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Year : 2016  |  Volume : 13  |  Issue : 2  |  Page : 77-79

Conservatively managed thyroglossal duct cyst carcinoma with lymph node metastasis: Long-term result

1 Tepecik Training and Research Hospital Head and Neck Surgery Clinic, Izmir, Turkey
2 Derince Training and Research Hospital Pathology Clinic, Kocaeli, Turkey
3 Derince Training and Research Hospital Head and Neck Surgery Clinic, Kocaeli, Turkey

Date of Web Publication1-Jun-2016

Correspondence Address:
Dr. Ilker Burak Arslan
Izmir Tepecik Egitimve Arastirma Hastanesi KBB Klinigi, Gaziler Caddesi No: 468, 35170 Yenisehir, Izmir
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-0354.157920

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Thyroid papillary carcinomas of thyroglossal duct cysts (TGDCs) are quite uncommon. Despite the identification of risk factors, the treatment course is controversial. A 34-year-old woman suffering sudden growth of a mass in the neck was clinically diagnosed TGDC. Ultrasonography and computed tomography (CT) showed irregular heterogeneous mass, with a large cystic component consisting of calcification and 7 × 6 mm solid smoothly contoured mass close to the left isthmus. Thyroid scintigraphy exposed normal thyroid gland uptake and a small cold nodule near the isthmus. Fine-needle aspiration cytology revealed signs of well-differentiated papillary TGDCs carcinoma. Sistrunk's procedure with an anterior neck dissection was performed. Postoperative histopathological examination revealed well-differentiated papillary carcinoma arising from thyroglossal cyst and lymph node metastasis. No further treatment was carried out and she was followed-up without recurrence for 44 months. A conservative procedure should be performed in well-differentiated papillary carcinoma of the TGDC.

Keywords: Lymphadenectomy, papillary carcinoma, Sistrunk's procedure, thyroglossal duct cyst

How to cite this article:
Arslan IB, Eruyar AT, Gumussoy M, Genc S. Conservatively managed thyroglossal duct cyst carcinoma with lymph node metastasis: Long-term result. Thyroid Res Pract 2016;13:77-9

How to cite this URL:
Arslan IB, Eruyar AT, Gumussoy M, Genc S. Conservatively managed thyroglossal duct cyst carcinoma with lymph node metastasis: Long-term result. Thyroid Res Pract [serial online] 2016 [cited 2022 Dec 5];13:77-9. Available from: https://www.thetrp.net/text.asp?2016/13/2/77/157920

  Introduction Top

Abnormalities in the pattern of thyroid development include arrested growth, accessory tissues and cystic development of the thyroglossal duct. Thyroglossal duct cysts (TGDCs), generally occurring just inferior to the hyoid bone is common; but malignant degeneration is rare, occurring only in 1% of the cases.[1] TGDC carcinoma is approximately 40 years of age and it shows female predominance.[2] Most benign TGDC contain microscopic foci of thyroid tissue, which are presumably the source of thyroglossal duct carcinoma.[3] The histological findings of thyroglossal duct carcinoma are most commonly papillary carcinoma (75-80%); these were either large or rapidly increasing in size TGDC.[4] Invasion into surrounding soft tissue is seen in only 17% of thyroglossal duct carcinomas. Metastatic disease is present in 1.3%, which is much lower than the rate from carcinoma arising in the thyroid gland.[4]

We report a rare case of papillary carcinoma of TGDC with lymph node metastasis treated without thyroidectomy and discuss the dilemma in treatment modalities.

  Case Report Top

A 34-year-old woman admitted with a right paramedian neck swelling that had been presented for 5 years and increased in size over the last 6 months. She was euthyroid and clinically diagnosed to have a TGDCs. The mass was a 3 × 4 cm soft, nontender mass below the hyoid bone. The thyroid gland was palpated at its original position and no cervical lymph nodes were found. Normal thyroid gland with mild hypertrophy and a 31 × 20 × 42 cm irregular heterogeneous mass, with a large cystic component consisting of calcification, between the hyoid bone and cricoid cartilage was visualized on ultrasonography (USG). Also a 7 × 6 mm solid, hypoechoic, and smoothly contoured lymph node distinguished from left isthmus were defined. Thyroid scintigraphy revealed normal thyroid gland uptake and a small cold nodule near the isthmus. Scintigraphic uptake was not observed from thyroid tissue inside the TGDC. Neck computed tomography (CT) showed a 4 × 3 cm heterogeneously enhancing mass with calcification [Figure 1] and a small mass similar dense with thyroid. Fine-needle aspiration cytology revealed well-differentiated papillary carcinoma of TGDCs.
Figure 1: Papillary carcinoma of the thyroglossal duct cyst. Axial contrast-enhanced computed tomography (CT) reveals a mass contained cystic and solid elements (white arrow). Calcifications are identified within the mass (black arrow)

