Papillary Thyroid Carcinoma Originated in Thyroglossal Duct Cyst: Case Report

Papillary Thyroid Carcinoma Originated in Thyroglossal Duct Cyst: Case Report | 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 Thyroid Carcinoma Originated in Thyroglossal Duct Cyst: Case Report Damaris Areli García-Cabra, Oscar Alejandro Ortega-Roman, Karla Pamela Carreón-Nava, and 1 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-5947493/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 Purpose : The Thyroglossal Duct Cyst (TDC) is the most prevalent congenital developmental anomaly in the neck, nevertheless, papillary carcinoma emerging from thyroglossal cyst (TGDC) represents an exceptional finding, and the associated appearance with Papillary Thyroid Carcinoma (PTC) is even more uncommon. The clinical presentation is usually almost identical from a benign lesion and the postoperative histopathological study defines the diagnosis. However, its surgical management and follow-up is still debatable because there is no standard consensus for this. The aim of this work is to describe a case of papillary thyroid carcinoma which arise in a histopathological confirmed thyroglossal duct cyst, associated with papillary thyroid carcinoma of different histological variant, an infrequent condition. Methods and Results: This is a case report of papillary thyroid carcinoma emerging from thyroglossal duct cyst, with clinical and imaging attribute of thyroglossal duct cyst, offering surgical management with Sistrunk Procedure (SP) with histopathological result of classical variant of PTC, with invasion to capsule wall. Cabinet tests in the absence of abnormal anatomical findings in thyroid gland, being considered an aspirant for total thyroidectomy (TT) in a second stage, with result of follicular variant of PTC. Conclusion: TGDC is unusual and is commonly detected after surgery. Due to its almost indistinguishable presentation from benign pathology, it constitutes a defiance for the surgeon, being SP the first therapeutic step, in some cases it is fulfill with TT and ablative therapy with radioiodine, nevertheless, there are no systematize recommendations concerning to the surgical therapeutic outcome and its follow-up. Thyroglossal cyst Thyroglossal duct cyst carcinoma Papillary thyroid carcinoma Sistrunk procedure Thyroidectomy Figures Figure 1 Figure 2 Figure 3 Figure 4 Introduction The thyroid gland emerges in the middle of the third and fourth week of embryonic development, from a midline invagination from endodermal cells, which come down from the lingual caecum foramen along the midline towards the anterior section of the neck as regards the first and second tracheal ring, then again, the hyoid bone develops from mesoderm originating at the second and third pharyngeal arch, so that the thyroglossal tract proceeds ventral to it [ 1 – 3 ]. Conventional thyroid tissue evolved anterior to the trachea in the seventh embryonic week, likewise, the thyroglossal duct obliterates its course among the eighth and tenth week of gestation, nonetheless, insufficient obliteration of the thyroglossal tract or preserve epithelial cysts produce a cystic defect known as Thyroglossal Duct Cyst (TDC) [ 1 , 4 – 6 ]. Ectopic thyroid is an exceptional embryologic variation caused by the atypical descent of primordial thyroid tissue and can take place at any time of thyroid descent, from the lingual cecum foramen to the sternal notch, it is in fact uncommon, with an incidence reported in previous works at 1:100,000 to 300,000 in the general adult population [ 7 ]. TDC is the most frequent congenital cervical tumor and accounts for more than 70% of cervical midline tumors in children, furthermore it can appear in the adult population in up to 7%. This tumor appears in the majority as a benign pathology, nevertheless, thyroid tissue can be identified in approximately two thirds of TDCs, consequently, the progression of malignant neoplasm has been reported in < 1%, of which up to 90% is acknowledge as papillary thyroid carcinoma [ 3 , 8 – 10 ]. Most patients who present with this anomaly do so in the first two decades of life, however, up to one third of these may manifest in early adulthood (20–40 years of age). Generally, TDC presents as a painless and growing cervical mass in young adults, in most cases, the diagnosis is made by clinical examination, however, thyroglossal duct carcinoma (TGDC) may be clinically indistinguishable from benign TDC and is mostly diagnosed incidentally after surgical resection [ 11 , 12 ]. Nearly all patients who present this abnormality do so in the first two decades of life, despite, up to one third of these may manifest in early adulthood (20–40 years of age). Typically, TDC presents as a painless and increasing cervical mass in young adults, most of the cases, the diagnosis is made by clinical examination, even tough, thyroglossal duct carcinoma (TGDC) may be clinically identical with from benign TDC and is mainly diagnosed incidentally after surgical resection [ 11 , 12 ]. The evaluation of TCD is made by neck ultrasound, despite, when there is clinical speculation of TGDC, fine needle aspiration (FNA) biopsy is the most precise method for pre-surgical diagnosis. Then again, the standard treatment for TDC is the Sistrunk Procedure (SP), nevertheless, the treatment for TGDC is still debatable, regarding use of lobectomy or thyroidectomy, in addition to the use of iodine ablation therapy [ 13 – 15 ]. The aim of this report is to describe a case of