Case Report
Head and Neck Oncology
Large cell neuroendocrine carcinoma of the tongue base: case report of an unusual location with immunohistochemical analysis

https://doi.org/10.1016/j.ijom.2008.12.012Get rights and content

Abstract

A case of large cell neuroendocrine carcinoma (LCNEC) of the tongue base is described. It was characterized by solid tumor nests with central necrosis and rosette formation resembling basaloid squamous cell carcinoma. Immunohistochemical examination revealed that this tumor had neuroendocrine differentiation. It was diagnosed as LCNEC of the tongue base.

Pulmonary LCNEC is a well-established entity, but LCNEC also occurs in other organs. This is the first report of mucosal LCNEC in the oral cavity. Basal cells in the normal squamous epithelium around the tumor indicated positivity for neural cell adhesion molecule and N-cadherin. These cells were considered neuroendocrine-related cells in the lingual squamous epithelium, which are related to the tumorigenesis of mucosal LCNEC in the tongue base.

Section snippets

Case report

The patient was a 79-year-old Japanese man who had had liver carcinoma (well-differentiated hepatocellular carcinoma) and hypopharyngeal carcinoma, which had been completely resected about 1 year before. Partial hepatectomy was performed, followed by total pharyngo-laryngo-esophagectomy and bilateral neck dissection for hypopharyngeal cancer that was diagnosed as moderately differentiated squamous cell carcinoma (SqCC). He had been followed up, and a mass, 10 × 10 mm, had been found on the left

Discussion

LCNECs were formally diagnosed as poorly differentiated SqCC and previous reports have pointed out that LCNEC was often misdiagnosed as poorly differentiated SqCC or poorly differentiated adenocarcinoma5, 11. Basaloid carcinoma of the lung also resembles LCNEC, but basaloid carcinoma lacks neuroendocrine features and TTF-1 expression7, 8. The diagnosis of LCNEC in the present case is based on criteria established for tumors of the lung10. The criteria included large cells, with a polygonal

Conflict interest statement

We declare that we have no conflict of interest.

Acknowledgements

The authors thank the staff of the Pathology Division, Shizuoka Cancer Center Hospital and Research Institute, Shizuoka, Japan (Isamu Hayashi, Kaori Nagata, Sachiko Oono, Yoichi Watanabe, Hiroshi Tashiro, Masatake Honda, Kumiko Hirobe, Masato Abe, Mihoko Shimura, Chiho Sugiyama, Takuya Kawasaki, and Koji Muramatsu) for excellent technical assistance.

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    There are some histopathological differences between this case of oral cavity LCNEC and the two previous descriptions in the literature (see Table 1). The case patient’s tumor originated in the FOM and RMT, similar to that described by Krishnamurthy et al.2 Esmati et al.3 reported a LCNEC originating from the oral tongue, while Kusafuka et al.1 reported an oral cavity LCNEC originating from the base of the tongue. The tongue base is an oropharyngeal subsite, meaning there have been only two true published reports of oral cavity primary LCNEC prior to the present case report.

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    In the oral cavity, Merkel cells are considered the origin, because they preferentially scatter in the basal layer of the keratinized epithelium in the hard palate and gingiva.7,11 Some researchers have proposed that LCNEC of the tongue base arises from some special cells that have neuroendocrine characteristics and are located in the basal cell layer of the squamous epithelium.23 Thus, LCNEC has been suggested to originate from neuroendocrine cells.

  • A neuroendocrine tumor in the maxilla

    2012, Journal of Oral and Maxillofacial Surgery
  • Primary typical carcinoid tumour in the retromolar region with prominent squamous differentiation: A case report

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    The Merkel cells show a preferential distribution along the basal layer of the keratinized epithelium of the hard palate and gingiva where they are present as widely scattered clear cells occurring singly or in clusters6. The presence of a novel cluster of neuroendocrine cells with a non-Merkel cell phenotype has been recently described in the squamous epithelium of the human tongue base5. These neuroendocrine cells normally found in the oral cavity are thought by some authors to be the cells of origin of neuroendocrine carcinomas3,5,6.

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