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Adult Thyroglossal Duct Carcinoma of Thyroid Epithelial Origin: A Retrospective Observational Study

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Indian Journal of Otolaryngology and Head & Neck Surgery Aims and scope Submit manuscript

Abstract

Thyroglossal duct or cyst carcinoma (TGDCCa) is uncommon. Current treatments follow those of thyroid cancer but controversy exists over the extent of thyroid and lymph node surgery. Our aim was to study the presentation, treatment strategies and clinical outcomes in patients presenting with TGDCCa. Of 637 patients diagnosed with thyroid cancer over 15-years, 4 patients (0.6 %) with TGDCCa were identified. Two patients were diagnosed outside this time period. Details of demographics, presentation, diagnosis, management and outcomes of all 6 patients were reviewed. 5 females and 1 male with a median (range) age of 41 (21–70) years were treated for TGDCCa. Five patients were diagnosed after initial surgery for thyroglossal cyst—cyst excision (n = 2) and Sistrunk’s procedure (n = 3). This was followed by a total thyroidectomy (TT) in 4 of these patients. One patient underwent Sistrunk’s operation, total thyroidectomy and central neck dissection at the first operation. I131 therapy was used in patients who had TT. Three patients had additional tumour foci in the thyroid. However, lymph node recurrence occurred in 2 patients at 6 and 16 months. At a median follow up of 55 months, all 6 patients were disease free. All patients had papillary TGDCCa and did well with conventional treatment as for thyroid cancer. The extent of treatment required is debatable as half the patients had additional thyroid foci and no patient had clinically or radiologically involved lymph nodes at presentation. A systematic review of published cases will help summarise the existing knowledge base.

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References

  1. 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 79:134–139 discussion 140-131

    Article  PubMed  Google Scholar 

  2. Carter Y, Yeutter N, Mazeh H (2014) Thyroglossal duct remnant carcinoma: beyond the sistrunk procedure. Surg Oncol 23:161–166

    Article  PubMed  PubMed Central  Google Scholar 

  3. Hirshoren N, Neuman T, Udassin R, Elidan J, Weinberger JM (2009) The imperative of the sistrunk operation: review of 160 thyroglossal tract remnant operations. Otolaryngol Head Neck Surg 140:338–342

    Article  PubMed  Google Scholar 

  4. Soni S, Poorey VK, Chouksey S (2014) Thyroglossal Duct Cyst, Variation in Presentation, Our Experience. Indian J Otolaryngol Head and Neck Surg 66:398–400

    Article  CAS  Google Scholar 

  5. Peretz A, Leiberman E, Kapelushnik J, Hershkovitz E (2004) Thyroglossal duct carcinoma in children: case presentation and review of the literature. Thyroid 14:777–785

    Article  PubMed  Google Scholar 

  6. Thabet H, Gaafar A, Nour Y (2011) Thyroglossal duct cyst: variable presentations. Egypt J Ear, Nose, Throat and Allied Sciences 12:13–20

    Article  Google Scholar 

  7. Mittal MK, Malik A, Sureka B, Thukral BB (2012) Cystic masses of neck: a pictorial review. Indian J Radiol and Imaging 22:334–343

    Article  Google Scholar 

  8. Mondin V, Ferlito A, Muzzi E, Silver CE, Fagan JJ, Devaney KO, Rinaldo A (2008) Thyroglossal duct cyst: personal experience and literature review. Auris Nasus Larynx 35:11–25

    Article  PubMed  Google Scholar 

  9. Weiss SD, Orlich CC (1991) Primary papillary carcinoma of a thyroglossal duct cyst: report of a case and literature review. Br J Surg 78:87–89

    Article  CAS  PubMed  Google Scholar 

  10. Renard TH, Choucair RJ, Stevenson WD, Brooks WC, Poulos E (1990) Carcinoma of the thyroglossal duct. Surg Gynecol Obstet 171:305–308

    CAS  PubMed  Google Scholar 

  11. 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 13:745–752

    Article  PubMed  Google Scholar 

  12. Luna-Ortiz K, Hurtado-Lopez LM, Valderrama-Landaeta JL, Ruiz-Vega A (2004) Thyroglossal duct cyst with papillary carcinoma: what must be done? Thyroid 14:363–366

    Article  PubMed  Google Scholar 

  13. Yang YJ, Haghir S, Wanamaker JR, Powers CN (2000) Diagnosis of papillary carcinoma in a thyroglossal duct cyst by fine-needle aspiration biopsy. Arch Pathol Lab Med 124:139–142

    CAS  PubMed  Google Scholar 

  14. Haymart MR, Greenblatt DY, Elson DF, Chen H (2008) The role of intraoperative frozen section if suspicious for papillary thyroid cancer. Thyroid 18:419–423

    Article  PubMed  Google Scholar 

  15. Narayana Moorthy S, Arcot R (2011) Thyroglossal duct cyst—more than just an embryological remnant. Indian J Surg 73:28–31

