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Year : 2013  |  Volume : 10  |  Issue : 1  |  Page : 20-22

Papillary carcinoma in a thyroglossalduct cyst with neck nodal metastases

1 Consultant Head and Neck Oncologist, Prince Aly Khan Hospital, Mumbai, India
2 Department of Internal Medicine, FLMC, Florida, USA
3 Department of ENT, Government Medical College Hospital, Srinagar, India
4 Department of Radiology, Government Medical College Hospital, Srinagar, India

Date of Web Publication10-Jan-2013

Correspondence Address:
Arsheed H Hakeem
Consultant Head and neck Oncology, Prince Aly Khan Hospital, Mumbai - 400 010
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-0354.105842

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Although thyroglossal duct cysts are the most common form of congenital anomalies in the neck, diagnosis of cancer in such cysts is rare with a reported incidence of 1.3% of all thyroglossal cysts. Presence of the metastatic neck nodes is still more uncommon. Papillary thyroid carcinoma is the most frequently encountered carcinoma in thyroglossal duct cyst. We present a case of young female with papillary carcinoma in the thyroglossal duct cyst with metastatic lymph nodes in level IB.

Keywords: Papillary thyroid carcinoma, sistrunk procedure, thyroglossal duct cysts

How to cite this article:
Hakeem AH, Hakeem IH, Ahmed R, Kirmani OS. Papillary carcinoma in a thyroglossalduct cyst with neck nodal metastases. Thyroid Res Pract 2013;10:20-2

How to cite this URL:
Hakeem AH, Hakeem IH, Ahmed R, Kirmani OS. Papillary carcinoma in a thyroglossalduct cyst with neck nodal metastases. Thyroid Res Pract [serial online] 2013 [cited 2022 Aug 17];10:20-2. Available from: https://www.thetrp.net/text.asp?2013/10/1/20/105842

  Introduction Top

Thyroglossal duct carcinoma is uncommon entity. Brenato was first to describe thyroglossal duct carcinoma in 1911, since that more than 200 cases have been reported in the world literature mostly as single case reports. [1] Papillary carcinoma in thyroglossal duct is a rare entity with a reported frequency of 1.8% (7 cases among 371 thyroglossal ducts or cysts) according to LiVolsiand, 1.7% (2 cases among 116 cysts) according to Keeling. [2],[3],[4] Presence of the metastatic nodes is still rarer in this condition. Van Vuuren reported a female: Male ratio of 1.6:1. [5] Fernandez, found that 55% of patients with thyroglossal duct carcinoma were younger than 40 years of age and approximately 30% were aged between 20 and 30 years. [6] LiVolsi reported that slowly growing lesions may continue to enlarge over several weeks to several years. [3] The size of the mass is variable but most are in the 2-4cm range. We report a case of papillary carcinoma in a thyroglossal duct cyst with metastatic nodes in right level IB region in a 25 year old female.

  Case Report Top

A 25 year-old female presented with a painless midline swelling in the front of the upper neck, which had been present for two years and had been increasing in size. Examination revealed a mobile 3 × 3 cm diameter cystic mass in the anterior midline of the neck in the hyoid region which moved on deglutition [Figure 1]. The thyroid gland was normal on palpation and two regional lymph nodes were palpable in right level IB region. Fine needle aspiration cytology revealed papillary carcinoma in the thyroglossal duct cyst and the level IB node. Magnetic resonance imaging revealed well circumscribed lesion with smooth outline with two metastatic nodes in the right upper neck [Figure 2]. MR Imaging did not pick up any abnormality of the thyroid gland [Figure 3]. Rest of the metastatic work up did not reveal any other foci of metastasis. Therefore, preoperative diagnosis of papillary carcinoma in thyroglossal duct cyst with metastatic nodes was made. Total thyroidectomy with Sistrunk's procedure with right selective neck dissection was done preserving all the three non lymphatic structures [Figure 4]. Histological examination showed hyalinised fibrous wall with thyroid follicles and scattered aggregates of lymphocytes. The cyst lining was thrown into papillae, composed of fibrovascular cores covered by cubical epithelium. Many of the stromal cores contained calcospherites or psammoma bodies. The features were of a papillary carcinoma arising within a thyroglossal cyst. There was no evidence of invasion beyond the cyst wall and two nodes showed metastases.
Figure 1: Clinical photograph showing thyoglossal duct cyst carcinoma front view

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Figure 2: MRI scan axial view at the level of the hyoid showing thyoglossal duct carcinoma with 2 level I B nodes

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Figure 3: MRI Scan at the level of the thyroid gland showing bilateral normal thyroid lobes

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Figure 4: Clinical picture of the surgical field after total thyroidectomy with modified neck dissection

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A post - operative isotope I 131 scan and ablation was done after five weeks. Three years post operative she is controlled and does not show any evidence of disease.

  Discussion Top

It is a well established fact that the thyroglossal duct contains normal thyroid tissue. So this thyroid tissue has the same potential to develop malignancy as the normal thyroid gland. Therefore some authors believe that the malignancy arises denovo in the thyroglossal duct cyst. [7],[8],[9] The presence of ectopic thyroid tissue in a thyroglossal duct cyst varies from 1.5% to 45% of cases and can be explained on the basis of the gland embryology. In this paper we report a case of papillary carcinoma in thyroglossal duct cyst with neck node metastases with management of the case.

