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Table of Contents
REPORT
Year : 2010  |  Volume : 12  |  Issue : 1  |  Page : 22-25

Papillary carcinoma of the thyroglossal duct cyst: A tertiary healthcare centre experience


1 Resident, Department of Otolaryngology - Head and Neck Surgery, Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia
2 Associate Professor and Consultant, Department of Otolaryngology - Head and Neck Surgery, Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia

Date of Web Publication24-Dec-2019

Correspondence Address:
MBBS Faisal Tarif Zawawi
P O Box 10659, Jeddah 21443
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1319-8491.273969

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  Abstract 


Although thyroglossal duct cyst (TDC) is a common problem, papillary carcinoma is a very rare finding. The management detail and protocol is yet to be well defined. This paper aims to highlight the experience of King Abdulaziz University Hospital in papillary carcinoma of TDC in the past five years. Four patients out of 315 patients reviewed retrospectively were diagnosed to have papillary carcinoma of the TDC. Fifty per cent had involvement of the thyroid. One patient (25%) was a child.
Conclusion: Papillary carcinoma of TDC has a favorable prognosis if managed properly by an experienced team.

Keywords: Papillary carcinoma, thyroglossal duct cyst,


How to cite this article:
Zawawi FT, Al-Muhayawi S. Papillary carcinoma of the thyroglossal duct cyst: A tertiary healthcare centre experience. Saudi J Otorhinolaryngol Head Neck Surg 2010;12:22-5

How to cite this URL:
Zawawi FT, Al-Muhayawi S. Papillary carcinoma of the thyroglossal duct cyst: A tertiary healthcare centre experience. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2010 [cited 2022 Dec 2];12:22-5. Available from: https://www.sjohns.org/text.asp?2010/12/1/22/273969




  Introduction Top


Thyroglossal duct cyst (TDC) is a congenital anomaly that forms an asymmetric midline neck mass. It is the most common midline neck mass found in children [1]. It is estimated that 7% of the population have some form of thyroglossal duct remnant [2],[3]. Malignancy is a rare transformation and is found in less than 1% of the TDC. In fact, not more than 250 cases have been reported, 22 of which are in the children [4]. The most common type is papillary carcinoma that accounts for more than 80% of all malignancies in TDC [5]. Less common are mixed papillary-follicular (8%) and squamous cell carcinoma (6%) [5]. Other reported malignancies are Follicular carcinoma, anaplastic carcinoma, adenocarcinoma and epidermoid carcinoma [5].

This was a retrospective study of 5 years experience of King Abdulaziz University Hospital (A tertiary health care centre),.Jeddah, Saudi Arabia with papillary carcinoma of TDC.


  Method and Material Top


The charts of 315 patients who had thyroid and TDC related surgeries at King Abdulaziz University Hospital, Jeddah, Saudi Arabia in the past 5 years were reviewed. Four patients were diagnosed as papillary carcinoma of the TDC. The medical history, investigations, surgical procedures and histopathology reports were recorded for those 4 patients.


  Results Top


Three adults and one child were diagnosed as papillary carcinoma of TDC. One adult and 1 child had malignancy in the thyroid gland. Two Adults had TDC excision done in a different centre [Table 1].
Table 1: This table shows the details of the four patients diagnosed to have Papillary carcinoma of TDC.

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Patient 1

A 47 years old patient was seen with a history of a midline neck mass for 2 years. He was attended to by a general surgeon and a the provisional diagnosis was TDC. A simple TDC excision was performed but the final histopathology report was papillary carcinoma. He was referred to the Otolaryngology, Head and Neck Surgery Clinic at King Abdulaziz University Hospital (KAUH), Jeddah, Saudi Arabia. The patient did not have a family history of thyroid or TDC cancers and did not receive radiotherapy for any other disease. His thyroid function test was normal. Examination of the ears, nose and throat including the nasopharynx were normal. He had an infrahyoid midline scar, swelling and indurations. No cervical lymphadenopathies were found. Neck ultrasound was performed which revealed bilateral multiple small nodules on the thyroid gland with possible remnants of the thyroglossal duct tract. The decision was to go for a total thyroidectomy. Sistrunk procedure (SP) was performed; remnants of the thyroglossal tract were found and excised. Total thyroidectomy with central neck nodal dissection was carried out in the same setting [Figure 1]. The final histopathology report was chronic thyroiditis without evidence of malignancy.
Figure 1: The Thyroid gland with bilateral multiple small nodules and thyroglossal duct cyst tract remnant. Histopathology of this specimen was chronic thyroiditis without evidence of malignancy

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The patient was put on TSH suppression therapy and was followed up using thyroglobulin antibodies. His 3 years follow up was cancer free.