papillary thyroid carcinoma arising in a histopathological confirmed thyroglossal duct cyst, associated with papillary thyroid carcinoma of different histological variant, an infrequent condition, as well as a review of the literature. Case description 28-year-old female, native and resident of State of Mexico, in the absence of chronic degenerative diseases and drug dependence. Began in 2021, noticing an increase in volume in the anterior region of the neck, denies pain, compressive symptoms, hyperthyroidism, hypothyroidism, family history of neck pathology, exposure to radiation or additional symptoms. On clinical examination, a tumor was detected in the anterior cervical region, solid, non-painful, estimated around 3x2 cm, which moves upward when swallowing and tongue retraction, unescorted by palpable cervical lymphadenopathy. Laboratory tests, including thyroid function tests (TFTs) were normal. The neck ultrasound revealed an ovoid, lobulated, hypoechogenic image inferior to the isthmus and left paramedial, with posterior enhancement, with mild flow uptake in its periphery, measuring about 30 x 22 x 21 mm, compatible with TDC. The thyroid gland with size, dimensions and echogenicity was preserved. Cervical lymph nodes were reported to be normal. Considering a benign pathology was suspected, FNA were not routinely offered. Sistrunk Procedure (SP) was performed, reporting trans-surgical findings of TDC measuring 45x25x17 mm, smooth well delimited borders, solid consistency, of infrahyoid location at 2 cm. Was completely removed along with the edge of the hyoid bone [Figure 1]. The concluding histopathologic result reveals Papillary Thyroid Carcinoma (probably originated in thyroglossal cyst) with classic pattern, which focally invades the capsule and adjacent fibro-adipose tissue, with associated vascular permeation, unescorted by invasion of adjacent muscle or bone tissue [Figure 2]. Throughout her clinical follow-up, a new thyroid ultrasound was requested, reporting no evidence of structural alteration, as well to a thyroid profile in normal ranges, although, considering the histopathological result, in a multidisciplinary meeting was decided to perform a total thyroidectomy (TT) [Figure 3]. She had successful postoperative progress and was discharged from the hospital without complications. The final histopathological report of the thyroid gland revealed infiltrating papillary thyroid carcinoma, follicular variant, unifocal and absence of lymph node involvement, concluding pathological stage pT1a. Clinical follow-up with ultrasound control, TFTs and Thyroglobulin level were decided. In addition, he was sent to radioiodine ablation therapy with a dose of 100 mCi [Figure 4]. Discussion TGDC is an unusual pathology, with an incidence reported in previous papers of 0.7 to 1.5% of all TDC, habitually appearing around the fourth decade, with female sex prevailing. The first case was described by Bretano et al [ 16 ], approximately just over 300 cases have been reported in the world medical literature. Even though, the concomitant phenomenon of TGDC and thyroid carcinoma is even more unusual [ 4 , 8 , 17 ]. The diagnosis is complicated, since the clinical and radiological findings are unspecific, and no alteration is observed in biochemical tests, being considered, by frequency as the first diagnostic suspicion a TCD. Though, it should be suspected in the appearance of a midline cyst, solid, irregular or associated with cervical lymphadenopathy. Nearly all cases the diagnosis of TGDC is made post-surgically in order to the histopathological exam corroborate the final diagnosis [ 8 , 13 ]. Josehp et al [ 18 ] conducted a study on the preoperative tests that are implemented when a TDC is suspected, concluding that the most requested study is neck ultrasound in 95%, followed by TFTs in 32%, radioisotope scintigraphy in 3%, FNA in 2% and finally CT in 1%. When TGDC is suspected, other imaging studies can be accomplished besides neck ultrasound for the purpose of assessing the anatomy, extent of the cyst or possible malignancy. Ultrasound-guided FNA has reported a PPV of 53% and NPV of 47%, this has been associated to the low cellular yield due to the cystic nature of the lesion, furthermore to the small malignant component in an ectopic thyroid tissue. In these circumstances, it is recommended to perform FNA in cases with high clinical suspicion of TGDC [ 4 , 8 , 19 ]. According to Widstrom et al [ 20 ], the diagnostic criteria for TGDC include histologic identification (epithelial lining of the ducts with normal thyroid follicles within the cyst walls), normal thyroid tissue close to the tumor, and ruling out primary carcinoma in the thyroid gland. In our case, an intentional search for thyroid malignant pathology was performed by imaging tests, nevertheless, they had no alterations. The histological findings of TGDC are most common papillary carcinoma (85–90%), followed by mixed papillary/follicular (8%), squamous cell (6%) and other types such as Hurtle cell carcinoma, follicular and anaplastic (6%). Metastasis to cervical lymph nodes has been described in 7–15% of cases, lower contrast to Papillary Thyroid Carcinoma. In our case we revealed a associated TGDC with Papillary Thyroid Carcinoma with contrasting histological variant, which is highly uncommon [ 3 , 4 , 11 ]. Presently, SP has become the standard surgical treatment for TDC, with a reported restore rate of up to 95%, nevertheless, malignant neoplasms in TDC are infrequently identified