Papillary Carcinoma Arising in a Thyroglossal Duct Cyst: A Case Report and Review of the Literature

Papillary Carcinoma Arising in a Thyroglossal Duct Cyst: A Case Report and Review of the Literature | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Papillary Carcinoma Arising in a Thyroglossal Duct Cyst: A Case Report and Review of the Literature Hamza BOUCHACHA, Ismail OUBLAHCEN, Nabil TOUIHEM, Hicham ATTIFI, and 2 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-9349382/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Background Thyroglossal duct cysts (TGDCs) represent the most common congenital cervical anomalies resulting from incomplete involution of the thyroglossal duct during embryologic development. Malignant transformation within a TGDC is rare, occurring in approximately 1–1.5% of cases , with papillary carcinoma being the most frequent histological type. Case Presentation We report the case of a 30-year-old woman presenting with a midline cervical swelling evolving over four years. Clinical examination revealed a firm, painless anterior cervical mass that was mobile with tongue protrusion . Imaging studies suggested a multiloculated thyroglossal duct cyst with a solid component . The patient underwent a Sistrunk procedure . Histopathological examination revealed papillary thyroid carcinoma measuring 1.5 cm arising within the thyroglossal duct cyst. Following multidisciplinary discussion, completion thyroidectomy was recommended but declined by the patient . Conclusion Papillary carcinoma arising in TGDC is rare and often diagnosed incidentally after surgical excision. The optimal therapeutic strategy remains controversial, particularly regarding the need for additional thyroidectomy. The Sistrunk procedure remains the cornerstone of treatment , while further management should be individualized according to clinical, radiological, and histopathological findings. Head & Neck Surgery Otorhinolaryngology Thyroglossal duct cyst Papillary thyroid carcinoma Sistrunk procedure Neck mass Case report Figures Figure 1 Figure 2 Figure 3 Figure 4 Introduction Thyroglossal duct cysts (TGDCs) are congenital cervical malformations resulting from the persistence of the thyroglossal duct , an embryologic structure that normally extends from the foramen cecum at the base of the tongue to the thyroid isthmus or the pyramidal lobe of Lalouette during thyroid gland descent [ 1 , 2 ]. Malignant transformation within a TGDC is rare, occurring in approximately 1–1.5% of cases , and is predominantly represented by papillary carcinoma [ 1 , 3 , 4 ]. We report the case of a 30-year-old woman who underwent surgery for a TGDC, in whom histopathological examination revealed papillary thyroid carcinoma arising within the cyst , and we review the relevant literature. Case Report A 30-year-old woman with no significant past medical history presented with a midline cervical swelling that had been evolving for four years . Clinical examination revealed a multilobulated anterior cervical mass , painless and non-compressive, mobile on tongue protrusion , firm in consistency, measuring approximately 5 cm in its greatest diameter . No signs of local inflammation or palpable cervical lymphadenopathy were observed. Cervical ultrasonography revealed a large septated thyroglossal duct cyst with vascularized septa and an isoechoic hypervascular solid component , associated with a nodular goiter . A cervical computed tomography (CT) scan demonstrated an anterior cervical mass located below the hyoid bone , predominantly cystic and multilocular, containing a central tissue component enhanced after contrast administration . The patient underwent a Sistrunk procedure , consisting of en bloc resection of the cyst, its tract, and the central portion of the hyoid bone . Histopathological examination of the surgical specimen revealed a papillary thyroid carcinoma measuring 1.5 cm in greatest dimension arising within the thyroglossal duct cyst . Following multidisciplinary discussion , completion treatment with total thyroidectomy was recommended. However, the patient declined further surgery after being informed of the potential benefits and associated risks . Discussion During the third week of embryonic development , the thyroid gland