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Sistrunk's procedure with anterior neck dissection and sentinel lymph node biopsy was performed. Frozen section analysis revealed papillarycarcinomaof the TGDC and metastasis of the preoperatively detected lymphnode. Neither thyroid nodule nor cervical lymphadenopathy in the lateral neck was detected by palpation during surgery. Operation was ended without thyroidectomy.

Post operative histopathological examination showed well-differentiated papillary carcinoma of both TGDC (25 × 15 × 11 mm) and lymph node (9 × 6 × 4 mm). She has remained free of disease on postoperative 44 months follow-up [Figure 2].
Figure 2: Axial CT scan of the normal thyroid gland

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  Discussion Top

There is a great deal of controversy around the surgical treatment of papillary carcinoma of TGDC. In patients with abnormal cervical lymph nodes by clinical or radiographic criteria, the Sistrunk's procedure combined with thyroidectomy and lymphadenectomy is widely accepted.[3] However, some surgeons consider that the Sistrunk's procedure and resection of the ectopic thyroid tissue alone to be sufficient for patients with had no signs of thyroid lesion.[5] Plaza et al., recommended more aggressive treatment including total thyroidectomy and 131 I (RAI) therapy for patients deemed to be at high risk, defined high risk characteristics as being older than 45 years, having a history of radiation exposure, a tumor in the thyroid on radiological evaluation, having clinical or radiological nodes, a tumor more than 1.5 cm in diameter, cyst wall invasion, or positive margins on histopathologic examination.[6] Choi et al., examined this recommendation for 56 subsequent cases, cannot find statistically significant relation to support Plaza's suggestion.[2] Although the present case include high risk due to tumor diameter and nodal metastasis according to above recommendation, Sistrunk's procedure with anterior neck dissection seems useful according to the long-term follow-up.

Any disease affecting the TGDC may also involve the ectopic thyroid and thyroid gland, including malignancy. The number of reported incidences of thyroid carcinoma, concomitant with TGDC carcinoma is between 11 and 33%, and the foci of carcinomas are small (ranging from 0.2 to 1.5 cm).[7] Nearly 1-4% of thyroglossal duct carcinomas were found to be locally invasive, while 11% were found to include metastasis to cervical lymph nodes. Prophylactic node dissection is not beneficial in papillary carcinoma of thyroid gland without palpable lymphadenopathy.[4],[8],[9] Total thyroidectomy could be a good alternative for papillary carcinoma of the TGDC patients to decrease the recurrence rate due to characteristic of multicentricity and to make postoperative follow-up easier by monitoring thyroglobulin level.[10],[11] In this treatment modality, over two-third of the patients became thyroid hormone addict in addition to the surgery-related complications. Sistrunk's procedure should be adequate in papillary carcinoma of the TGDC with utrasonographically normal thyroid gland because of the benign behavior than the primary thyroid gland papillary carcinoma. Some authors concluded that the addition of total thyroidectomy to the Sistrunk operation did not have a significant impact on recurrence and survival.[8],[12],[13] Regular follow-up to detect any recurrence in the thyroid gland is essential in cases where the thyroid gland is not resected. USG, capable of detecting thyroid mass as small as 2 mm in size, should contribute identification of recurrence.[14] Papillary cancer is known to have a prolonged clinical course; therefore, long-term follow-up (>20 years) is recommended. We prefer to close follow-up with USG and no further treatment was carried out.[8]

The diagnosis of well-differentiated thyroglossal duct carcinoma can be made preoperatively with FNA.[15] Preoperative thyroid scans confirms the presence of ectopic thyroid tissue in 33% of TGDCs.[16] Such scans may play an important role in establishing diagnosis of lymph node metastasis including functional thyroid tissue in well-differentiated papillary carcinoma of TGDC as in the present case. Also this should be a prognostication for noninvasive surgery.