preoperatively, most TGDC are initially treated as a benign tumor. There is no consensus on following management after SP, particularly in the role of Total Thyroidectomy (TT) and radioiodine ablative therapy [ 1 , 3 , 4 ]. In patients with TGDC, thyroid gland involvement has been reported in 33–45% of the cases, so the concept of prognostic risk group in these patients becomes meaningful, additionally risk-based treatment decisions. In these conditions, for low-risk patients (patients < 45 years and in the absence of history of low-dose neck radiation in childhood, clinically or radiologically normal thyroid and small tumors, without histologically positive margins, cyst wall invasion or metastatic spread to lymph nodes) only SP is recommended. On the contrary, for high-risk patients (patients > 45 years, tumors > 4 cm, soft tissue extension, distant metastases) and those with clinically or radiologically abnormal regional lymph nodes, more aggressive treatment including SP, TT and radioiodine ablative therapy is necessary. In the case presented, invasion of the capsule was observed, so aggressive management with TT and ablative therapy with radioactive iodine was decided [ 4 , 9 ]. Despite multiple reviews, the treatment of TGDC is still disputable; there are no standardized recommendations concerning surgical management and post-surgical follow-up. The prognosis of TGDC is usually magnificent, with metastatic lesions reported in less than 2% of cases. Some authors recommend post-surgical scrutiny and follow-up with serum thyroglobulin levels, ultrasound monitoring of the post-surgical site and entire-body scintigraphy. [ 1 , 3 , 11 ] Conclusion TGDC is uncommon and is habitually detected after surgery. Due to its almost indistinguishable presentation from benign pathology, it represents a diagnostic challenge for the surgeon, being SP the first therapeutic step; nevertheless, there are no establish recommendations concerning the surgical therapeutic development and its follow-up. Due to its oddity, the presentation of new cases contributes to the unification of criteria of the management. The individualization of each case plays a fundamental role in its prognosis. Statements and assertions Funding: This study was not supported by any funding. Conflict of Interest: The authors declare that they have no conflict of interest. Ethical approval: All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. Consent to Participate: Informed consent was obtained from all individual participants included in the study. Consent for publication: Consent for publication was obtained for every individual person’s data included in this case report. References Gómez-Álvarez LR, Treviño-Lozano MA, de la O-Escamilla ME, Vergara-Miranda H, Anda LAM, Falcón LÁC, Tafoya AR. (2022) Papillary thyroid carcinoma from a thyroglossal cyst: case series. J Surg Case Rep. 2:rjab613. doi: 10.1093/jscr/rjab613. Fernandez JF, Ordoñez NG, Schultz PN, Samaan NA, Hickey RC. (1991) Thyroglossal duct carcinoma. Surgery. 110(6):928-34. Alqahtani SM, Rayzah M, Al Mutairi A, Alturiqy M, Hendam A, Bin Makhashen M. (2022) Papillary carcinoma arising from a thyroglossal duct cyst: A case report and literature review. Int J Surg Case Rep. 94:107106. https://doi.org/10.1016/j.ijscr.2022.107106. Li CF, Zhang LH, Li XS, Yu LS. (2023) Individual treatment strategy of thyroglossal duct cyst carcinoma: a case report. Gland Surg. 12(4):555-561. https://doi.org/10.21037/gs-23-102. Adelchi C, Mara P, Melissa L, De Stefano A, Cesare M. (2014) Ectopic thyroid tissue in the head and neck: a case series. BMC Res Notes. 7:790. doi: 10.1186/1756-0500-7-790. Balalaa N, Megahed M, Ashari MA, Branicki F. (2011) Thyroglossal duct cyst papillary carcinoma. Case Rep Oncol. 4(1):39-43. https://doi.org/10.1159/000324405. Naik K. (2014) Redefining anatomy in a case of midline subhyoid ectopic thyroid. Indian J Surg. 76(4):333-5. https://doi.org/10.1007/s12262-013-0846-z. Charbel N, Chadi F, Bassam T. (2012) Two cases of papillary carcinoma in a thyroglossal duct cyst. EJENTAS. 13:133-135. https://doi.org/10.1016/j.ejenta.2012.10.003. Plaza CP, López ME, Carrasco CE, Meseguer LM, Perucho Ade L. (2006) Management of well-differentiated thyroglossal remnant thyroid carcinoma: time to close the debate? Report of five new cases and proposal of a definitive algorithm for treatment. Ann Surg Oncol. 13(5):745-52. https://doi.org/10.1245/ASO.2006.05.022. Stein T, Murugan P, Li F, El Hag MI. (2018) Can Medullary Thyroid Carcinoma Arise in Thyroglossal Duct Cysts? A Search for Parafollicular C-cells in 41 Resected Cases. Head Neck Pathol. 12(1):71-74. https://doi.org/10.1007/s12105-017-0826-x. Yang SI, Park KK, Kim JH. (2013) Papillary carcinoma arising from thyroglossal duct cyst with thyroid and lateral neck metastasis. Int J Surg Case Rep. 4(8):704-7. https://doi.org/10.1016/j.ijscr.2013.05.007. Telander RL, Deane SA. (1977) Thyroglossal and branchial cleft cysts and sinuses. Surg Clin North Am. 57(4):779-91. https://doi.org/10.1016/s0039-6109(16)41288-0. Palomino Martínez BD, Beristain Hernández JL, Piscil Salazar MA, Villalpando Mendoza CJ, Velázquez García JA. (2014) Thyroglossal cyst as primary site of papillary thyroid carcinoma metastatic to jugular lymph node chain. Endocrinol Nutr. 61(3):e11-2. https://doi.org/10.1016/j.endonu.2013.10.005. Luna-Ortiz K, Hurtado-Lopez LM, Valderrama-Landaeta JL, Ruiz-Vega A. (2004) Thyroglossal duct cyst with papillary carcinoma: what must be done? Thyroid. 