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Patient records from a tertiary referral center were analyzed for individuals diagnosed with TDCC between 2002 and 2023. A systematic review followed the PRISMA guidelines, encompassing studies from Medline and PubMed. Patient data, including demographics, imaging results, and histological findings were extracted. The primary outcome assessed was tumor-free and recurrence-free survival, while additional variables included treatment regimens and follow-up data. The analysis identified a total of 484 TDCC cases, confirming papillary thyroid carcinoma (PTC) as the predominant type (94.2%), with synchronous thyroid gland carcinomas observed in 34.6%. The age range was 8 to 76 with median age of 40 years. Women were affected in 62%, men in 36%. The recurrence rate was 7.4%, with distant metastases observed in 1% of cases. The overall survival rate was 99.1%, regardless of the treatment regimen. Although rare, TDCC predominantly presents as PTC, with a favorable prognosis. Sistrunk\u2019s procedure remains the primary surgical approach, but optimal management regarding therapies such as total thyroidectomy, neck dissection, or radioiodine ablation requires careful evaluation of risk factors like rare tumor types, suspicious lymph nodes, thyroid nodules, age, radiation exposure, and molecular patterns. Emphasizing more individualized treatment strategies, we propose an algorithm that can help to reduce invasiveness and overtreatment risks in selected cases.<\/jats:p>","DOI":"10.1007\/s00428-025-04125-2","type":"journal-article","created":{"date-parts":[[2025,5,10]],"date-time":"2025-05-10T12:51:52Z","timestamp":1746881512000},"page":"1139-1151","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Thyroglossal duct cyst carcinomas \u2014 a retrospective study and systematic review of the literature"],"prefix":"10.1007","volume":"486","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7631-7336","authenticated-orcid":false,"given":"Vivian","family":"Thimsen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5790-0841","authenticated-orcid":false,"given":"Sarina Katrin","family":"M\u00fcller","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0445-8161","authenticated-orcid":false,"given":"Abbas","family":"Agaimy","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4948-8882","authenticated-orcid":false,"given":"Konstantinos","family":"Mantsopoulos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2523-6266","authenticated-orcid":false,"given":"Michael","family":"Beck","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7838-6490","authenticated-orcid":false,"given":"Michael","family":"Koch","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7441-2265","authenticated-orcid":false,"given":"Heinrich","family":"Iro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0741-4049","authenticated-orcid":false,"given":"Matti","family":"Sievert","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,5,10]]},"reference":[{"key":"4125_CR1","doi-asserted-by":"publisher","first-page":"161","DOI":"10.1016\/j.suronc.2014.07.002","volume":"23","author":"Y Carter","year":"2014","unstructured":"Carter Y, Yeutter N, Mazeh H (2014) Thyroglossal duct remnant carcinoma: beyond the Sistrunk procedure. 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