{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,27]],"date-time":"2025-10-27T16:14:30Z","timestamp":1761581670492,"version":"3.37.3"},"reference-count":18,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2018,2,23]],"date-time":"2018-02-23T00:00:00Z","timestamp":1519344000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Head &amp; Neck"],"published-print":{"date-parts":[[2018,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Primary intestinal\u2010type 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.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A 52\u2010year\u2010old man presented with a midline tongue mass that was connected to the mucosal surface with an epithelial\u2010lined sinus tract. Morphological examination and immunohistochemical profiling of the neoplasm were performed.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Histopathological evaluation showed a primary MANEC arising within a preexisting congenital enteric cyst, comprising both adenocarcinoma and neuroendocrine components. The adenocarcinoma had a colonic\u2010type morphology and coexpressed CK7, CK20, and CDX2. Imaging and colonoscopy excluded a distant primary colorectal neoplasm.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>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.<\/jats:p><\/jats:sec>","DOI":"10.1002\/hed.25117","type":"journal-article","created":{"date-parts":[[2018,2,23]],"date-time":"2018-02-23T06:34:29Z","timestamp":1519367669000},"source":"Crossref","is-referenced-by-count":7,"title":["Mixed adenoneuroendocrine carcinoma of the tongue arising within a congenital enteric cyst"],"prefix":"10.1002","volume":"40","author":[{"given":"Louis J.","family":"Ligthelm","sequence":"first","affiliation":[{"name":"Ampath Laboratories, Drs Du Buisson, Kramer, Swart, Bouwer Inc  South Africa"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Belinda K.","family":"Bunn","sequence":"additional","affiliation":[{"name":"Department of Oral Pathology and Oral Biology, School of Dentistry, Faculty of Health Sciences University of Pretoria  South Africa"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Erich J.","family":"Raubenheimer","sequence":"additional","affiliation":[{"name":"Ampath Laboratories, Drs Du Buisson, Kramer, Swart, Bouwer Inc  South Africa"},{"name":"Department of Oral Pathology and Oral Biology, School of Dentistry, Faculty of Health Sciences University of Pretoria  South Africa"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2494-667X","authenticated-orcid":false,"given":"Willie F. P.","family":"van Heerden","sequence":"additional","affiliation":[{"name":"Department of Oral Pathology and Oral Biology, School of Dentistry, Faculty of Health Sciences University of Pretoria  South Africa"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2018,2,23]]},"reference":[{"key":"e_1_2_4_2_1","first-page":"13","volume-title":"WHO Classification of Tumours of the Digestive System","author":"Rindi G","year":"2010"},{"key":"e_1_2_4_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/modpathol.2016.167"},{"key":"e_1_2_4_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12105-011-0301-z"},{"key":"e_1_2_4_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2009.01.028"},{"key":"e_1_2_4_6_1","doi-asserted-by":"publisher","DOI":"10.1001\/archotol.127.12.1484"},{"key":"e_1_2_4_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2015.08.006"},{"key":"e_1_2_4_8_1","doi-asserted-by":"publisher","DOI":"10.1245\/s10434-016-5179-2"},{"key":"e_1_2_4_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0b013e318159371c"},{"key":"e_1_2_4_10_1","doi-asserted-by":"publisher","DOI":"10.21614\/chirurgia.112.2.152"},{"key":"e_1_2_4_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bjoms.2012.04.003"},{"key":"e_1_2_4_12_1","doi-asserted-by":"publisher","DOI":"10.1001\/archotol.133.7.717"},{"key":"e_1_2_4_13_1","doi-asserted-by":"publisher","DOI":"10.4132\/KoreanJPathol.2013.47.3.289"},{"key":"e_1_2_4_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0194-5998(98)70382-1"},{"key":"e_1_2_4_15_1","doi-asserted-by":"publisher","DOI":"10.1097\/PAS.0000000000000883"},{"key":"e_1_2_4_16_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2559.2003.01545.x"},{"key":"e_1_2_4_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.oooo.2017.03.049"},{"key":"e_1_2_4_18_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12105-016-0740-7"},{"key":"e_1_2_4_19_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoto.2013.3240a"}],"container-title":["Head &amp; Neck"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fhed.25117","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/hed.25117","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,24]],"date-time":"2023-09-24T17:00:11Z","timestamp":1695574811000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/hed.25117"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,2,23]]},"references-count":18,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2018,5]]}},"alternative-id":["10.1002\/hed.25117"],"URL":"https:\/\/doi.org\/10.1002\/hed.25117","archive":["Portico"],"relation":{},"ISSN":["1043-3074","1097-0347"],"issn-type":[{"type":"print","value":"1043-3074"},{"type":"electronic","value":"1097-0347"}],"subject":[],"published":{"date-parts":[[2018,2,23]]}}}