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Year : 2005  |  Volume : 15  |  Issue : 4  |  Page : 531-533
"Papillary carcinoma in a thyroglossal duct cyst" - a case report and review of literature


Government Medical College Nagpur, India

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Keywords: Thyroglossal Cyst, Papillary Carcinoma, Computed Tomography

How to cite this article:
Taori K, Rohatgi S, Mahore D M, Dubey J, Saini T. "Papillary carcinoma in a thyroglossal duct cyst" - a case report and review of literature. Indian J Radiol Imaging 2005;15:531-3

How to cite this URL:
Taori K, Rohatgi S, Mahore D M, Dubey J, Saini T. "Papillary carcinoma in a thyroglossal duct cyst" - a case report and review of literature. Indian J Radiol Imaging [serial online] 2005 [cited 2019 Oct 15];15:531-3. Available from: http://www.ijri.org/text.asp?2005/15/4/531/28790

   Introduction Top


Thyroglossal duct cysts are the most common nonodontogenic cysts that occur in the neck. They account for approximately 70% of congenital neck abnormalities. The coexistence of a carcinoma in a thyroglossal duct cyst is rare. It occurs in less than 1% of patients and is almost invariably of thyroid gland origin. In majority of the patients, the histological features of papillary carcinoma of the thyroid tissue are demonstrated. Relevant review of literature reveals only 200 such cases. We report one such case suspected on spiral CT and confirmed on histopathology.


   Case report Top


A 25 years old female presented with complaints of a painless midline swelling over the anterior aspect of neck, which had been growing slowly in size over last 3 months [Figure - 1]. On examination the swelling moved with deglutition and protrusion of the tongue. Thyroid function tests were within normal limits. CT was done to know the origin and extent of the lesion. CT Neck showed a midline multicystic lesion with an attenuation value of 20-70 with foci of irregular calcification, anterosuperior to the isthmus of the thyroid gland and to the right of hyoid bone in its upper part [Figure - 2],[Figure - 3],[Figure - 4]. The lesion was intimately associated with the strap muscles. The thyroid gland was normal and no neck lymph nodes were found. A diagnosis of Thyroglossal Duct Cyst with possible malignant transformation was kept. Patient underwent excision by Sistrunk's operation. There were no local signs of invasion of the tissue surrounding the cyst at surgery. Gross examination of specimen showed, thyroglossal cyst partially filled by a solid brownish tissue [Figure - 5]. Histological sections showed a papillary carcinoma in the thyroid tissue of the thyroglossal cyst, with normal thyroid tissue at the boundary of carcinoma. There was no capsular invasion and the margins were negative. The patient has been followed up for two years with no further evidence of the disease.


   Discussion Top


Thyroglossal cysts are common abnormalities of thyroid development that present as palpable and symptomatic neck masses. These cysts, which originate from epithelial remnants of a persisting thyroglossal duct, may occur twice as often as branchial cleft abnormalities. In general Thyroglossal cyst (TGC) occur more frequently in younger patients [1]. Papillary carcinoma arising in a thyroglossal duct cyst is seen most frequently in young women with a sex ratio of 1.5:1. [2]. These can be located anywhere along the course of the thyroglossal duct, the majority are located below the level of the hyoid bone [3]. The clinical presentation of thyroglossal duct carcinoma may be indistinguishable from that of benign thyroglossal duct cysts however Thyroglossal duct carcinoma may present with a rapidly enlarging neck mass [4].

The first case of carcinoma in a thyroglossal duct cyst was reported by Uchermannin 1915 [2]. In 75% - 80% of patients, the histological features of papillary carcinoma of thyroid are demonstrated [3]. Other types although rarer include follicular variant of papillary carcinoma and pure follicular carcinoma. Squamous and anaplastic carcinomas have also been described [1]. There are different theories regarding the origin for these malignancies. In the sixties some authors thought that these carcinomas were metastases of thyroid carcinomas. Now, following demonstration of normal thyroid tissue occurrence in the wall of thyroglossal duct cysts, it is almost universally accepted that a carcinoma may arise from thyroglossal remnants [5].

Joseph and Komorowski proposed a criteria for an unequivocal diagnosis of thyroglossal duct cyst carcinoma which included: the presence of carcinoma in the duct or cyst, combined with squamous epithelium lining and normal thyroid follicles nests in the duct or cyst wall and the presence of a normal thyroid gland [1].

The detection of these carcinomas usually occurs as an incidental finding at the time of surgery. On CT, a benign thyroglossal duct cyst appears as a midline, fluid-attenuated mass near the level of the hyoid bone, with a thin, smooth wall. Infrahyoid thyroglossal duct cysts may be slightly off midline and are intimately associated with the strap muscles [4]. Calcifications are present in 60% of papillary carcinomas arising in the thyroid gland [4]. These calcifications correspond to the psammoma bodies that are visible histologically. To the best of our knowledge, there are no reports in the English-language literature of calcifications in a benign thyroglossal duct cyst.

A high attenuation value of 20-70 was found in our case. The high-density areas were not found to correspond with a high protein cystic component but with a solid thyroid papillary carcinoma coexisting within the low-density thyroglossal duct cyst [4]. Other entities may have CT findings similar to thyroglossal duct carcinoma. A congenital dermoid tumor may appear as a cystic midline mass, A dermoid tumor can be differentiated from a thyroglossal duct cyst if fat is detected within the lesion, and unlike a thyroglossal duct remnant, a dermoid tumor usually is not intimately associated with the strap muscles [4].

The prognosis for papillary thyroglossal duct cyst carcinoma is excellent, with occurrence of metastatic lesions in less than 2% of cases [6].

So, we would like to conclude that Carcinoma should be considered in thyroglossal duct cysts that have irregular calcification and high attenuation values. This case is reported in view of its extreme rarity.

 
   References Top

1.Fernandez JF, Ordoρez NG, Schultz PN, Samaan NA, Hickey RC. Thyroglossal duct carcinoma. Surgery 1991; 110:928-35.  Back to cited text no. 1    
2.Kristensen S, Juul A, Morsener J. Thyroglossal cyst carcinoma. J Laryngol Otol 1984; 98:1277-80.  Back to cited text no. 2    
3.Reede DL, Bergeron RT, Som PM. CT of thyroglossal duct cysts. Radiology 1985; 157:121-125.  Back to cited text no. 3  [PUBMED]  
4.Barton F. Branstetter, Jane L. Weissman, Thomas L. Kennedy and Mark Whitaker. The CT Appearance of Thyroglossal Duct Carcinoma. American Journal of Neuroradiology (2000) 21:1547-1550.  Back to cited text no. 4    
5.Sorrenti G, Cavazzuti PP, Zanetti G.Papillary carcinoma arising in thyroglossal duct cyst: a case report and review of the literature. Acta Otorhinolaryngol Ital. 1995 Dec; 15(6): 460-4.  Back to cited text no. 5    
6.Martins AS, Melo GM, Tincani AJ, Lage HT, Matos PS.Papillary carcinoma in a thyroglossal duct: case report Sao Paulo Med J. 1999 Nov 4; 117(6): 248-50.  Back to cited text no. 6    

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Correspondence Address:
K Taori
Radio-Diagnosis, Professor And Head; Dept. Of Radio-Diagnosis, Govt. Medical College, Nagpur (M.S.). 440003
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0971-3026.28790

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    Figures

[Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5]

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