14(5):363-6. https://doi.org/10.1089/105072504774193195. Kermani W, Belcadhi M, Abdelkéfi M, Bouzouita K. (2008) Papillary carcinoma arising in a thyroglossal duct cyst: case report and discussion of management modalities. Eur Arch Otorhinolaryngol. 265(2):233-6. https://doi.org/10.1007/s00405-007-0405-y. Brentano H. (1911) Struma aberrata lingual mit druzen metastasen. Dtsch Med Wochenschr 37:665-6. Cherian MP, Nair B, Thomas S, Somanathan T, Sebastian P. (2009) Synchronous papillary carcinoma in thyroglossal duct cyst and thyroid gland: case report and review of literature. Head Neck. 31(10):1387-91. https://doi.org/10.1002/hed.21029. Joseph J, Lim K, Ramsden J. (2012) Investigation prior to thyroglossal duct cyst excision. Ann R Coll Surg Engl. 94(3):181-4. https://doi.org/10.1308/003588412X13171221589892. Thompson LDR, Herrera HB, Lau SK. (2017) Thyroglossal Duct Cyst Carcinomas: A Clinicopathologic Series of 22 Cases with Staging Recommendations. Head Neck Pathol. 11(2):175-185. https://doi.org/10.1007/s12105-016-0757-y. Thompson LDR, Herrera HB, Lau SK. (2017) Thyroglossal Duct Cyst Carcinomas: A Clinicopathologic Series of 22 Cases with Staging Recommendations. Head Neck Pathol. 11(2):175-185. https://doi.org/10.1007/s12105-016-0757-y. Additional Declarations No competing interests reported. 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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-5947493","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":410564644,"identity":"dc312cd3-588c-4889-b844-cfb3f8815804","order_by":0,"name":"Damaris Areli García-Cabra","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAABA0lEQVRIiWNgGAWjYDCCAyDCAETwAHHFAYjgA+K1nDkApg4kENTCANXC2AbRwoBPC9/x04mfKwrqGPjbzx778HPeHTl7scMPgbbYyek2YNcieSZ3s+QZg8MMEmfykmf2bntmzCOdZgDUkmxsdgC7FoMDuRskGwwOAL2TY8zAu+1wYo90AkjLgcRtuLScf7v5Z4NBHYMB/xtjxr9zQFrSP+DXciN3G9AWZgYDiRxjZt4GkJYc/LZI3ni7zbLB4DCPxI13ycwyxw4b89zOKTiQYIDbL3znczffbPhTJ8ffn3uY8U3NYTn22embP3yosJPDpQUGeNAdjF/5KBgFo2AUjAL8AAAdgmQqoWjDbQAAAABJRU5ErkJggg==","orcid":"","institution":"Hospital de Especialidades, Centro Medico Nacional \"La Raza\"","correspondingAuthor":true,"prefix":"","firstName":"Damaris","middleName":"Areli","lastName":"García-Cabra","suffix":""},{"id":410564645,"identity":"17d5fc00-6fe3-4ebf-8bae-7f165018535d","order_by":1,"name":"Oscar Alejandro Ortega-Roman","email":"","orcid":"","institution":"Hospital de Especialidades, Centro Medico Nacional \"La Raza\"","correspondingAuthor":false,"prefix":"","firstName":"Oscar","middleName":"Alejandro","lastName":"Ortega-Roman","suffix":""},{"id":410564646,"identity":"fd95c10f-ee1e-4283-af55-7a2e3b5363ad","order_by":2,"name":"Karla Pamela Carreón-Nava","email":"","orcid":"","institution":"Hospital de Especialidades, Centro Medico Nacional \"La Raza\"","correspondingAuthor":false,"prefix":"","firstName":"Karla","middleName":"Pamela","lastName":"Carreón-Nava","suffix":""},{"id":410564647,"identity":"95918cdb-cc9f-4ec6-a7e5-fc716a749d4e","order_by":3,"name":"Erick Rueda-Don Juan","email":"","orcid":"","institution":"Hospital de Especialidades, Centro Medico Nacional \"La Raza\"","correspondingAuthor":false,"prefix":"","firstName":"Erick","middleName":"Rueda-Don","lastName":"Juan","suffix":""}],"badges":[],"createdAt":"2025-02-02 21:53:10","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-5947493/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-5947493/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":75994964,"identity":"a4b17b17-079a-4dae-acb7-1e309fbb4cfb","added_by":"auto","created_at":"2025-02-11 09:44:30","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":623812,"visible":true,"origin":"","legend":"\u003cp\u003eBloc resection of thyroglossal cyst (Sistrunk procedure)\u003c/p\u003e","description":"","filename":"1.png","url":"https://assets-eu.researchsquare.com/files/rs-5947493/v1/ca80edc680dc6b0d6b793a47.png"},{"id":75995561,"identity":"8a53d770-5b25-46d2-b153-6aa67d112659","added_by":"auto","created_at":"2025-02-11 09:52:30","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":463156,"visible":true,"origin":"","legend":"\u003cp\u003ePapillary thyroid carcinoma originated in thyroglossal duct cyst. a) Thyroglossal cyst capsule with invasion of malignant thyroid tissue. b and c) Cubic epithelium with empty nuclei with ground-glass image characteristic and thick nuclear membrane\u003c/p\u003e","description":"","filename":"2.png","url":"https://assets-eu.researchsquare.com/files/rs-5947493/v1/ccdd42d0af95c4542aca6460.png"},{"id":75994971,"identity":"acd474a2-6a30-47ec-856e-5b05f6567fd0","added_by":"auto","created_at":"2025-02-11 09:44:30","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":251741,"visible":true,"origin":"","legend":"\u003cp\u003eThyroid ultrasound. In usual situation, with homogeneous echogenicity, without evidence of structural alteration, without evidence of abnormal lymph nodes. a) Right lobe with measures of 38.2 x 16.6 x14.4 mm. b) Isthmus of 2.2 mm in its diameter. c) Right lobe with measures of 38.2 x 16.6 x14.4 mm.\u003c/p\u003e","description":"","filename":"3.png","url":"https://assets-eu.researchsquare.com/files/rs-5947493/v1/f83cd8fc6fc5fd3987bc082c.png"},{"id":75997130,"identity":"f5f14b06-afd1-4963-9084-0efb28703c3f","added_by":"auto","created_at":"2025-02-11 10:00:30","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":174131,"visible":true,"origin":"","legend":"\u003cp\u003ePost-surgical clinical follow-up (14 days)\u003c/p\u003e","description":"","filename":"4.png","url":"https://assets-eu.researchsquare.com/files/rs-5947493/v1/1c88be495211b5d0abd3241e.png"},{"id":75998329,"identity":"1f8be944-739a-42fb-ae80-e8a6a7dd7a33","added_by":"auto","created_at":"2025-02-11 10:08:53","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":2277806,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-5947493/v1/8683ef03-a573-465a-9732-c94100fdb7c3.