originates as an epithelial proliferation at the floor of the primitive pharynx at the level of the foramen cecum . The thyroid primordium then forms a duct that descends anterior to the pharyngeal gut, known as the thyroglossal duct , which normally involutes and disappears. Failure of this involution results in persistence of the duct and the formation of a thyroglossal duct cyst (TGDC) . TGDCs may occur anywhere along the course of the duct, extending from the base of the tongue to the pyramidal lobe of Lalouette . Malignant transformation within TGDCs is rare, accounting for 1–1.5% of cases , and occurs mainly in adults during the fourth decade of life [1,3,6]. The first description of carcinoma arising in a TGDC was reported by Brentano in 1911 [3]. The papillary histological subtype is the most common, accounting for approximately 83% of cases . Other reported histological types include: Mixed papillary–follicular carcinoma (8%) Squamous cell carcinoma (6%) Rare cases of Hürthle cell carcinoma Follicular carcinoma Anaplastic carcinoma Epidermoid carcinoma No cases of medullary carcinoma arising from TGDC have been reported in the literature [1,3,6–7]. Most authors consider that TGDC carcinomas arise de novo within ectopic thyroid tissue present in the cyst , suggesting that they represent primary thyroglossal duct carcinomas . However, other authors, including Belnoue , have suggested that the thyroglossal duct may serve as a pathway for the spread of primary thyroid carcinoma , which explains the differing therapeutic approaches proposed in the literature [5]. Clinically, TGDC carcinoma usually presents similarly to a benign thyroglossal duct cyst , and the diagnosis is frequently incidental following histopathological examination of the surgical specimen [1]. However, certain clinical features may raise suspicion of malignancy, including: A hard or fixed cervical mass Irregular borders Rapid increase in size Presence of cervical lymphadenopathy [3] The management of TGDC carcinoma remains controversial , particularly regarding the need for thyroidectomy following the Sistrunk procedure [6]. Some authors advocating a more aggressive approach , such as Belnoue [5] and Miccoli [8] , recommend systematic total thyroidectomy due to the relatively high frequency of associated thyroid carcinoma. Indeed, 11–27% of patients undergoing thyroidectomy are found to have synchronous thyroid carcinoma . In addition, thyroidectomy facilitates postoperative surveillance using thyroglobulin levels and radioactive iodine therapy when indicated . In a meta-analysis , Patel [9] demonstrated that the extent of the initial surgical treatment was the only significant factor influencing survival outcomes. Conversely, other authors supporting a more conservative strategy , such as Baïzri [1] , recommend limiting treatment to the Sistrunk procedure alone , citing the slow progression and excellent prognosis of papillary carcinoma arising in TGDCs, provided that regular follow-up is ensured . Overall, the prognosis of TGDC carcinoma is favorable , with a lower incidence of distant metastases compared with primary thyroid carcinoma [5]. According to Patel [9] , the 5-year and 10-year survival rates are approximately 100% and 95.6%, respectively . Conclusion Papillary carcinoma arising in a thyroglossal duct cyst is a rare clinical entity that is most often diagnosed incidentally following surgical excision . Because clinical and radiological findings cannot reliably distinguish a benign TGDC from malignant transformation , the Sistrunk procedure remains the standard initial treatment . However, the optimal postoperative management remains controversial , particularly regarding the indication for completion thyroidectomy . Therefore, therapeutic decisions should be individualized based on clinical, radiological, and histopathological findings, as well as patient preferences. Declarations Written informed consent was obtained from the patient for participation and for publication of this clinical case and accompanying images. References Ghfir I, Ouboukdir R, M'Hamdi F, Ben Rais Aouad N (2010) Degenerated thyroglossal duct cyst revealing a multifocal micropapillary thyroid carcinoma. J Med Nucl 34(1):29–33 Kandogan T, Erkan N, Vardar E (2008) Papillary carcinoma arising in a thyroglossal duct cyst with associated microcarcinoma of the thyroid and without cervical lymph node metastasis: a case report. J Med Case Rep 2:42 Tharmabala M, Kanthan R (2013) Incidental thyroid papillary carcinoma in a thyroglossal duct cyst: management dilemmas. Int J Surg Case Rep 4(1):58–61 Charbel N, Chadi F, Bassam T (2012) Two cases of papillary carcinoma in a thyroglossal duct cyst. Egypt J Ear Nose Throat Allied Sci 13(3):133–135 Belnoue A, Poupart M, Pignat JC (2004) Intérêt de la thyroïdectomie dans la prise en charge des kystes du tractus thyréoglosse dégénérés. Ann Chir 129:523–525 Rogério AD, Daniela LC, Gisele LP, Leonardo W, André VG (2002) Thyroglossal duct: a review of 55 cases. J Am Coll Surg 194(3):274–277 David SF, Mary EF (2006) Thyroglossal duct and other congenital midline cervical anomalies. Semin Pediatr Surg 15(2):70–75 Miccoli P, Minuto MN, Galleri D, Puccini M, Berti P (2004) Extent of surgery in thyroglossal duct carcinoma: reflections on a series of 18 cases. Thyroid 14:121–123 Patel SG, Escrig M, Shaha AR, Singh B, Shah JP (2002) Management of well-differentiated thyroid carcinoma presenting within a thyroglossal duct cyst. J Surg Oncol 79:134–141 Additional Declarations The authors declare no competing interests. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-9349382","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":619201320,"identity":"9007b7b5-2a73-4962-94a0-6ef80a7b9fcf","order_by":0,"name":"Hamza 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Morocco","correspondingAuthor":false,"prefix":"","firstName":"Ismail","middleName":"","lastName":"OUBLAHCEN","suffix":""},{"id":619201873,"identity":"8464bb75-6047-4bc7-8cbd-3cdbfe6285b2","order_by":2,"name":"Nabil TOUIHEM","email":"","orcid":"","institution":"Department of Otolaryngology, Head and Neck Surgery, Moulay Ismail Military Hospital, Meknes, Morocco","correspondingAuthor":false,"prefix":"","firstName":"Nabil","middleName":"","lastName":"TOUIHEM","suffix":""},{"id":619201874,"identity":"bbc7a018-1789-4ae1-81a4-140bcc29e759","order_by":3,"name":"Hicham ATTIFI","email":"","orcid":"","institution":"Department of Otolaryngology, Head and Neck Surgery, Moulay Ismail Military Hospital, Meknes, Morocco","correspondingAuthor":false,"prefix":"","firstName":"Hicham","middleName":"","lastName":"ATTIFI","suffix":""},{"id":619201875,"identity":"0ef52d09-5ed6-4bd5-9ddd-cb1e424cdf9e","order_by":4,"name":"Ali BOUKHARI","email":"","orcid":"","institution":"Department of Otolaryngology, Head and Neck Surgery, Moulay Ismail Military Hospital, Meknes, Morocco","correspondingAuthor":false,"prefix":"","firstName":"Ali","middleName":"","lastName":"BOUKHARI","suffix":""},{"id":619201876,"identity":"44b7a6ab-22be-4a34-a11f-c4515710b9e0","order_by":5,"name":"Mounir HMIDI","email":"","orcid":"","institution":"Department of Otolaryngology, Head and Neck Surgery, Moulay Ismail Military Hospital, Meknes, Morocco","correspondingAuthor":false,"prefix":"","firstName":"Mounir","middleName":"","lastName":"HMIDI","suffix":""}],"badges":[],"createdAt":"2026-04-07 21:22:10","currentVersionCode":1,"declarations":{"humanSubjects":true,"vertebrateSubjects":false,"conflictsOfInterestStatement":false,"humanSubjectEthicalGuidelines":true,"humanSubjectConsent":true,"humanSubjectClinicalTrial":true,"humanSubjectCaseReport":true,"vertebrateSubjectEthicalGuidelines":false},"doi":"10.21203/rs.3.rs-9349382/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-9349382/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":106542930,"identity":"f98e67f7-56a1-4458-ba38-0092ba136a52","added_by":"auto","created_at":"2026-04-09 16:32:10","extension":"jpeg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":228384,"visible":true,"origin":"","legend":"\u003cp\u003eMidline cervical swelling suggestive of a thyroglossal duct cyst.\u003c/p\u003e","description":"","filename":"WhatsAppImage20240225at18.16.25.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-9349382/v1/7a5ecd50c65cebf7a6d0109c.jpeg"},{"id":106725999,"identity":"d122dbec-c5c2-45fa-ad7e-5843d339050a","added_by":"auto","created_at":"2026-04-12 18:34:50","extension":"jpeg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":104018,"visible":true,"origin":"","legend":"\u003cp\u003eCervical CT scan (axial view) showing a multilocular cystic mass suggestive of TGDC.