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Mixed adenoneuroendocrine carcinoma of the tongue arising within a congenital enteric cyst
Author(s) -
Ligthelm Louis J.,
Bunn Belinda K.,
Raubenheimer Erich J.,
Heerden Willie F. P.
Publication year - 2018
Publication title -
head and neck
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.012
H-Index - 127
eISSN - 1097-0347
pISSN - 1043-3074
DOI - 10.1002/hed.25117
Subject(s) - cyst , medicine , pathology , adenocarcinoma , tongue , gastrointestinal tract , cdx2 , biology , cancer , biochemistry , gene expression , homeobox , gene
Background Primary intestinal‐type adenocarcinoma of the tongue is rare. This represents the first reported case of a primary mixed adenoneuroendocrine carcinoma (MANEC) of the tongue arising within a congenital enteric cyst. Methods A 52‐year‐old man presented with a midline tongue mass that was connected to the mucosal surface with an epithelial‐lined sinus tract. Morphological examination and immunohistochemical profiling of the neoplasm were performed. Results Histopathological evaluation showed a primary MANEC arising within a preexisting congenital enteric cyst, comprising both adenocarcinoma and neuroendocrine components. The adenocarcinoma had a colonic‐type morphology and coexpressed CK7, CK20, and CDX2. Imaging and colonoscopy excluded a distant primary colorectal neoplasm. Conclusion The association of primary MANEC of the tongue with a gastrointestinal heterotopic cyst supports an origin from entrapped endodermal elements as opposed to salivary duct origin. This case raises the awareness of a rare yet prognostically important complication of a gastrointestinal heterotopic cyst.