Literature search revealed few cases of thyroglossal duct papillary carcinoma with neck metastases. Often the patient has noticed the mass many months or even years before presentation. Carcinoma should be suspected in any thyroglossal duct cyst that is hard, fixed and irregular or which has undergone recent change and associated palpable lymphnodes. Rapid enlargement may also induce suspicion, but infections in this area may also present similar clinical picture. Full knowledge of the embryogenesis of the thyroid gland and the pathology of carcinomas arising in these structures is mandatory for the appropriate management of thyroglossal duct diseases. [8],[10],[11]

The important controversy in such cases is for management of the thyroid gland. This is because between 11% and 33% of the cases reported have also had a second focus of tumor in the thyroid gland. [12] These second foci are often not detectable by either physical examination or imaging as is true with our case. Weiss and Orlich believed that because papillary thyroid carcinoma has a prolonged course in the absence of objective suspicion of a thyroid mass only long-term follow-up is warranted. [1] In contrast, Heshmati et al, recommended thyroidectomy on all patients with thyroglossal duct carcinoma. [12]

Therefore significant attention must be paid to the pre-operative evaluation of a patient affected by a thyroglossal duct cyst carcinoma which should include a complete physical examination, accurate head and neck examination and palpation of the thyroid gland, thyroid function laboratory tests and also a thyroid scan and USG to rule out synchronous lesion in the thyroid and metastatic neck nodes. Although removal of the thyroglossal tract is a standard procedure, the actual extent of surgery depends on the degree of tumor involvement as detected through USG neck, thyroid scan and surgery. If the thyroid and lymph nodes are largely normal on clinical and radiological examination Sistrunk's procedure, is justified. [6] If however, USG and thyroid scan shows nodules in the thyroid gland or metastatic lymph nodes or thyroid mass is identified during surgery a thyroidectomy is recommended which may include also regional lymph node dissection as indicated.

In our case preoperative diagnosis of thyroglossal duct papillary carcinoma with neck node metastases was made and confirmed on fine needle aspiration cytology and imaging. Therefore, total thyroidectomy with sistrunk's operation with neck dissection was done [Figure 4]. Six weeks post surgery, I 131 scan and ablation was done which did not pick up significant disease. At more than three years of follow up she is free of disease.

  Conclusion Top

Malignancy within a thyroglossal duct cyst is rare and association with metastatic neck nodes is more uncommon. This condition can be diagnosed preoperatively on high clinical suspicion, imaging and FNAC. Once diagnosed therapy includes total thyroidectomy with sistrunk's operation and appropitate neck dissection. Radioactive iodine ablation and thyroxin are used as adjuvant, as is the case for differentiated thyroid cancers.

  References Top

1.Brenato H. Struma aberrata lingual mitdruzenmetastasen. Deutsch Med Wschr 1911;37:665.  Back to cited text no. 1
2.Roses DF, Snively SL, Phelps RG, Cohen N, Blum M. Carcinoma of the thyroglossal duct. Am J Surg 1983;145:266-9.  Back to cited text no. 2
3.LiVolsi VA, Perzin KH, Savetsky L. Carcinoma arising in median ectopic thyroid (including thyroglossal duct tissue). Cancer 1974;34:1303-15.  Back to cited text no. 3
4.Keeling JH, Ochsner A. Carcinoma in thyroglossal duct remnants; Review of literature and report of 2 cases. Cancer 1959;12:596-600.  Back to cited text no. 4
5.Van Vuuren PA, Balm AJ, Greger RT, Hilgers FJ, Loftus BM, Delprat CC, et al. Carcinoma arising in thyroglossal remnants. Clin Otolaryngol Allied Sci 1994;19:509-15.  Back to cited text no. 5
6.Fernandez JF, Ordonez NG, Schultz PN, Samaan NA, Hickey RC. Thyroglossal duct carcinoma. Surgery 1991;110:928-34.  Back to cited text no. 6
7.Kazemi M, Assadi M, Kazemi AA, Ghazvini LA. Primary papillary carcinoma of thyroglossal duct cyst. Hell J Nucl Med 2006;9:39-40.  Back to cited text no. 7
8.Astl J, Dusková J, Kraus J, Vlcek P, Kodet R, Last°uvka P, et al. Coincidence of thyroid tumor and thyroglossal duct remnants. Review of the literature and presentation of three cases. Tumori 2003;89:314-20.  Back to cited text no. 8
9.Ferrer C, Ferrández A, Dualde D, Rodriguez M, Ferrer E, Pinazo J, et al. Squamous cell carcinoma of the thyroglossal duct cyst: Report of a new case and literature review. J Otolaryngol 2000;29:311-4.  Back to cited text no. 9
10.Organ GM, Organ CH Jr. Thyroid gland and surgery of the thyroglossal duct: Exercise in applied embryology. World J Surg 2000 ; 24:866-90.  Back to cited text no. 10
11.Allard RH. The thyroglossal cyst. Head Neck Surg 1982;5:134-46.  Back to cited text no. 11
12.Weiss SD, Orlich CC. Primary papillary carcinoma of a thyroglossal duct cyst: Report of a case and literature review. Br J Surg 1991;78:87-9.  Back to cited text no. 12


  [Figure 1], [Figure 2], [Figure 3], [Figure 4]


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