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Japanese Journal of Clinical Oncology Pages 340-342

Thyroglossal Duct Carcinoma: a Case Report
Introduction
Case Report
Discussion
References

Thyroglossal Duct Carcinoma: a Case Report

Thyroglossal Duct Carcinoma: a Case Report Takahiro Asakage, Satoshi Nara, Takashi Yoshizumi and Satoshi Ebihara

Department of Head and Neck Surgery, National Cancer Center Hospital East, Kashiwa, Chiba, Japan

We describe a 47-year-old woman with a 13-year history of asymptomatic midline submental swelling. Cytologic examination of a fine-needle aspiration specimen from the solid mass revealed adenocarcinoma. The preoperative diagnosis was thyroglossal duct carcinoma. A Sistrunk procedure was performed and microscopic examination revealed papillary adenocarcinoma. The postoperative course was uneventful and there were no signs of local recurrence or metastasis at one year after surgery.

Key words: thyroglossal duct carcinoma - papillary carcinoma - Sistrunk procedure

INTRODUCTION

Although thyroglossal duct remnants are common developmental abnormalities, carcinomas arising from them are rare and about 100 such cases have been reported in the English language literature (1 ). However, only 21 cases of thyroglossal duct carcinoma have been reported in the Japanese literature (2 ). Here we report an additional case.


Figure 1. Cervical X-ray demonstrates a calcified lesion in front of the hyoid bone (white arrowheads, the thyroglossal duct carcinoma; black arrows, hyoid bone).

CASE REPORT

A 47-year-old woman presented with a 13-year history of asymptomatic submental midline swelling. Because the swelling had recently become larger, she sought medical advice. On physical examination, a mass measuring approximately 3.3 * 5.0 * 2.8 cm was palpated at the level of the hyoid bone. The mass was well circumscribed, firm and slightly fluctuant. No cervical lymphadenopathy was evident. The thyroid gland was normally located and thyroid hormone levels were normal. A cervical X-ray examination revealed calcification anterior to the hyoid bone (Fig. 1 ) and ultrasonography revealed several cystic lesions measuring 1.0-1.5 cm. A solid component was present anterior to the hyoid bone. Small cystic thyroid lesions were present, two in the left lobe and one in the right. Computed tomography also showed cystic lesions with calcification anterior to the hyoid bone (Fig. 2 ). Cytologic examination of a fine-needle aspiration specimen from the solid mass revealed adenocarcinoma. A preoperative diagnosis of thyroglossal duct carcinoma and adenomatous goiter was made.

A Sistrunk procedure was performed. The resected specimen measured 3.5 * 5.0 * 3.5 cm and was attached to a segment of the hyoid bone (Fig. 3 ). On cross-sectional examination, the cyst was a yellow, multilobular, solid lesion with calcification. Microscopic examination revealed papillary adenocarcinoma with a follicular pattern (Fig. 4 ), but without a fibrous capsule. Although the cancer showed invasion through the cyst wall, it was completely resected. Normal thyroid gland cells were evident around the malignant component. The postoperative course was uneventful and follow-up examination at one year after surgery revealed no local recurrence or metastasis.


Figure 2. Computed tomography demonstrates some cystic lesions measuring about 1.0 or 1.5 cm in diameter. There is a solid component in front of the hyoid bone (small arrowheads, thyroglossal duct carcinoma; large arrowheads, hyoid bone).


Figure 3. Gross specimen measuring 3.5 * 5.0 * 3.5 cm with a segment of the hyoid bone attached (white arrowheads, thyroglossal duct carcinoma; black arrows, hyoid bone).


Figure 4. (A) Microscopic examination shows papillary adenocarcinoma with a follicular pattern. (B) High-power view reveals papillary infoldings of nuclei, a ground-glass appearance and nuclear grooves.

DISCUSSION

Developmental abnormalities of the thyroid gland may be divided into two types: ectopic thyroid gland and thyroglossal duct cysts. Ectopic thyroids result from incomplete descent of the gland. Thyroglossal duct cysts result from persistence of a portion of the embryonic duct (3 ).

Most thyroglossal duct cysts are located in the midline (4 ). Among the various cyst locations, 2.1% are intralingual, 24.1% suprahyoidal, 60.9% thyroidal and 12.9% suprasternal (5 ). Between 24% and 62% of thyroglossal duct specimens contain normal thyroid tissue (6 ). Thyroglossal duct carcinomas originate in the thyroid remnant of the wall of the cyst (7 ).