Patient 2

A 13 years old girl presented to our clinic complaining from a central neck lump for one year. The lump was round in shape and was progressive in size. It was not painful nor did it affect her swallowing or breathing. She did not suffer from any other illnesses nor had any other complaints. She had no previous exposure to radiation and she did not have a family history of thyroid cancer.

On examination, the neck lump was at the midline measuring 2x2 cm, cystic in nature and was not tender or hot. The lump moved with swallowing and with protrusion of the tongue. The rest of the ear, nose and throat examination was normal. Neck Ultrasound showed 2.2 x 2.5 cm thyroglossal duct cyst. The thyroid gland was normal.

Due to the patient’s preferences, surgery was performed 7 months later. Thyroglossal duct cyst excision was done. The histopathologic examination showed papillary carcinoma. Neck ultrasound was repeated, which showed 1 x 0.8 cm left lobe thyroid nodule. Thyroid function test was normal. SP and total thyroidectomy with central neck nodal dissection were performed. No extra thyroidal extensions or enlarged lymph nodes were noted. Histopathologic examination of the thyroid gland was positive for papillary carcinoma. Post operatively the patient was referred to a pediatric endocrinologist and an oncologist in another city, due to social reasons, to decide on TSH suppression therapy and radioactive Iodine 131 (I 131) body scan and ablation. The patient was lost to follow up.

Patient 3:

A 19 years old male patient presented to our clinic with a 1-year history of a midline neck swelling. He had a history of TDC excision done in another centre two years earlier. Neck Ultrasound showed a midline cyst suggestive of persistent TDC and a normal looking thyroid gland. TDC excision was performed. The histopathology report was papillary carcinoma of the TDC. SP and total thyroidectomy with central nodal dissection were carried out two weeks later. The histopathology showed foci of papillary carcinoma in the right lobe of the thyroid gland. The patient was put on TSH suppression therapy and was referred to endocrinology and oncology for radioactive iodine body scan and ablation therapy.

The patient was free of cancer at two years follow up.

Patient 4:

A 41 years old gentleman presented with a one-year history of a midline neck swelling. Neck Ultrasound showed midline cystic mass suggestive of TDC with a normal looking thyroid. TDC excision was performed and histopathology confirmed the diagnosis of papillary carcinoma of the TDC. SP and total thyroidectomy were carried out. The histopathology report of this specimen was benign thyroid gland. The patient was put in TSH replacement therapy and was referred to oncology and endocrinology for radioactive Iodine body scan. The patient was free of cancer at two years follow up.


  Discussion Top


During the development of the thyroid gland, a diverticulum descends from the tongue base anterior to the trachea. A remnant of this diverticulum is called the thyroglossal duct which usually disappears by the 7th week of intrauterine life. However, it may persist. Any dilatation of this remnant is thought to form the TDC [6]. It is often seen between the thyroid gland the hyoid bone in (61%), suprahyoid (24%), suprasternal (13%) and at the intralingual (2%) areas. A small number (<1%) undergoes neoplastic changes [7]. Malignant TDC are more prevalent in females (1.5:1) [8] and are often found in adults (mean age of 39 years). While benign TDC are usually found in children (mean age of 5.5 years); both can be found at any age [8]. In most malignant TDC patients, presentation is similar to the benign TDC. Page et al reported that pain, hoarseness, sudden growth of a mass, weight loss, regional lymphadenopathy and respiratory involvement suggest malignant changes [9]. In our four cases only a progressive mass was noted.

The use of radiological aids to assess the TDC is controversial. Samara et al reported 12 cases of TDC in which the most common radiological finding in Computed Tomography scan (CT Scan) was solid nodule in the cyst [7]. Two of the cases reported in Samara’s study had no evidence of malignancy on CT scan. Thyroid scanning is not of value in diagnosing TDC malignancy unless there is an obvious thyroid involvement [7]. The only form of imaging used in our patients was ultrasound.

Fine needle aspiration cytology (FNAC) was used in the initial investigation in some of cases in the literature, where the same criteria for diagnosing papillary carcinoma of the thyroid apply for TDC malignancy [10]. However, caution is advised since while interpreting a FNAC from a TDC in half of the reported cases cytology smears were inconclusive and may give false negative and false positive results [10]. Sending the TDC intra operatively for frozen section routinely to screen for malignancies is not cost effective. Malignancy remains a very rare finding in the TDC that comes with good prognosis if managed properly. This is why in most cases sending the TDC for permanent histopathology is the only thing that is required. Frozen section may play a role if malignancy was suspected before hand and the patient was consented for the SP, thyroidectomy and central nodal dissection with the TDC excision procedure.