\u003c/p\u003e","description":"","filename":"WhatsAppImage20240225at18.24.30.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-9349382/v1/309abee0da6ff14264171f28.jpeg"},{"id":106726033,"identity":"51fed2c2-e48b-4a0b-a0c5-d01a858ffda5","added_by":"auto","created_at":"2026-04-12 18:35:00","extension":"jpeg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":376233,"visible":true,"origin":"","legend":"\u003cp\u003eIntraoperative view of the thyroglossal duct cyst.\u003c/p\u003e","description":"","filename":"WhatsAppImage20240225at18.16.28.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-9349382/v1/111f344454736e842961dd35.jpeg"},{"id":106725094,"identity":"42ed4ddf-51b7-4005-984d-fc94691e9b43","added_by":"auto","created_at":"2026-04-12 18:31:19","extension":"jpeg","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":151493,"visible":true,"origin":"","legend":"\u003cp\u003eSurgical specimen following excision using the Sistrunk procedure.\u003c/p\u003e","description":"","filename":"WhatsAppImage20240225at18.16.29.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-9349382/v1/f8b4db5649df41b286423ed1.jpeg"},{"id":106727790,"identity":"b9f6bf87-9054-4b15-a881-bfd745bdcd6b","added_by":"auto","created_at":"2026-04-12 18:40:52","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":2516124,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-9349382/v1/fedf59d7-ef94-4944-aa24-8cfaae0551f3.pdf"}],"financialInterests":"The authors declare no competing interests.","formattedTitle":"\u003cp\u003ePapillary Carcinoma Arising in a Thyroglossal Duct Cyst: A Case Report and Review of the Literature\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eThyroglossal duct cysts (TGDCs) are \u003cb\u003econgenital cervical malformations resulting from the persistence of the thyroglossal duct\u003c/b\u003e, an embryologic structure that normally extends from the \u003cb\u003eforamen cecum at the base of the tongue to the thyroid isthmus or the pyramidal lobe of Lalouette\u003c/b\u003e during thyroid gland descent [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eMalignant transformation within a TGDC is rare, occurring in approximately \u003cb\u003e1\u0026ndash;1.5% of cases\u003c/b\u003e, and is predominantly represented by \u003cb\u003epapillary carcinoma\u003c/b\u003e [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eWe report the case of a \u003cb\u003e30-year-old woman\u003c/b\u003e who underwent surgery for a TGDC, in whom \u003cb\u003ehistopathological examination revealed papillary thyroid carcinoma arising within the cyst\u003c/b\u003e, and we review the relevant literature.\u003c/p\u003e"},{"header":"Case Report","content":"\u003cp\u003eA \u003cb\u003e30-year-old woman\u003c/b\u003e with no significant past medical history presented with a \u003cb\u003emidline cervical swelling\u003c/b\u003e that had been evolving for \u003cb\u003efour years\u003c/b\u003e.\u003c/p\u003e \u003cp\u003eClinical examination revealed a \u003cb\u003emultilobulated anterior cervical mass\u003c/b\u003e, painless and non-compressive, \u003cb\u003emobile on tongue protrusion\u003c/b\u003e, firm in consistency, measuring approximately \u003cb\u003e5 cm in its greatest diameter\u003c/b\u003e. No signs of local inflammation or \u003cb\u003epalpable cervical lymphadenopathy\u003c/b\u003e were observed.\u003c/p\u003e \u003cp\u003e \u003cb\u003eCervical ultrasonography\u003c/b\u003e revealed a large \u003cb\u003eseptated thyroglossal duct cyst\u003c/b\u003e with vascularized septa and an \u003cb\u003eisoechoic hypervascular solid component\u003c/b\u003e, associated with a \u003cb\u003enodular goiter\u003c/b\u003e.\u003c/p\u003e \u003cp\u003eA \u003cb\u003ecervical computed tomography (CT) scan\u003c/b\u003e demonstrated an \u003cb\u003eanterior cervical mass located below the hyoid bone\u003c/b\u003e, predominantly cystic and multilocular, containing a \u003cb\u003ecentral tissue component enhanced after contrast administration\u003c/b\u003e.\u003c/p\u003e \u003cp\u003eThe patient underwent a \u003cb\u003eSistrunk procedure\u003c/b\u003e, consisting of \u003cb\u003een bloc resection of the cyst, its tract, and the central portion of the hyoid bone\u003c/b\u003e.