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      Papillary carcinoma arising in a submentalintralingual thyroglossal duct cyst Translated title: Carcinoma papillare a sede sottomentoniera-intralinguale insorto da cisti del dotto tiroglosso

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          SUMMARY

          An extremely rare case of papillary carcinoma is described arising in a submental-intralingual thyroglossal duct cyst. A 27-year-old male presented with a painless swelling in the submental region extending to the tongue which had gradually been increasing in size over the last three months. Upon examination of the neck, a tender right paramedian mass was found in the submental region just above the hyoid bone, it was mobile upon swallowing without clinical evidence of cervical nodes. The Sistrunk surgical technique, extended to the submental and intra lingual region was used to radically remove the mass. The occurrence of carcinoma of the thyroglossal duct cyst, even though rare and unexpected, should always be considered an option, and histologically ruled out mainly on account of the atypical localization, such as the submental-intralingual reported herein. The two different surgical approaches reported in the literature, one more conservative and the other more aggressive, apparently alternatives, are, instead, complementary and adequate when strict diagnostic criteria and adequate follow-up, are observed.

          RIASSUNTO

          Gli Autori riportano un caso estremamente raro di un carcinoma papillare a sede sottomentoniera-sottolinguale insorto da cisti del dotto tiroglosso. Un giovane di 27 anni si è presentato alla nostra osservazione riferendo dolore in regione sottomentoniera e linguale con difficoltà alla deglutizione da circa tre mesi. Alla palpazione del collo si evidenziò la presenza di una tumefazione in regione sottomentoniera, a sede paramediana destra, al di sopra dell'osso ioide, mobile alla deglutizione, senza linfonodi clinicamente palpabili. La massa fu radicalmente asportata effettuando l'exeresi mediante tecnica descritta da Sistrunk allargata alla regione sottomentoniera. L'insorgenza di un carcinoma papillare da una cisti del dotto tiroglosso è un evento raro e inaspettato. Tuttavia deve essere sempre sospettato e istologicamente escluso specie nelle tumefazioni a localizzazione rara quale quella a sede sottomentoniera descritta. In letteratura sono riportati due differenti approcci chirurgici per la relativa exeresi, uno più demolitivo, l'altro più conservativo, con risultati sovrapponibili; in ogni caso è necessario, in questi casi, seguire adeguati criteri diagnostici e stretti controlli post-operatori.

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          Most cited references17

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          THE SURGICAL TREATMENT OF CYSTS OF THE THYROGLOSSAL TRACT.

          W Sistrunk (1920)
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            Thyroid gland ectopias.

            Failures of descent of the medial anlage of the thyroid and incomplete obliteration of its vertical tract lead to midline or near-midline ectopias such as lingual thyroid and thyroglossal cysts. Each poses special diagnostic and therapeutic considerations. "Ectopias" of the thyroid gland lateral to the carotid artery and jugular vein, however, cannot be readily explained by current embryological information. In these instances, and especially for intranodal thyroid tissue, a metastasis from an occult thyroid primary is the mandatory first consideration.
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              The thyroglossal cyst.

              R Allard (2015)
              The thyroglossal cyst is the most common nonodontogenic cyst in the neck. This cyst may also occur in the lingual or submental areas, though more rarely. Malignant changes have been described. Clinically, no differentiation between a benign cyst and a malignancy can be made. The literature is reviewed regarding the embryology, epidemiology, etiology, symptomatology, radiology, histology, and treatment of this cyst and its malignant counterpart.
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                Author and article information

                Journal
                Acta Otorhinolaryngol Ital
                Pacini
                Acta Otorhinolaryngologica Italica
                Pacini Editore SpA
                0392-100X
                1827-675X
                December 2010
                : 30
                : 6
                : 313
                Affiliations
                Department of Otolaryngology-Head and Neck Surgery, University of Naples "Federico II", Naples;
                [1 ] Institute of Otolaryngology, Sacro Cuore Catholic University, Rome, Italy
                Author notes
                Address for correspondence: Dr. M. Mesolella, via G. Filangieri 36, 80121 Napoli, Italy. Fax: +39 081 415321. E-mail: massimo.mesolella@ 123456tin.it
                Article
                Pacini
                3146324
                21808454
                b6c697cd-3991-4abb-8165-9ed0859e4e7a
                © Copyright by Società Italiana di Otorinolaringologia e Chirurgia Cervico-Facciale

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial No Derivatives License, which permits for noncommercial use, distribution, and reproduction in any digital medium, provided the original work is properly cited and is not altered in any way. For details, please refer to http://creativecommons.org/licenses/by-nc-nd/3.0/

                History
                : 30 March 2009
                : 03 May 2010
                Categories
                Case Report

                Otolaryngology
                head and neck neoplasm,papillary carcinoma,sistrunk procedure,thyroglossal cyst
                Otolaryngology
                head and neck neoplasm, papillary carcinoma, sistrunk procedure, thyroglossal cyst

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