Eur J Cardiothorac Surg 2002;22:842-844
© 2002 Elsevier Science NL
Papillary carcinoma arising on cervico-mediastinal thyroglossal ductal cyst resected via transcervical and partial upper sternotomy incision
S. Volkan Baysungur,
Erdal Okur*,
Semih Halezeroglu,
Ali Atasalihi
SSK Sureyyapasa Thoracic and Cardiovascular Diseases Teaching Hospital, Istanbul, Turkey
Received 11 May 2002;
received in revised form 24 July 2002;
accepted 30 July 2002.
* Corresponding author. Kardelen 4.1, D 34 Atasehir, Istanbul 81120, Turkey. Tel.: +90-216-455-3538; fax: +90-212-225-9934
e-mail: erdalokur{at}turk.net
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Abstract
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Thyroglossal cyst is the most common congenital cervical pathology of childhood. Malign transformation in thyroglossal cyst is very rare and seen generally in adults. Here, we report on a 40-year-old female patient who presented with progressive dyspnea and enlarging cervical masses. Radiological examination revealed multiple cystic lesions in cervical and mediastinal region. The cysts were resected surgically via transcervical and partial upper sternotomy incision. Pathological examination revealed malignant change in the cervico-mediastinal thyroglossal cysts.
Key Words: Thyroglossal duct cyst • Papillary carcinoma • Surgical resection
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1. Introduction
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Thyroglossal ductal cysts are the most common congenital cervical masses seen in childhood [1]. They develop due to a failure in complete involution of the thyroglossal duct during embryonal life. They are rarely seen in adults [2]. Malignant transformation of the cyst is quite rare (about 0.7–1%) and is encountered mostly in adults [1,3]. Here, we present a case of papillary carcinoma arising on the thyroglossal ductal cyst which has an unusual cervico-mediastinal location.
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2. Case report
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A 40-year-old female patient was admitted with right cervical multiple cystic masses, first noticed 4 months previuosly. The cysts were enlarging over time. For the last 2 weeks, she had also symptoms of dyspnea when she was in a supine position. She had a history of subtotal thyroidectomy 4 years earlier for hyperthyroidism. The pathologic result of the resected thyroid gland was a diffuse thyroid hyperplasia with no malignancy within the specimen. Chest X-ray of the patient revealed a large, spherical and sharp-edged homogeneous density in a right paratracheal location. Computed tomography (CT) showed six cystic lesions between 0.2 and 2 cm in size in the right cervical region and a 7-cm cyst, which was partially calcified and located in anterior mediastinal compartment. Magnetic resonance imaging (MRI) was performed in order to demonstrate the relation of the lesion with mediastinal structures, which revealed a giant mediastinal cyst with no invasion to mediastinal structures (Fig. 1A)
. There was no abnormality in routine laboratory examinations. There was no pathologic cell in cytological examination of aspirated cyst fluid. Our most probable preoperative diagnosis was ‘cervico-mediastinal cystic hygroma’. The patient was operated on for diagnostic and therapeutic purposes. She was placed in a supine position. For better exploration of cervical and mediastinal regions altogether in the same session, a right cervical incision anterior to the sternocleidomastoid muscle extending through jugular notch down to mid-sternum with partial sternotomy was done (Fig. 1B). After six cervical cysts were extirpated, the mediastinal cyst was aspirated and removed (Fig. 2A)
. The postoperative pathologic result was reported as ‘papillary thyroid carcinoma arising on thyroglossal ductal cyst’ (Fig. 2B). Thyroid scintigraphy was performed postoperatively and revealed no pathology in her thyroid gland. Prophylactic radioactive iodine treatment was done. The patient is still in follow-up and no pathology was detected in her MR investigation at the third postoperative month.
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Fig. 1. (A) MRI of patient showing cervical and mediastinal cyst. (B) Exploration of cervical and mediastinal cysts via transcervical and partial sternotomy incision is shown.
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Fig. 2. (A) Cysts resected are seen (mediastinal cyst was aspirated before resection). (B) Pathologic section stained with hemotoxylin–eosin showing carcinoma areas (ca) and normal thyroid follicle (TF) x20.
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3. Discussion
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Thyroglossal ductal cyst is the most common congenital cervical pathology. Development of malignancy in thyroglossal ductal cyst is very rare (0.7–1%) and only few cases have been reported [1,3–6]. Thyroglossal cysts are generally located in the midline, beneath the hyoid bone. Cyst walls are covered by columnar, cuboidal and non-keratinized squamous epithelium and thyroid tissue can be found in 65% of the cases. Carcinoma arising from these thyroid islets is mostly papillary carcinoma (75–80%), while other cell types such as folliculo-papillar (8%), squamous (6%) and follicular (3%) types are rarely encountered [3,5,6].