\u003c/p\u003e \u003cp\u003eHistopathological examination of the surgical specimen revealed a \u003cb\u003epapillary thyroid carcinoma measuring 1.5 cm in greatest dimension arising within the thyroglossal duct cyst\u003c/b\u003e.\u003c/p\u003e \u003cp\u003eFollowing \u003cb\u003emultidisciplinary discussion\u003c/b\u003e, completion treatment with \u003cb\u003etotal thyroidectomy\u003c/b\u003e was recommended. However, the patient \u003cb\u003edeclined further surgery after being informed of the potential benefits and associated risks\u003c/b\u003e.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eDuring the \u003cstrong\u003ethird week of embryonic development\u003c/strong\u003e, the thyroid gland originates as an epithelial proliferation at the floor of the primitive pharynx at the level of the \u003cstrong\u003eforamen cecum\u003c/strong\u003e. The thyroid primordium then forms a duct that descends anterior to the pharyngeal gut, known as the \u003cstrong\u003ethyroglossal duct\u003c/strong\u003e, which normally involutes and disappears.\u003c/p\u003e\n\u003cp\u003eFailure of this involution results in persistence of the duct and the formation of a \u003cstrong\u003ethyroglossal duct cyst (TGDC)\u003c/strong\u003e.\u003c/p\u003e\n\u003cp\u003eTGDCs may occur anywhere along the course of the duct, extending from the \u003cstrong\u003ebase of the tongue to the pyramidal lobe of Lalouette\u003c/strong\u003e.\u003c/p\u003e\n\u003cp\u003eMalignant transformation within TGDCs is rare, accounting for \u003cstrong\u003e1–1.5% of cases\u003c/strong\u003e, and occurs mainly in \u003cstrong\u003eadults during the fourth decade of life\u003c/strong\u003e [1,3,6].\u003c/p\u003e\n\u003cp\u003eThe first description of carcinoma arising in a TGDC was reported by \u003cstrong\u003eBrentano in 1911\u003c/strong\u003e [3].\u003c/p\u003e\n\u003cp\u003eThe \u003cstrong\u003epapillary histological subtype\u003c/strong\u003e is the most common, accounting for approximately \u003cstrong\u003e83% of cases\u003c/strong\u003e. Other reported histological types include:\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eMixed \u003cstrong\u003epapillary–follicular carcinoma (8%)\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eSquamous cell carcinoma (6%)\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003eRare cases of \u003cstrong\u003eHürthle cell carcinoma\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eFollicular carcinoma\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eAnaplastic carcinoma\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eEpidermoid carcinoma\u003c/strong\u003e\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eNo cases of \u003cstrong\u003emedullary carcinoma arising from TGDC\u003c/strong\u003e have been reported in the literature [1,3,6–7].\u003c/p\u003e\n\u003cp\u003eMost authors consider that TGDC carcinomas arise \u003cstrong\u003ede novo within ectopic thyroid tissue present in the cyst\u003c/strong\u003e, suggesting that they represent \u003cstrong\u003eprimary thyroglossal duct carcinomas\u003c/strong\u003e. However, other authors, including \u003cstrong\u003eBelnoue\u003c/strong\u003e, have suggested that the thyroglossal duct may serve as a \u003cstrong\u003epathway for the spread of primary thyroid carcinoma\u003c/strong\u003e, which explains the differing therapeutic approaches proposed in the literature [5].\u003c/p\u003e\n\u003cp\u003eClinically, TGDC carcinoma usually presents similarly to a \u003cstrong\u003ebenign thyroglossal duct cyst\u003c/strong\u003e, and the diagnosis is frequently \u003cstrong\u003eincidental following histopathological examination of the surgical specimen\u003c/strong\u003e [1].\u003c/p\u003e\n\u003cp\u003eHowever, certain clinical features may raise suspicion of malignancy, including:\u003c/p\u003e\n\u003cul\u003e\n \u003cli\u003eA \u003cstrong\u003ehard or fixed cervical mass\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eIrregular borders\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003e\u003cstrong\u003eRapid increase in size\u003c/strong\u003e\u003c/li\u003e\n \u003cli\u003ePresence of \u003cstrong\u003ecervical lymphadenopathy\u003c/strong\u003e [3]\u003c/li\u003e\n\u003c/ul\u003e\n\u003cp\u003eThe management of TGDC carcinoma remains \u003cstrong\u003econtroversial\u003c/strong\u003e, particularly regarding the need for \u003cstrong\u003ethyroidectomy following the Sistrunk procedure\u003c/strong\u003e [6].