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"\u003cp\u003ePapillary Thyroid Carcinoma Originated in Thyroglossal Duct Cyst: Case Report\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eThe thyroid gland emerges in the middle of the third and fourth week of embryonic development, from a midline invagination from endodermal cells, which come down from the lingual caecum foramen along the midline towards the anterior section of the neck as regards the first and second tracheal ring, then again, the hyoid bone develops from mesoderm originating at the second and third pharyngeal arch, so that the thyroglossal tract proceeds ventral to it [\u003cspan additionalcitationids=\"CR2\" citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eConventional thyroid tissue evolved anterior to the trachea in the seventh embryonic week, likewise, the thyroglossal duct obliterates its course among the eighth and tenth week of gestation, nonetheless, insufficient obliteration of the thyroglossal tract or preserve epithelial cysts produce a cystic defect known as Thyroglossal Duct Cyst (TDC) [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan additionalcitationids=\"CR5\" citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eEctopic thyroid is an exceptional embryologic variation caused by the atypical descent of primordial thyroid tissue and can take place at any time of thyroid descent, from the lingual cecum foramen to the sternal notch, it is in fact uncommon, with an incidence reported in previous works at 1:100,000 to 300,000 in the general adult population [\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eTDC is the most frequent congenital cervical tumor and accounts for more than 70% of cervical midline tumors in children, furthermore it can appear in the adult population in up to 7%. This tumor appears in the majority as a benign pathology, nevertheless, thyroid tissue can be identified in approximately two thirds of TDCs, consequently, the progression of malignant neoplasm has been reported in \u0026lt;\u0026thinsp;1%, of which up to 90% is acknowledge as papillary thyroid carcinoma [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan additionalcitationids=\"CR9\" citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eMost patients who present with this anomaly do so in the first two decades of life, however, up to one third of these may manifest in early adulthood (20\u0026ndash;40 years of age). Generally, TDC presents as a painless and growing cervical mass in young adults, in most cases, the diagnosis is made by clinical examination, however, thyroglossal duct carcinoma (TGDC) may be clinically indistinguishable from benign TDC and is mostly diagnosed incidentally after surgical resection [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e, \u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eNearly all patients who present this abnormality do so in the first two decades of life, despite, up to one third of these may manifest in early adulthood (20\u0026ndash;40 years of age). Typically, TDC presents as a painless and increasing cervical mass in young adults, most of the cases, the diagnosis is made by clinical examination, even tough, thyroglossal duct carcinoma (TGDC) may be clinically identical with from benign TDC and is mainly diagnosed incidentally after surgical resection [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e, \u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe evaluation of TCD is made by neck ultrasound, despite, when there is clinical speculation of TGDC, fine needle aspiration (FNA) biopsy is the most precise method for pre-surgical diagnosis. Then again, the standard treatment for TDC is the Sistrunk Procedure (SP), nevertheless, the treatment for TGDC is still debatable, regarding use of lobectomy or thyroidectomy, in addition to the use of iodine ablation therapy [\u003cspan additionalcitationids=\"CR14\" citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe aim of this report is to describe a case of papillary thyroid carcinoma arising in a histopathological confirmed thyroglossal duct cyst, associated with papillary thyroid carcinoma of different histological variant, an infrequent condition, as well as a review of the literature.\u003c/p\u003e"},{"header":"Case description","content":"\u003cp\u003e28-year-old female, native and resident of State of Mexico, in the absence of chronic degenerative diseases and drug dependence. Began in 2021, noticing an increase in volume in the anterior region of the neck, denies pain, compressive symptoms, hyperthyroidism, hypothyroidism, family history of neck pathology, exposure to radiation or additional symptoms. On clinical examination, a tumor was detected in the anterior cervical region, solid, non-painful, estimated around 3x2 cm, which moves upward when swallowing and tongue retraction, unescorted by palpable cervical lymphadenopathy.