The presence of papillary carcinoma in TDC should raise the suspicion of a primary origin. The tumor may arise from the thyroid tissue found in the TDC wall or it maybe the presentation of a metastatic thyroid cancer [11]. Papillary thyroid cancer has been shown to metastasize without a detectable lesion in the thyroid. Further more, up to 40% of reported TDC malignancy had a second focus in the thyroid hence thyroidectomy and long term follow up is advised [12],[13]. These numbers are comparable to what we present. Two of the patients (50%) had papillary carcinoma involving the thyroid gland.

Although 7% of TDC cancer patients have regional lymph node metastasis, the patients involved in our paper did not have any lymph node involvement or invasion to the cyst wall.

The criteria of diagnosing a primary carcinoma of the TDC include localization of the carcinoma to the TDC and tract, with absence of the malignancy in a resected thyroid gland [14]. This was the case in 2 of our patients (50%).

Due to its rarity, the management of malignant TDC is not fully defined and requires further understanding of the embryology and pathophysiology of the disease. It is still debatable as to when the thyroid is to be removed. All four patients went through SP and total thyroidecto-my with central nodal dissection after the TDC malignancy was diagnosed. Ramirez Plaza et al, suggested an Algorithm treatment for patients with TDC malignancy [15]. His protocol for thyroidectomy depends on the presence of a clinically detectable thyroid abnormality prior to surgery. In contrast to his suggestion, this paper is leaning towards a more aggressive approach to the thyroid gland. Two of the patients (50%) had involvement of the thyroid in which one of these two (25%) did not have any detectable thyroid involvement preoperatively.

Papillary carcinoma often carry good prognosis with rare instances of metastasis therefore it can be successfully managed surgically and with good follow up. The use of I131 ablation therapy and TSH suppression therapy depends on the presence of thyroid involvement.


  Conclusion Top


Malignancy in TDC is a rare finding but should not be over looked. There is yet a reliable method to diagnose malignant TDC preoperatively. Ruling out extensions of the disease, especially thyroid involvement remains the key of management. Malignant TDC patients usually have a favorable outcome if managed and followed up properly by experienced head and neck surgery, endocrinology andoOncology Team.



 
  References Top

1.
Mondin V, Ferlito A, Muzzi E, et al. Thyroglossal duct cyst: personal experience and literature review. Auris Nasus Larynx. 2008;35:11-25.  Back to cited text no. 1
    
2.
Ellis PD, van Nostrand AW. The applied anatomy of thyroglossal tract remnants. Laryngoscope. 1977;87:765-70.  Back to cited text no. 2
    
3.
Burnell I, Tomkinson A, Hourihan M, et al. Mucin-secreting papillary adenocarcinoma of the hyoid bone: a unique case.J Laryngol Otol. 2005;119:498-502.  Back to cited text no. 3
    
4.
Heshmati HM, Fatourechi V, van Heerden JA, et al. Thyroglossal duct carcinoma: report of 12 cases. Mayo Clin Proc. 1997;72:315-9.  Back to cited text no. 4
    
5.
Motamed M, McGlashan JA. Thyroglossal duct carcinoma. Curr Opin Otolaryngol Head Neck Surg. 2004;12:106-9.  Back to cited text no. 5
    
6.
O’Connell M, Grixti M, Harmer C. Thyroglossal duct carcinoma: presentation and management, including eight cases reports. Clin Oncol (R Coll Radiol). 1998;10:186-90.  Back to cited text no. 6
    
7.
Samara C, Bechrakis I, Kavadias S, et al. Thyroglossal duct cyst carcinoma: case report and review of the literature, with emphasis on CT findings. Neuroradiol. 2001;43:647-9.  Back to cited text no. 7
    
8.
Allard RH. The thyroglossal cyst. Head Neck Surg 1982;5:134-46.  Back to cited text no. 8
    
9.
Page CP, Kemmerer WT, Haff RC, et al. Thyroid carcinomas arising in thyroglossal ducts. Ann Surg. 1974;180:799-803.  Back to cited text no. 9
    
10.
Doshi SV, Cruz RM, Hilsinger RL, Jr. Thyroglossal duct carcinoma: a large case series. Ann Otol Rhinol Laryngol. 2001;110:734-8.  Back to cited text no. 10
    
11.
Wigley TL, Chonkich GD, Wat BY. Papillary carcinoma arising in a thyroglossal duct cyst. Otolaryngol Head Neck Surg. 1997;116:386-8.  Back to cited text no. 11
    
12.
Pacheco-Ojeda L, Micheau C, Stafford N, et al. Papillary carcinoma in thyroglossal duct remnants. Eur Arch Otorhinolaryngol. 1991;248:268-70.  Back to cited text no. 12
    
13.
Brown PM, Judd ES. Thyroglossal duct cysts and sinuses: results of radical (Sistrunk) operation. Am J Surg. 1961;102:494-501.  Back to cited text no. 13
    
14.
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. 14
    
15.
Plaza CP, Lopez ME, Carrasco CE, et al. 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. 15
    


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