\u003c/p\u003e\n\u003cp\u003eSome authors advocating a \u003cstrong\u003emore aggressive approach\u003c/strong\u003e, such as \u003cstrong\u003eBelnoue [5]\u003c/strong\u003e and \u003cstrong\u003eMiccoli [8]\u003c/strong\u003e, recommend \u003cstrong\u003esystematic total thyroidectomy\u003c/strong\u003e due to the relatively high frequency of associated thyroid carcinoma. Indeed, \u003cstrong\u003e11–27% of patients undergoing thyroidectomy are found to have synchronous thyroid carcinoma\u003c/strong\u003e. In addition, thyroidectomy facilitates \u003cstrong\u003epostoperative surveillance using thyroglobulin levels and radioactive iodine therapy when indicated\u003c/strong\u003e.\u003c/p\u003e\n\u003cp\u003eIn a \u003cstrong\u003emeta-analysis\u003c/strong\u003e, \u003cstrong\u003ePatel [9]\u003c/strong\u003e demonstrated that the \u003cstrong\u003eextent of the initial surgical treatment\u003c/strong\u003e was the only significant factor influencing survival outcomes.\u003c/p\u003e\n\u003cp\u003eConversely, other authors supporting a \u003cstrong\u003emore conservative strategy\u003c/strong\u003e, such as \u003cstrong\u003eBaïzri [1]\u003c/strong\u003e, recommend limiting treatment to the \u003cstrong\u003eSistrunk procedure alone\u003c/strong\u003e, citing the \u003cstrong\u003eslow progression and excellent prognosis\u003c/strong\u003e of papillary carcinoma arising in TGDCs, provided that \u003cstrong\u003eregular follow-up is ensured\u003c/strong\u003e.\u003c/p\u003e\n\u003cp\u003eOverall, the \u003cstrong\u003eprognosis of TGDC carcinoma is favorable\u003c/strong\u003e, with a lower incidence of distant metastases compared with primary thyroid carcinoma [5]. According to \u003cstrong\u003ePatel [9]\u003c/strong\u003e, the \u003cstrong\u003e5-year and 10-year survival rates are approximately 100% and 95.6%, respectively\u003c/strong\u003e.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003ePapillary carcinoma arising in a thyroglossal duct cyst is a \u003cb\u003erare clinical entity\u003c/b\u003e that is most often diagnosed \u003cb\u003eincidentally following surgical excision\u003c/b\u003e. Because \u003cb\u003eclinical and radiological findings cannot reliably distinguish a benign TGDC from malignant transformation\u003c/b\u003e, the \u003cb\u003eSistrunk procedure remains the standard initial treatment\u003c/b\u003e. However, the \u003cb\u003eoptimal postoperative management remains controversial\u003c/b\u003e, particularly regarding the indication for \u003cb\u003ecompletion thyroidectomy\u003c/b\u003e. Therefore, \u003cb\u003etherapeutic decisions should be individualized\u003c/b\u003e based on clinical, radiological, and histopathological findings, as well as patient preferences.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003eWritten informed consent was obtained from the patient for participation and for publication of this clinical case and accompanying images.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eGhfir I, Ouboukdir R, M'Hamdi F, Ben Rais Aouad N (2010) Degenerated thyroglossal duct cyst revealing a multifocal micropapillary thyroid carcinoma. J Med Nucl 34(1):29\u0026ndash;33\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eKandogan T, Erkan N, Vardar E (2008) Papillary carcinoma arising in a thyroglossal duct cyst with associated microcarcinoma of the thyroid and without cervical lymph node metastasis: a case report. J Med Case Rep 2:42\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTharmabala M, Kanthan R (2013) Incidental thyroid papillary carcinoma in a thyroglossal duct cyst: management dilemmas. Int J Surg Case Rep 4(1):58\u0026ndash;61\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eCharbel N, Chadi F, Bassam T (2012) Two cases of papillary carcinoma in a thyroglossal duct cyst. Egypt J Ear Nose Throat Allied Sci 13(3):133\u0026ndash;135\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBelnoue A, Poupart M, Pignat JC (2004) Int\u0026eacute;r\u0026ecirc;t de la thyro\u0026iuml;dectomie dans la prise en charge des kystes