Seventy percent of benign thyroglossal cysts are seen in first 3 decades. Malignant transformation within the cysts are most commonly observed in thin ladies in the fourth decade of life [3,4]. Squamous type is seen in patients over 50 years of age [4]. No predisposing factor has been described although some claim that radiation is a risk factor [4]. Clinically it is impossible to differentiate a benign cyst from a malignant one, but increased age and rapid growth should alert for malignancy [1]. However, it should be remembered that infected cysts may also grow rapidly.
Patients with clinical symptoms should be assessed by ultrasonography, CT and MRI. Non-palpable cysts can be detected by ultrasonography. Calcification and nodules at the cyst wall are seen in malignant form [1,4] and can be visualized by CT scanning. Calcification at the cyst wall is especially specific for papillary carcinoma. While some authors report that cytological examination is diagnostic in up to 40% of the cases, in most cases, as in ours, cytology is negative. In papillary carcinoma, thyroglobulin level in the cyst fluid is diagnostically high. Similarly, T3 level is found to be high in follicular carcinoma [7]. In most cases, definitive diagnosis can only be reached by pathologic examination of the resected cyst.
When the thyroglossal cyst is beneath the hyoid bone and in the midline, as in most of the cases, the sistrunk procedure is the resection technique of choice [8]. However, we needed a different approach in order to reach both cervical and mediastinal compartments at the same time in our patient. When the malignancy is encountered in any cyst, total or subtotal thyroidectomy should be performed in addition to the surgical excision [1,9]. This is necessary because in 11–40% of the cases, there is also a malignancy in the thyroid gland [2,4,10]. Since metastasis to cervical lymph nodes is seen in 7.9–10% of the cases [4,10] and there may be an aberrant cervical thyroid tissue, all patients should be screened by cervical scintigraphy and should undergo radioactive iodine therapy [10], as in our case. Long-term prognosis is excellent after this treatment and the recurrence rate is almost 0% in cases with papillary carcinoma [2,9]. Heshmati et al. reported no local recurrence or distant metastasis in his series of 12 cases after a mean follow-up of 13 years [3], but the prognosis is poor in squamous cell carcinoma [2].
In conclusion, differential diagnosis of a cervico-mediastinal cyst must include the thyroglossal cyst. When rapidly growing cervical or mediastinal masses are found in a middle-aged woman together with increased thyroglobulin or T3 level in aspirated cyst fluid and when there is calcification or nodule in the cyst wall seen on CT, one should suspect carcinoma arising on the thyroglossal ductal cyst.
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References
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- Barton F.B., Jane L.W., Thomas L.K. The CT appearance of thyroglossal duct carcinoma. Am J Neuroradiol 2000;21:1547-1550.[Abstract/Free Full Text]
- Martins A.S., Melo G.M., Tincani A.J., Lage H.T., Matos P.S. Papillary carcinoma in a thyroglossal duct: case report. Sao Paulo Med J 1999;117:248-250.[Medline]
- Heshmati H.M., Fatourechi V., van Heerden J.A., Hay I.D., Goelner J.R. Thyroglossal duct carcinoma: report of 12 cases. Mayo Clin Proc 1997;72:315-319.[Abstract]
- Christine M.G., Christian D., Jeffrey R.H. The CT and MR imaging features of carcinoma arising in thyroglossal duct remnants. Am J Neuroradiol 2000;21:770-774.[Abstract/Free Full Text]
- Hilger A.W., Thomson S.D., Smallman L.A., Watkinson J.C. Papillary carcinoma arising in a thyroglossal duct cyst: a case report and literature review. J Laryngol Otol 1995;109:1124-1127.[Medline]
- Weiss S.D., Orlich C.C. Primary papillary carcinoma of a thyroglossal duct cyst: report of a case and literature review. Br J Surg 1991;78:87-89.[Medline]
- Kawamura S., Kishino B., Miyauchi A., Takai S., Tajima K., Mashita K., Tarui S. The differential diagnosis of cystic neck masses by the determination of thyroglobulin concentrations in the aspirates. Clin Endocrinol (Oxf) 1984;20:261-267.[Medline]
- Sistrunk W.E. The surgical treatment of cyst of the thyroglossal tract. Ann Surg 1920;71:121-122.[Medline]
- Walton B.R., Koch K.E. Presentation and management of a thyroglossal duct cyst with a papillary carcinoma. South Med J 1997;90:758-761.[Medline]
- Miccoli P., Pacini F., Basolo S., Iacconi P., Puccini M., Pinchera A. Thyroid carcinoma in a thyroglossal duct cyst: tumor resection alone or a total thyroidectomy?. Ann Chir 1998;52:452-454.[Medline]
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Abstract
1. Introduction
