Ectopic thyroid papillary carcinoma of nasopharynx associated with adenoid hypertrophy: an unusual presentation
© Tian et al.; licensee BioMed Central Ltd. 2014
Received: 25 June 2014
Accepted: 18 September 2014
Published: 20 September 2014
Ectopic thyroid tissue of nasopharynx is an uncommon phenomenon and papillary thyroid carcinoma arising from the tissue is extremely rare. The authors report a rare case of 16-year-old girl with papillary thyroid carcinoma of nasopharynx. Clinicians were ever confused by adenoid hypertrophy and solved the diagnostic dilemma by adequate examinations. In the case, we mainly emphasize that surgeons should be aware of and actively consider such a possibility of ectopic papillary thyroid carcinoma of nasopharynx in children and adolescents with long-term nasal obstruction, even if thyroid carcinoma is a rare tumor.
KeywordsEctopic thyroid papillary carcinoma Nasopharynx Adenoid hypertrophy Nasal obstruction
Ectopic thyroid tissue is rare, which the reported incidence is 1 in every 100,000 to 300,000 in the general population[1, 2]. A thyroid gland can reside anywhere along thyroid embryologic path from the foramen cecum to its normal site such as lingua, thyroglossal duct and laryngotrachea. The abnormal migration of the thyroid is known as ectopic thyroid. Ectopic thyroid tissue, especially ectopic papillary thyroid carcinoma (PTC) of nasopharynx, is extremely rare, and may cause diagnostic and therapic dilemma for clinicians just as our case. To our knowledge, very few reports of ectopic nasopharyngeal thyroid cancer with a normal eutopic thyroid gland have been published to date[5, 6]. Herein we present an uncommon case of ectopic PTC of nasopharynx associated with adenoidal hypertrophy and share our experience of the successful management about such a rare case.
Thyroid carcinoma is a relatively rare pediatric pathology, comprising around 3% of children and adolescents tumors. Papillary thyroid carcinoma, a kind of differentiated thyroid cancer, is the most common neoplasm in the thyroid gland and accounts for about 80% of all thyroid cancers. Ectopic papillary thyroid carcinoma has been found in some places such as lingua, mediastinum and thyroglossal duct. Ectopic PTC of nasopharynx is extremely scarce especially with normal eutopic thyroid gland and no lymph node involvement just as our case.
The girl of our case had long-term nasal obstruction and was diagnosed as adenoid hypertrophy before 5 years by means of simple CT scan examination. Because of increasing nasal obstruction, the patient was checked by further examinations including CT scan and endoscopy and an oval neoplasm with intact capsule was found to be located on nasopharynx near hypertrophic adenoid. This case suggested that nasal obstruction of children and adolescent could be caused by not only lymphoid tissue hyperplasia and common neoplasms but also unusual tumors. Then, biopsy showed histological characteristics of papillarity, ground glass nuclei and nuclear grooves of cells, suggesting this was a thyroid-like tumor of malignant origin. Additional examination of immunohistochemistry revealed that the positive expressions of TTF-1, CK and TG and negative for P63. TTF-1 is currently used in routine surgical pathology as an immunohischemical marker of primary carcinomas arising in thyroid and lung organs. It has also been reported to be expressed in other tumors such as thymoma, ovarian, endocervical and endometrial neoplasms[11, 12]. According to the histological characteristics and immunohistochemical features, we made a final diagnosis of papillary thyroid carcinoma on nasopharynx. Other thyroid-like tumors such as low-grade papillary adenocarcinoma of the nasopharynx (LGPACNP) also exhibit similar features[13, 14]. However, the fact that the positive expression of TG has hitherto not been mentioned in previous reports of LGPACNP could exclude this diagnostic possibility. In addition, normal thyroid gland in its pretracheal position and no cervical lymphadenopathy gave us evidence to support our diagnosis of ectopic PTC rather than metastasis of thyroid carcinoma.
Treatment outcomes of PTC are very favorable and have an excellent prognosis with 10-year survival rates of more than 90%[17, 18]. The optional treatment methods have surgery, radiotherapy, radioactive iodine therapy, and chemotherapy according to the current management guidelines for patients with differentiated thyroid cancer issued by the American Thyroid Association. Adequate surgical treatment followed by postoperative radioactive iodine therapy can offer very promising results for ectopic PTC.
In conclusion, even if thyroid carcinoma is a rare tumor in children and adolescents, surgeons should be aware of and actively consider such a possibility of ectopic papillary thyroid carcinoma of nasopharynx in patients with nasal obstruction. Since the clinical signs and symptoms are most often vague mimicking other nasopharyngeal diseases, ectopic thyroid carcinoma should be considered in the differential diagnosis even in the presence of a normal thyroid gland. Imageological examinations such as CT scan and biopsy are usually necessary for diagnosis and treatment. The present case report shows that there is always a chance to encounter exceedingly rare disease and adequate examinations of pre-operation are necessary for appropriate diagnosis and treatment.
Written informed consent was obtained from the patient’s next of kin for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal. The review board of Harbin Medical University approved publication of this case report.