    Article  PubMed  Google Scholar 

  16. Rohof D, Honings J, Theunisse HJ, Schutte HW, van den Hoogen FJ, van den Broek GB, Takes RP, Wijnen MH, Marres HA (2014) Recurrences after thyroglossal duct cyst surgery: results in 207 consecutive cases and review of the literature. Head Neck 37(12):1699–1704

    Article  PubMed  Google Scholar 

  17. Charbel N, Chadi F, Bassam T (2012) Two cases of papillary carcinoma in a thyroglossal duct cyst. Egypt J Ear, Nose, Throat and Allied Sci 13:133–135

    Article  Google Scholar 

  18. Ishay A, Elmalah I, Luboshitzky R (2008) Papillary carcinoma in a thyroglossal duct cyst. Isr Med Assoc J 10:312–313

    PubMed  Google Scholar 

  19. Dzodic R, Markovic I, Stanojevic B, Saenko V, Buta M, Djurisic I, Oruci M, Pupic G, Milovanovic Z, Yamashita S (2012) Surgical management of primary thyroid carcinoma arising in thyroglossal duct cyst: an experience of a single institution in Serbia. Endocr J 59:517–522

    Article  PubMed  Google Scholar 

  20. Kum CK, Goh P, Teh M (1993) Papillary carcinoma arising in a thyroglossal cyst. Aust N Z J Surg 63:738–740

    Article  CAS  PubMed  Google Scholar 

  21. Tharmabala M, Kanthan R (2013) Incidental thyroid papillary carcinoma in a thyroglossal duct cyst—management dilemmas. Int J Sur Case Rep 4:58–61

    Article  Google Scholar 

  22. Sprinzl GM, Koebke J, Wimmers-Klick J, Eckel HE, Thumfart WF (2000) Morphology of the human thyroglossal tract: a histologic and macroscopic study in infants and children. Ann Otol Rhinol Laryngol 109:1135–1139

    Article  CAS  PubMed  Google Scholar 

  23. LiVolsi VA, Perzin KH, Savetsky L (1974) Carcinoma arising in median ectopic thyroid (including thyroglossal duct tissue). Cancer 34:1303–1315

    Article  CAS  PubMed  Google Scholar 

  24. Martin Perez M, Burguno Garcia M, Echevarria Iturbe C, Martinez Perez D (1993) Papillary carcinoma arising in a thyroglossal duct cyst: report of two cases. J Oral Maxillofac Surg 51:89–93

    Article  CAS  PubMed  Google Scholar 

  25. Pollock WF, Stevenson EO (1966) Cysts and sinuses of the thyroglossal duct. Am J Surg 112:225–232

    Article  CAS  PubMed  Google Scholar 

  26. Patti G, Ragni G, Calisti A (2000) Papillary thyroid carcinoma in a thyroglossal duct cyst in a child. Med Pediatr Oncol 34:67–69

    Article  CAS  PubMed  Google Scholar 

  27. Pribitkin EA, Friedman O (2002) Papillary carcinoma in a thyroglossal duct remnant. Arch Otolaryngol Head Neck Surg 128:461–462 discussion 463–464

    Article  PubMed  Google Scholar 

  28. Flynn RW, Bonellie SR, Jung RT, MacDonald TM, Morris AD, Leese GP (2010) Serum thyroid-stimulating hormone concentration and morbidity from cardiovascular disease and fractures in patients on long-term thyroxine therapy. J Clin Endocrinol Metab 95:186–193

    Article  CAS  PubMed  Google Scholar 

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Authors and Affiliations

  1. University of Sheffield, Beech Hill Rd, Sheffield, South Yorkshire, S10 2RX, M14 7DN, England, UK

    Rahim Akram & Joseph J Wiltshire

  2. Weston Park Hospital, Sheffield, Whitham Rd, Sheffield, Sheffield, South Yorkshire, S10 2SJ, England, UK

    Jonathan Wadsley

  3. Sheffield Teaching Hospitals, Sheffield, Glossop Rd, Sheffield, South Yorkshire, S10 2JF, England, UK

    Sabapathy P Balasubramanian

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  1. Rahim Akram
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  2. Joseph J Wiltshire
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  4. Sabapathy P Balasubramanian
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Correspondence to Rahim Akram.

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This article does not contain any studies with human participants or animals performed by any of the authors.

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Informed consent was obtained from all individual participants included in the study.

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Akram, R., Wiltshire, J.J., Wadsley, J. et al. Adult Thyroglossal Duct Carcinoma of Thyroid Epithelial Origin: A Retrospective Observational Study. Indian J Otolaryngol Head Neck Surg 68, 522–527 (2016). https://doi.org/10.1007/s12070-016-1019-8

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  • DOI: https://doi.org/10.1007/s12070-016-1019-8

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