Although surgery is mandatory for well-differentiated papillary carcinoma in TGDC, it is unclear that Sistrunk's procedure alone to be sufficient or total thyroidectomy and radioactive iodine ablation procedure is necessary. The role of the scintigraphic uptake on tumor differentiation and its effect on surgery must be further evaluated.

  References Top

Sevinç AI, Unek T, Canda AE, Guray M, Kocdor MA, Saydam S, et al. Papillary carcinoma arising in subhyoid ectopic thyroid gland with no orthotopic thyroid tissue. Am J Surg 2010;200:e17-8.  Back to cited text no. 1
Choi YM, Kim TY, Song DE, Hong SJ, Jang EK, Jeon MJ, et al. Papillary thyroid carcinoma arising from a thyroglossal duct cyst: A single institution experience. Endocr J 2013;60:665-70.  Back to cited text no. 2
Branstetter BF, Weissman JL, Kennedy TL, Whitaker M. The CT appearance of thyroglossal duct carcinoma. AJNR Am J Neuroradiol 2000;21:1547-50.  Back to cited text no. 3
Sturniolo G, Moleti M, Violi MA, Di Bella B, Presti S, Trimarchi F, et al. Prevalence of thyroglossal duct cyst carcinoma in adults having surgery for thyroglossal duct cysts. Thyroid 2012;22:1191-2.  Back to cited text no. 4
Chrisoulidou A, Iliadou P, Doumala E, Mathiopoulou L, Boudina M, Alevizaki M, et al. Thyroglossal duct cyst carcinomas: Is there a need for thyroidectomy? Hormones (Athens) 2013;12:522-8.  Back to cited text no. 5
Plaza CP, Lopez ME, Carrasco CE, Meseguer LM, Perucho Ade L. 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 2006;13:745-52.  Back to cited text no. 6
Park MH, Yoon JH, Jegal YJ, Lee JS. Papillary thyroglossal duct cyst carcinoma with synchronous occult papillary thyroid microcarcinoma. Yonsei Med J 2010;51:609-11.  Back to cited text no. 7
Yang S, Park KK, Kim JH. Papillary carcinoma arising from throglossal duct cyst with thyroid and lateral neck metastasis. Int J Surg Case Rep 2013;4:704-7.  Back to cited text no. 8
Wada N, Duh QY, Sugino K, Iwasaki H, Kameyama K, Mimura T, et al. Lymph node metastasis from 259 papillary thyroid microcarcinomas frequency, pattern of occurrence and recurrence, and optimal strategy for neck dissection. Ann Surg 2003;237:399-407.  Back to cited text no. 9
Pellegriti G, Lumera G, Malandrino P, Latina A, Masucci R, Scollo C, et al. Thyroid cancer in throglossal duct cysts requires a specific approach due to its unpredictable extension. J Clin Endocrinol Metab 2013;98:458-65.  Back to cited text no. 10
Dzodic R, Markovic I, Stanojevic B, Saenko V, Buta M, Djurisic I, et al. Surgical management of primary thyroid carcinoma arising in thyroglossal duct cyst: An experience of a single institution in Serbia. Endocr J 2012;59:517-22.  Back to cited text no. 11
Patel SG, Escrig M, Shaha AR, Singh B, Shah JP. Management of well-differentiated thyroid carcinoma presenting within a thyroglossal duct cyst. J Surg Oncol 2002;79:134-9.  Back to cited text no. 12
Yü Y, Wang XL, Xu ZG, Liu SY, Wang JY. Management of thyroglossal duct carcinoma: Report of five cases. Zhonghua Er Bi Yan Hou Tou Jing Wai Ke Za Zhi 2012;47:1013-6.  Back to cited text no. 13
Polyzos SA, Kita M, Avramidis A. Thyroid nodules-Stepwise diagnosis and management. Hormones (Athens) 2007;6:101-19.  Back to cited text no. 14
Jang DW, Sikora AG, Leytin A. Thyroglossal duct cyst carcinoma: Case report and review of the literature. Ear Nose Throat J 2013;92:E12-4.  Back to cited text no. 15
Tharmabala M, Kanthan R. Incidental thyroid papillary carcinoma in a thyroglossal duct cyst-management dilemmas. Int J Surg Case Rep 2013;4:58-61.  Back to cited text no. 16


  [Figure 1], [Figure 2]


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