\u003c/p\u003e\n\u003cp\u003eLaboratory tests, including thyroid function tests (TFTs) were normal. The neck ultrasound revealed an ovoid, lobulated, hypoechogenic image inferior to the isthmus and left paramedial, with posterior enhancement, with mild flow uptake in its periphery, measuring about 30 x 22 x 21 mm, compatible with TDC. The thyroid gland with size, dimensions and echogenicity was preserved. Cervical lymph nodes were reported to be normal. Considering a benign pathology was suspected, FNA were not routinely offered.\u003c/p\u003e\n\u003cp\u003eSistrunk Procedure (SP) was performed, reporting trans-surgical findings of TDC measuring 45x25x17 mm, smooth well delimited borders, solid consistency, of infrahyoid location at 2 cm. Was completely removed along with the edge of the hyoid bone [Figure 1].\u003c/p\u003e\n\u003cp\u003eThe concluding histopathologic result reveals Papillary Thyroid Carcinoma (probably originated in thyroglossal cyst) with classic pattern, which focally invades the capsule and adjacent fibro-adipose tissue, with associated vascular permeation, unescorted by invasion of adjacent muscle or bone tissue [Figure 2].\u003c/p\u003e\n\u003cp\u003eThroughout her clinical follow-up, a new thyroid ultrasound was requested, reporting no evidence of structural alteration, as well to a thyroid profile in normal ranges, although, considering the histopathological result, in a multidisciplinary meeting was decided to perform a total thyroidectomy (TT) [Figure 3].\u003c/p\u003e\n\u003cp\u003eShe had successful postoperative progress and was discharged from the hospital without complications. The final histopathological report of the thyroid gland revealed infiltrating papillary thyroid carcinoma, follicular variant, unifocal and absence of lymph node involvement, concluding pathological stage pT1a.\u003c/p\u003e\n\u003cp\u003eClinical follow-up with ultrasound control, TFTs and Thyroglobulin level were decided. In addition, he was sent to radioiodine ablation therapy with a dose of 100 mCi [Figure 4].\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eTGDC is an unusual pathology, with an incidence reported in previous papers of 0.7 to 1.5% of all TDC, habitually appearing around the fourth decade, with female sex prevailing. The first case was described by Bretano et al [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e], approximately just over 300 cases have been reported in the world medical literature. Even though, the concomitant phenomenon of TGDC and thyroid carcinoma is even more unusual [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e, \u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eThe diagnosis is complicated, since the clinical and radiological findings are unspecific, and no alteration is observed in biochemical tests, being considered, by frequency as the first diagnostic suspicion a TCD. Though, it should be suspected in the appearance of a midline cyst, solid, irregular or associated with cervical lymphadenopathy. Nearly all cases the diagnosis of TGDC is made post-surgically in order to the histopathological exam corroborate the final diagnosis [\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e, \u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eJosehp et al [\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e] conducted a study on the preoperative tests that are implemented when a TDC is suspected, concluding that the most requested study is neck ultrasound in 95%, followed by TFTs in 32%, radioisotope scintigraphy in 3%, FNA in 2% and finally CT in 1%. When TGDC is suspected, other imaging studies can be accomplished besides neck ultrasound for the purpose of assessing the anatomy, extent of the cyst or possible malignancy. Ultrasound-guided FNA has reported a PPV of 53% and NPV of 47%, this has been associated to the low cellular yield due to the cystic nature of the lesion, furthermore to the small malignant component in an ectopic thyroid tissue. In these circumstances, it is recommended to perform FNA in cases with high clinical suspicion of TGDC [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e, \u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eAccording to Widstrom et al [\u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e], the diagnostic criteria for TGDC include histologic identification (epithelial lining of the ducts with normal thyroid follicles within the cyst walls), normal thyroid tissue close to the tumor, and ruling out primary carcinoma in the thyroid gland. In our case, an intentional search for thyroid malignant pathology was performed by imaging tests, nevertheless, they had no alterations. The histological findings of TGDC are most common papillary carcinoma (85\u0026ndash;90%), followed by mixed papillary/follicular (8%), squamous cell (6%) and other types such as Hurtle cell carcinoma, follicular and anaplastic (6%). Metastasis to cervical lymph nodes has been described in 7\u0026ndash;15% of cases, lower contrast to Papillary Thyroid Carcinoma. In our case we revealed a associated TGDC with Papillary Thyroid Carcinoma with contrasting histological variant, which is highly uncommon [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e].