- Sevinç AI, Unek T, Canda AE, Guray M, Kocdor MA, Saydam S, Harmancioglu O: Papillary carcinoma arising in subhyoid ectopic thyroid gland with no orthotopic thyroid tissue. Am J Surg. 2010, 200 (1): e17-e18. 10.1016/j.amjsurg.2009.06.032.View ArticlePubMedGoogle Scholar
- Noussios G, Anagnostis P, Goulis DG, Lappas D, Natsis K: Ectopic thyroid tissue: anatomical, clinical, and surgical implications of a rare entity. Eur J Endocrinol. 2011, 165 (3): 375-382. 10.1530/EJE-11-0461.View ArticlePubMedGoogle Scholar
- Massine RE, Durning SJ, Koroscil TM: Lingual thyroid carcinoma: a case report and review of the literature. Thyroid. 2001, 11 (12): 1191-1196. 10.1089/10507250152741055.View ArticlePubMedGoogle Scholar
- Hazarika P, Murty PS, Nooruddin SM, Zachariah J, Rao NR: Lingual thyroid. Ear Nose Throat J. 1988, 67 (3): 161-165.PubMedGoogle Scholar
- Tong A, Xia W, Qi F, Jin Z, Yang D, Zhang Z, Li F, Xing X, Lian X: Hyperthyroidism caused by an ectopic thyrotropin-secreting tumor of the nasopharynx: a case report and review of the literature. Thyroid. 2013, 23 (9): 1172-1177. 10.1089/thy.2012.0574.View ArticlePubMedGoogle Scholar
- Karras S, Anagnostis P, Noussios G, Pontikides N: Thyroid papillary carcinoma arising in ectopic thyroid tissue within a branchial cleft cyst. BMJ Case Rep. 2013, 22: 2013-Google Scholar
- Dzepina D: Surgical and pathological characteristics of papillary thyroid cancer in children and adolescents. Int J Pediatr. 2012, 2012: 125389-View ArticlePubMedGoogle Scholar
- Hodak SP, Burman KD: The calcitonin conundrum–is it time for routine measurement of serum calcitonin in patients with thyroid nodules?. J Clin Endocrinol Metab. 2004, 89 (2): 511-514. 10.1210/jc.2003-031983.View ArticlePubMedGoogle Scholar
- Choi JY, Kim JH: A case of an ectopic thyroid gland at the lateral neck masquerading as a metastatic papillary thyroid carcinoma. J Korean Med Sci. 2008, 23 (3): 548-550. 10.3346/jkms.2008.23.3.548.View ArticlePubMedPubMed CentralGoogle Scholar
- Lianos G, Bali C, Tatsis V, Anastasiadi Z, Lianou E, Papathanasiou V, Messinis T: Ectopic thyroid carcinoma. Case report. G Chir. 2013, 34 (4): 114-116.PubMedPubMed CentralGoogle Scholar
- Kubba LA, McCluggage WG, Liu J, Malpica A, Euscher ED, Silva EG, Deavers MT: Thyroid transcription factor-1 expression in ovarian epithelial neoplasms. Mod Pathol. 2008, 21: 485-490. 10.1038/modpathol.2008.4.View ArticlePubMedGoogle Scholar
- Siami K, McCluggage WG, Ordonez NG, Euscher ED, Malpica A, Sneige N, Silva EG, Deavers MT: Thyroid transcription factor-1 expression in endometrial and endocervical adenocarcinomas. Am J Surg Pathol. 2007, 31: 1759-1763. 10.1097/PAS.0b013e3181131e21.View ArticlePubMedGoogle Scholar
- Wenig BM, Hyams VJ, Heffner DK: Nasopharyngeal papillary adenocarcinoma. A clinicopathologic study of a low-grade carcinoma. Am J Surg Pathol. 1988, 12: 946-953. 10.1097/00000478-198812000-00005.View ArticlePubMedGoogle Scholar
- Petersson F, Pang B, Loke D, Hao L, Yan B: Biphasic low-grade nasopharyngeal papillary adenocarcinoma with a prominent spindle cell component: report of a case localized to the posterior nasal septum. Head Neck Pathol. 2011, 5 (3): 306-313. 10.1007/s12105-011-0252-4.View ArticlePubMedPubMed CentralGoogle Scholar
- Glicksman JT, Franklin JH, Shepherd J, Rotenberg BW: An endonasal approach to the resection of a papillary seromucinous adenocarcinoma of the Eustacian tube. J Otolaryngol Head Neck Surg. 2013, 42 (1): 12-10.1186/1916-0216-42-12.View ArticlePubMedPubMed CentralGoogle Scholar
- Haq M, Harmer C: Thyroid cancer: an overview [J]. Nucl Med Commun. 2004, 25 (9): 861-867. 10.1097/00006231-200409000-00001.View ArticlePubMedGoogle Scholar
- Volante M, Collini P, Nikiforov YE, Sakamoto A, Kakudo K, Katoh R, Lloyd RV, LiVolsi VA, Papotti M, Sobrinho-Simoes M, Bussolati G, Rosai J: Poorly differentiated thyroid carcinoma: the turin proposal for the use of uniform diagnostic criteria and an algorithmic diagnostic approach. Am J Surg Pathol. 2007, 31 (8): 1256-1264. 10.1097/PAS.0b013e3180309e6a.View ArticlePubMedGoogle Scholar
- Verburg FA, Mader U, Luster M, Reiners C: Primary tumour diameter as a risk factor for advanced disease features of differentiated thyroid carcinoma. Clin Endocrinol (Oxf). 2009, 71 (2): 291-297. 10.1111/j.1365-2265.2008.03482.x.View ArticleGoogle Scholar
- Cooper DS, Doherty GM, Haugen BR, Kloos RT, Lee SL, Mandel SJ, Mazzaferri EL, McIver B, Pacini F, Schlumberger M, Sherman SI, Steward DL, Tuttle RM, American Thyroid Association (ATA) Guidelines Taskforce on Thyroid Nodules and Differentiated Thyroid Cancer: Revised American Thyroid Association management guidelines for patients with thyroid nodules and differentiated thyroid cancer. Thyroid. 2009, 19 (11): 1167-1214. 10.1089/thy.2009.0110.View ArticlePubMedGoogle Scholar
This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.