcinoma presenting as an extrapulmonary neoplasm: sites of origin and response to chemotherapy.

            John Minna, D Ihde, R Levenson … (1981)
            Eight (4%) of 203 consecutive prospectively staged and treated patients with small cell carcinoma (SCC) had no evidence of pulmonary or mediastinal tumor on chest roentgenogram or at fiberoptic bronchoscopy at the time of diagnosis. There were two distinct clinical presentations in these SCC patients with exclusively extrapulmonary tumors. Four had discrete localized extrapulmonary neoplasms, presumably originating in these sites. In the other 4 cases with either regional or widely metastatic disease, no obvious primary tumor could be documented in the lungs or elsewhere. One complete and two partial responses to chemotherapy (duration 6 to greater than 11 mo) occurred in 6 evaluable patients. Two remaining patients were inevaluable for response because they received adjuvant chemotherapy after irradiation or excision of the primary tumor and are free of disease at 15 and 28 months. Results document two clinicopathologic entities of extrapulmonary SCC, more firmly establish that it can be responsive to chemotherapy, and encourage systemic therapy as part of initial treatment planning.
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              Neuroendocrine carcinoma of the extrahepatic bile duct: case report and literature review.

              Eizaburo Sasatomi, Michael Nalesnik, J Wallis Marsh (2013)
              Neuroendocrine carcinoma (NEC) of the extrahepatic bile duct is rare, and only 22 cases have been reported. Only two of these were large-cell NEC (LCNEC); the vast majority were small-cell NEC. Here, we report a third case of LCNEC of the extrahepatic bile duct. A 76-year-old male presented to a local hospital with painless jaundice. Imaging studies revealed a tumor at the hepatic hilum. The patient underwent right hepatic lobectomy, bile duct resection, and cholecystectomy. The resection specimen showed a 5.0-cm invasive neoplasm involving the hilar bile ducts and surrounding soft tissue. Histologically, the tumor consisted of nests of medium to large cells with little intervening stroma. The tumor invaded a large portal vein branch. All four excised lymph nodes were positive for metastasis, and metastatic deposits were also present in the gallbladder wall. The tumor was diffusely positive for synaptophysin and focally positive for chromogranin A. Approximately 70%-80% of the tumor cells were positive for Ki-67, indicating strong proliferative activity. A diagnosis of LCNEC was made. A few bile ducts within and adjacent to the invasive tumor showed dysplasia of the intestinal phenotype and were focally positive for synaptophysin and chromogranin A, suggesting that the dysplastic intestinal-type epithelium played a precursor role in this case. A postoperative computer tomography scan revealed rapid enlargement of the abdominal and retroperitoneal lymph nodes. The patient died 21 d after the operation. NEC of the bile duct is an aggressive neoplasm, and its biological characteristics remain to be better defined.
              Article 0 comments Cited 15 times – based on 0 reviews     Review now
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                Author and article information

                Contributors
                Yasuhiro Kihara: +81-(0) 96-384-2111 , flinge21@yahoo.co.jp
                Hiroshi Yokomizo: h-yokomizo@kumamoto-med.jrc.or.jp
                Takahiro Urata: tagatame@yahoo.co.jp
                Michiko Nagamine: nagamine.michiko.jx@kumamoto-med.jrc.or.jp
                Toshihiko Hirata: krchirata@kumamoto-med.jrc.or.jp
                Journal
                Journal ID (nlm-ta): BMC Surg
                Journal ID (iso-abbrev): BMC Surg
                Title: BMC Surgery
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1471-2482
                Publication date (Electronic): 10 December 2015
                Publication date PMC-release: 10 December 2015
                Publication date Collection: 2015
                Volume: 15
                Electronic Location Identifier: 125
                Affiliations
                [ ]Division of General Surgery, Japanese Red Cross Kumamoto Hospital, Nagamineminami 2-1-1, Higashiku, Kumamoto city, Kumamoto 861-8520 Japan
                [ ]Division of Gastroenterology, Japanese Red Cross Kumamoto Hospital, Kumamoto, Japan
                [ ]Division of Diagnostic Pathology, Japanese Red Cross Kumamoto Hospital, Kumamoto, Japan
                Article
                Publisher ID: 116
                DOI: 10.1186/s12893-015-0116-z
                PMC ID: 4676104
                SO-VID: fcb5a80a-7d91-4a80-be40-b062a050e8f3
                Copyright © © Kihara et al. 2015
                License:

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Date received : 18 September 2015
                Date accepted : 7 December 2015
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2015

                ScienceOpen disciplines: Surgery
                Keywords: neuroendocrine carcinoma,bile duct,small cell carcinoma
                Data availability:
                ScienceOpen disciplines: Surgery
                Keywords: neuroendocrine carcinoma, bile duct, small cell carcinoma

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