\u003c/p\u003e \u003cp\u003ePresently, SP has become the standard surgical treatment for TDC, with a reported restore rate of up to 95%, nevertheless, malignant neoplasms in TDC are infrequently identified preoperatively, most TGDC are initially treated as a benign tumor. There is no consensus on following management after SP, particularly in the role of Total Thyroidectomy (TT) and radioiodine ablative therapy [\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\u003eIn patients with TGDC, thyroid gland involvement has been reported in 33\u0026ndash;45% of the cases, so the concept of prognostic risk group in these patients becomes meaningful, additionally risk-based treatment decisions. In these conditions, for low-risk patients (patients\u0026thinsp;\u0026lt;\u0026thinsp;45 years and in the absence of history of low-dose neck radiation in childhood, clinically or radiologically normal thyroid and small tumors, without histologically positive margins, cyst wall invasion or metastatic spread to lymph nodes) only SP is recommended. On the contrary, for high-risk patients (patients\u0026thinsp;\u0026gt;\u0026thinsp;45 years, tumors\u0026thinsp;\u0026gt;\u0026thinsp;4 cm, soft tissue extension, distant metastases) and those with clinically or radiologically abnormal regional lymph nodes, more aggressive treatment including SP, TT and radioiodine ablative therapy is necessary. In the case presented, invasion of the capsule was observed, so aggressive management with TT and ablative therapy with radioactive iodine was decided [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eDespite multiple reviews, the treatment of TGDC is still disputable; there are no standardized recommendations concerning surgical management and post-surgical follow-up. The prognosis of TGDC is usually magnificent, with metastatic lesions reported in less than 2% of cases. Some authors recommend post-surgical scrutiny and follow-up with serum thyroglobulin levels, ultrasound monitoring of the post-surgical site and entire-body scintigraphy. [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eTGDC is uncommon and is habitually detected after surgery. Due to its almost indistinguishable presentation from benign pathology, it represents a diagnostic challenge for the surgeon, being SP the first therapeutic step; nevertheless, there are no establish recommendations concerning the surgical therapeutic development and its follow-up. Due to its oddity, the presentation of new cases contributes to the unification of criteria of the management. The individualization of each case plays a fundamental role in its prognosis.\u003c/p\u003e"},{"header":"Statements and assertions","content":"\u003cp\u003e\u003cstrong\u003eFunding:\u0026nbsp;\u003c/strong\u003eThis study was not supported by any funding.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConflict of Interest:\u0026nbsp;\u003c/strong\u003eThe authors declare that they have no conflict of interest.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthical approval:\u0026nbsp;\u003c/strong\u003eAll procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent to Participate:\u0026nbsp;\u003c/strong\u003eInformed consent was obtained from all individual participants included in the study.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication:\u0026nbsp;\u003c/strong\u003eConsent for publication was obtained for every individual person’s data included in this case report.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n \u003cli\u003eG\u0026oacute;mez-\u0026Aacute;lvarez LR, Trevi\u0026ntilde;o-Lozano MA, de la O-Escamilla ME, Vergara-Miranda H, Anda LAM, Falc\u0026oacute;n L\u0026Aacute;C, Tafoya AR. (2022) Papillary thyroid carcinoma from a thyroglossal cyst: case series. J Surg Case Rep. 2:rjab613. doi: 10.1093/jscr/rjab613.\u003c/li\u003e\n \u003cli\u003eFernandez JF, Ordo\u0026ntilde;ez NG, Schultz PN, Samaan NA, Hickey RC. (1991) Thyroglossal duct carcinoma. Surgery. 110(6):928-34.\u003c/li\u003e\n \u003cli\u003eAlqahtani SM, Rayzah M, Al Mutairi A, Alturiqy M, Hendam A, Bin Makhashen M. (2022) Papillary carcinoma arising from a thyroglossal duct cyst: A case report and literature review. Int J Surg Case Rep. 94:107106.\u0026nbsp;https://doi.org/10.1016/j.ijscr.2022.107106.\u0026nbsp;\u003c/li\u003e\n \u003cli\u003eLi CF, Zhang LH, Li XS, Yu LS. (2023) Individual treatment strategy of thyroglossal duct cyst carcinoma: a case report. Gland Surg. 12(4):555-561.\u0026nbsp;https://doi.org/10.21037/gs-23-102.\u003c/li\u003e\n \u003cli\u003eAdelchi C, Mara P, Melissa L, De Stefano A, Cesare M. (2014) Ectopic thyroid tissue in the head and neck: a case series. BMC Res Notes. 7:790. doi: 10.1186/1756-0500-7-790.\u003c/li\u003e\n \u003cli\u003eBalalaa N, Megahed M, Ashari MA, Branicki F. (2011) Thyroglossal duct cyst papillary carcinoma. Case Rep Oncol. 4(1):39-43.\u0026nbsp;https://doi.org/10.1159/000324405.\u003c/li\u003e\n \u003cli\u003eNaik K. (2014) Redefining anatomy in a case of midline subhyoid ectopic thyroid. Indian J Surg. 76(4):333-5.\u0026nbsp;https://doi.org/10.1007/s12262-013-0846-z.\u003c/li\u003e\n \u003cli\u003eCharbel N, Chadi F, Bassam T. (2012) Two cases of papillary carcinoma in a thyroglossal duct cyst. EJENTAS. 13:133-135.\u0026nbsp;https://doi.org/10.1016/j.ejenta.2012.10.003.\u003c/li\u003e\n \u003cli\u003ePlaza CP, L\u0026oacute;pez ME, Carrasco CE, Meseguer LM, Perucho Ade L. (2006) Management of well-differentiated thyroglossal remnant thyroid carcinoma: time to close the debate? Report of five new cases and proposal of a definitive algorithm for treatment. Ann Surg Oncol. 13(5):745-52.\u0026nbsp;https://doi.org/10.1245/ASO.2006.05.022.\u003c/li\u003e\n \u003cli\u003eStein T, Murugan P, Li F, El Hag MI. (2018) Can Medullary Thyroid Carcinoma Arise in Thyroglossal Duct Cysts? A Search for Parafollicular C-cells in 41 Resected Cases. Head Neck Pathol. 12(1):71-74.\u0026nbsp;https://doi.org/10.1007/s12105-017-0826-x.\u003c/li\u003e\n \u003cli\u003eYang SI, Park KK, Kim JH. (2013) Papillary carcinoma arising from thyroglossal duct cyst with thyroid and lateral neck metastasis. Int J Surg Case Rep. 4(8):704-7.