Thyroglossal duct carcinoma is a relatively rare lesion (8 ), with a reported frequency of 1.8% (7 cases among 371 thyroglossal ducts or cysts) according to LiVolsi (6 ) and 1.7% (2 cases among 116 cysts) according to Keeling (9 ). Van Vuuren (1 ) reported a female:male ratio of 1.6:1. Fernandez (10 ) 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. LiVolsi (6 ) reported that slowly growing lesions may continue to enlarge over several weeks to several years. The size of the mass is variable, but most are in the 2-4 cm range (11 ). Generally, the findings of thyroid function tests and thyroid scans are within normal limits (10 ). Computed tomography or ultrasonography shows a cyst and peripheral calcification anterior to the hyoid bone (10 ).

The Sistrunk procedure (12 ), involving excision of the mid-portion of the hyoid bone, is the most common treatment for thyroglossal duct carcinoma (13 ,14 ). Kristensen et al. (15 ) have suggested that the Sistrunk procedure is appropriate if 1, histologically normal thyroid follicles are found in the cyst wall; 2, the tumor has not extended through the cyst wall; 3, the thyroid gland is normal; and 4, no lymph node involvement is evident. If lymphadenopathy is found during surgery, conservative neck dissection should be considered.

Histologically, most thyroglossal duct carcinomas are papillary carcinoma (79.9%). Other histologic types are mixed follicular and papillary carcinoma (9.5%), squamous cell carcinoma (7.6%), follicular carcinoma (0.6%), anaplastic carcinoma (0.6%) and Hürthle cell carcinoma (0.6%) (1 ). The thyroid gland is normal on pathological examination (16 ). Generally, the prognosis of papillary carcinoma arising from a median ectopic thyroid is extremely good. In many reported cases, there was no evidence of recurrent tumor following local excision (6 ).

References

1. Van Vuuren PAC, Balm AJM, Greger RT, Hilgers FJM, Loftus BM, Delprat CC, et al. Carcinoma arising in thyroglossal remnants. Clin Otolaryngol 1994;19:509/P-15.

2. Taketani S, Yoshikawa K, Hashimoto T, Yamaguchi T, Dousei T, Moriguchi A, et al. A case report of ectopic thyroid cancer originated from the thyroglossal duct remnant. Nippon Rinsho Geka Igakkai Zassi 1995;56:948-52 (in Japanese).

3. Hawkins DB, Jacoobson BE, Klatt EC. Cysts of the thyroglossal duct. Laryngoscope 1982;92:1254>/PG>-8. MEDLINE Abstract

4. Brintnall ES, Davis J, Huffman WC, Lierle DM. Thyroglossal ducts and cysts. Arch Otolaryngol 1954;59:282-9.

5. Allard RHB. The thyroglossal cyst. Head Neck 1982;5:134-46.

6. LiVolsi VA, Perzin KH, Savetsky L. Carcinoma arising in median ectopic thyroid. Cancer 1974;34:1303-15. MEDLINE Abstract

7. Nuttal FQ. Cystic metastasis from papillary adenocarcinoma of the thyroid with comment concerning carcinoma associated with thyroglossal remnants. Am J Surg 1965;109:500-5.

8. Roses DF, Snively SL, Phelps RG, Cohen N, Blum M. Carcinoma of the thyroglossal duct. Am J Surg 1983;145:266-9. MEDLINE Abstract

9. Keeling JH, Ochsner A. Carcinoma in thyroglossal duct remnants. Cancer 1959;12:596-600.

10. Fernandez JF, Ordonez NG, Schultz PN, Samaan NA, Hickey RC. Thyroglossal duct carcinoma. Surgery 1991;110:928-35.

11. Trail ML, Zeringue GP, Chicola JP. Carcinoma in thyroglossal duct remnants. Laryngoscope 1997;87:1685-91.

12. Sistrunk WE. The surgical treatment of cyst of the thyroglossal tract. Ann Surg 1920;71::121-2.

13. 13. Pacheco OL, Micheau C, Stufford N, Marandas P, Luboinski B, Martinez AL. Papillary carcinoma in thyroglossal duct remnants. Eur Arch Otorhinolaryn-gol 1991;248:268-70. MEDLINE Abstract

14. Yildiz K, Koksal H, Ozoran Y, Muhtar H, Teltar M. Papillary carcinoma in thyroglossal duct remnant with normal thyroid gland. J Laryngol Otol 1993;107:1174-6.

15. Kristensen S, Juula A, Moesner J. Thyroglossal cyst carcinoma. J Laryngol Otol 1984:98:1277-80.

16. Weiss SD, Orich CC. Primary papillary carcinoma of a thyroglossal duct cyst; report of a case and literature review. Br J Surg 1991;78:87-9. MEDLINE Abstract

Received December 27, 1996; accepted May 9, 1997
For reprints and all correspondence: Takahiro Asakage, Department of Head and Neck Surgery, National Cancer Center Hospital East, 5-1, Kashiwanoha 6-chome, Kashiwa, Chiba 277, Japan

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Copyright© Japanese Journal of Clinical Oncology, 1997.
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This Article
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