du tractus thyr\u0026eacute;oglosse d\u0026eacute;g\u0026eacute;n\u0026eacute;r\u0026eacute;s. Ann Chir 129:523\u0026ndash;525\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRog\u0026eacute;rio AD, Daniela LC, Gisele LP, Leonardo W, Andr\u0026eacute; VG (2002) Thyroglossal duct: a review of 55 cases. J Am Coll Surg 194(3):274\u0026ndash;277\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eDavid SF, Mary EF (2006) Thyroglossal duct and other congenital midline cervical anomalies. Semin Pediatr Surg 15(2):70\u0026ndash;75\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMiccoli P, Minuto MN, Galleri D, Puccini M, Berti P (2004) Extent of surgery in thyroglossal duct carcinoma: reflections on a series of 18 cases. Thyroid 14:121\u0026ndash;123\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003ePatel SG, Escrig M, Shaha AR, Singh B, Shah JP (2002) Management of well-differentiated thyroid carcinoma presenting within a thyroglossal duct cyst. J Surg Oncol 79:134\u0026ndash;141\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"hideJournal":true,"highlight":"","institution":"Moulay Ismail Military Hospital, Meknes, Morocco","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Thyroglossal duct cyst, Papillary thyroid carcinoma, Sistrunk procedure, Neck mass, Case report","lastPublishedDoi":"10.21203/rs.3.rs-9349382/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-9349382/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eBackground\u003c/p\u003e\n\u003cp\u003eThyroglossal duct cysts (TGDCs) represent the most common congenital cervical anomalies resulting from incomplete involution of the thyroglossal duct during embryologic development. Malignant transformation within a TGDC is rare, occurring in approximately \u003cstrong\u003e1–1.5% of cases\u003c/strong\u003e, with \u003cstrong\u003epapillary carcinoma\u003c/strong\u003e being the most frequent histological type.\u003c/p\u003e\n\u003cp\u003eCase Presentation\u003c/p\u003e\n\u003cp\u003eWe report the case of a \u003cstrong\u003e30-year-old woman\u003c/strong\u003epresenting with a \u003cstrong\u003emidline cervical swelling\u003c/strong\u003e evolving over four years. Clinical examination revealed a \u003cstrong\u003efirm, painless anterior cervical mass\u003c/strong\u003e that was \u003cstrong\u003emobile with tongue protrusion\u003c/strong\u003e. Imaging studies suggested a \u003cstrong\u003emultiloculated thyroglossal duct cyst with a solid component\u003c/strong\u003e. The patient underwent a \u003cstrong\u003eSistrunk procedure\u003c/strong\u003e. Histopathological examination revealed \u003cstrong\u003epapillary thyroid carcinoma measuring 1.5 cm\u003c/strong\u003e arising within the thyroglossal duct cyst. Following multidisciplinary discussion, \u003cstrong\u003ecompletion thyroidectomy was recommended but declined by the patient\u003c/strong\u003e.\u003c/p\u003e\n\u003cp\u003eConclusion\u003c/p\u003e\n\u003cp\u003ePapillary carcinoma arising in TGDC is rare and often diagnosed incidentally after surgical excision. The optimal therapeutic strategy remains controversial, particularly regarding the need for additional thyroidectomy. \u003cstrong\u003eThe Sistrunk procedure remains the cornerstone of treatment\u003c/strong\u003e, while further management should be individualized according to clinical, radiological, and histopathological findings.\u003c/p\u003e","manuscriptTitle":"Papillary Carcinoma Arising in a Thyroglossal Duct Cyst: A Case Report and Review of the Literature","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2026-04-09 16:32:05","doi":"10.21203/rs.3.rs-9349382/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"3d92cf8e-99de-4f5f-ab45-0ffc31da5e68","owner":[],"postedDate":"April 9th, 2026","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[{"id":65891401,"name":"Head \u0026 Neck Surgery"},{"id":65891402,"name":"Otorhinolaryngology"}],"tags":[],"updatedAt":"2026-04-09T16:32:05+00:00","versionOfRecord":[],"versionCreatedAt":"2026-04-09 16:32:05","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-9349382","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-9349382","identity":"rs-9349382","version":["v1"]},"buildId":"XKTyCvWXoU3ODBz1xrDgd","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}