\u0026nbsp;https://doi.org/10.1016/j.ijscr.2013.05.007.\u003c/li\u003e\n \u003cli\u003e\u0026nbsp;Telander RL, Deane SA. (1977) Thyroglossal and branchial cleft cysts and sinuses. Surg Clin North Am. 57(4):779-91.\u0026nbsp;https://doi.org/10.1016/s0039-6109(16)41288-0.\u0026nbsp;\u003c/li\u003e\n \u003cli\u003ePalomino Mart\u0026iacute;nez BD, Beristain Hern\u0026aacute;ndez JL, Piscil Salazar MA, Villalpando Mendoza CJ, Vel\u0026aacute;zquez Garc\u0026iacute;a JA. (2014) Thyroglossal cyst as primary site of papillary thyroid carcinoma metastatic to jugular lymph node chain. Endocrinol Nutr. 61(3):e11-2.\u0026nbsp;https://doi.org/10.1016/j.endonu.2013.10.005.\u003c/li\u003e\n \u003cli\u003eLuna-Ortiz K, Hurtado-Lopez LM, Valderrama-Landaeta JL, Ruiz-Vega A. (2004) Thyroglossal duct cyst with papillary carcinoma: what must be done? Thyroid. 14(5):363-6.\u0026nbsp;https://doi.org/10.1089/105072504774193195.\u003c/li\u003e\n \u003cli\u003eKermani W, Belcadhi M, Abdelk\u0026eacute;fi M, Bouzouita K. (2008) Papillary carcinoma arising in a thyroglossal duct cyst: case report and discussion of management modalities. Eur Arch Otorhinolaryngol. 265(2):233-6.\u0026nbsp;https://doi.org/10.1007/s00405-007-0405-y.\u0026nbsp;\u003c/li\u003e\n \u003cli\u003e\u003ccite\u003eBrentano H. (1911) Struma aberrata lingual mit druzen metastasen. Dtsch Med Wochenschr \u0026nbsp;37:665-6.\u003c/cite\u003e\u0026nbsp;\u003c/li\u003e\n \u003cli\u003eCherian MP, Nair B, Thomas S, Somanathan T, Sebastian P. (2009) Synchronous papillary carcinoma in thyroglossal duct cyst and thyroid gland: case report and review of literature. Head Neck. 31(10):1387-91.\u0026nbsp;https://doi.org/10.1002/hed.21029.\u003c/li\u003e\n \u003cli\u003eJoseph J, Lim K, Ramsden J. (2012) Investigation prior to thyroglossal duct cyst excision. Ann R Coll Surg Engl. 94(3):181-4.\u0026nbsp;https://doi.org/10.1308/003588412X13171221589892.\u003c/li\u003e\n \u003cli\u003eThompson LDR, Herrera HB, Lau SK. (2017) Thyroglossal Duct Cyst Carcinomas: A Clinicopathologic Series of 22 Cases with Staging Recommendations. Head Neck Pathol. 11(2):175-185.\u0026nbsp;https://doi.org/10.1007/s12105-016-0757-y.\u003c/li\u003e\n \u003cli\u003eThompson LDR, Herrera HB, Lau SK. (2017) Thyroglossal Duct Cyst Carcinomas: A Clinicopathologic Series of 22 Cases with Staging Recommendations. Head Neck Pathol. 11(2):175-185. https://doi.org/10.1007/s12105-016-0757-y.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"hideJournal":true,"highlight":"","institution":"","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 cyst, Thyroglossal duct cyst carcinoma, Papillary thyroid carcinoma, Sistrunk procedure, Thyroidectomy","lastPublishedDoi":"10.21203/rs.3.rs-5947493/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-5947493/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003ePurpose\u003c/strong\u003e: The Thyroglossal Duct Cyst (TDC) is the most prevalent congenital developmental anomaly in the neck, nevertheless, papillary carcinoma emerging from thyroglossal cyst (TGDC) represents an exceptional finding, and the associated appearance with Papillary Thyroid Carcinoma (PTC) is even more uncommon. The clinical presentation is usually almost identical from a benign lesion and the postoperative histopathological study defines the diagnosis. However, its surgical management and follow-up is still debatable because there is no standard consensus for this. The aim of this work is to describe a case of papillary thyroid carcinoma which arise in a histopathological confirmed thyroglossal duct cyst, associated with papillary thyroid carcinoma of different histological variant, an infrequent condition.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eMethods and Results:\u003c/strong\u003e This is a case report of papillary thyroid carcinoma emerging from thyroglossal duct cyst, with clinical and imaging attribute of thyroglossal duct cyst, offering surgical management with Sistrunk Procedure (SP) with histopathological result of classical variant of PTC, with invasion to capsule wall. Cabinet tests in the absence of abnormal anatomical findings in thyroid gland, being considered an aspirant for total thyroidectomy (TT) in a second stage, with result of follicular variant of PTC.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusion:\u003c/strong\u003e TGDC is unusual and is commonly detected after surgery. Due to its almost indistinguishable presentation from benign pathology, it constitutes a defiance for the surgeon, being SP the first therapeutic step, in some cases it is fulfill with TT and ablative therapy with radioiodine, nevertheless, there are no systematize recommendations concerning to the surgical therapeutic outcome and its follow-up.\u003c/p\u003e","manuscriptTitle":"Papillary Thyroid Carcinoma Originated in Thyroglossal Duct Cyst: Case Report","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-02-11 09:44:25","doi":"10.21203/rs.3.rs-5947493/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":"1bf78393-e983-4fa2-bbd9-4d32bcc3c255","owner":[],"postedDate":"February 11th, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2025-02-11T09:44:25+00:00","versionOfRecord":[],"versionCreatedAt":"2025-02-11 09:44:25","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-5947493","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-5947493","identity":"rs-5947493","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}