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Published in: BMC Endocrine Disorders 1/2015

Open Access 01-12-2015 | Research article

New insight into ectopic thyroid glands between the neck and maxillofacial region from a 42-case study

Authors: Ting Gu, Boren Jiang, Ningjian Wang, Fangzhen Xia, Lizhen Wang, Aichun Gu, Feng Xu, Yongshun Han, Qin Li, Yingli Lu

Published in: BMC Endocrine Disorders | Issue 1/2015

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Abstract

Background

Ectopic thyroid is a rare disease. In the present study at the 9th People’s Hospital in Shanghai, China, 42 patients’ ectopic thyroid glands between the neck and maxillofacial region were subjected to a retrospective and transverse study based on data from 1978 to 2012 to explore the natural characteristics of ectopic thyroid.

Methods

The patients’ clinical data were collected. In addition, scintigraphy (Tc-99 m, Iodine-131), CT scan, histology and pathology were performed. The protein expression of thyroid transcription factor-1 (TTF-1), thyroglobulin (TG), calcitonin (CT), Ki-67 and parathyroid hormone (PTH) were analyzed from paraffin wax-stored specimens of ectopic thyroid tissue compared with those of orthotopic thyroid tissue.

Results

There were 42 total ectopic thyroid patients, approximately 1.24 patients per year on average at our hospital. These patients were aged from 6 to 85 years old, and there were 35 females (83.3 %), seven males (16.7 %). In total, 27 of the patients had lingual thyroid (64 %); seven, sublingual thyroid (17 %); five, dual areas occupied by ectopic thyroid (12 %) and three, other types (7 %). The following conditions were also presented: nodular goiter (13 %), adenoma (8.7 %) and Hashimoto’s thyroiditis (4.3 %), no malignancy and no accompanying ectopic parathyroid. TTF-1 expression was significantly higher in ectopic samples than that in orthotopic samples (P = 0.007), but CT and Ki-67 levels displayed no difference. PTH was negative in ectopic tissue.

Conclusion

Ectopic thyroid is a rare disease and females were more prone to the disease. The most frequent location was lingual thyroid. Nodular goiter, adenoma and Hashimoto’s thyroiditis was observed as orthotopic thyroid without accompanying ectopic parathyroid. TTF-1 was highly expressed in ectopic tissue, which may be related to abnormal embryogenesis leading to the thyroid gland being in an abnormal position. The expression of calcitonin (CT) and Ki-67 was not increased, and there were no malignant cells in any sample, which could indicate that it is not easy for ectopic thyroids to become malignant between the neck and maxillofacial region.
Literature
1.
go back to reference Ibrahim NA, Fadeyibi IO. Ectopic thyroid: etiology, pathology and management. Hormones (Athens). 2011;10(4):261–9.CrossRef Ibrahim NA, Fadeyibi IO. Ectopic thyroid: etiology, pathology and management. Hormones (Athens). 2011;10(4):261–9.CrossRef
2.
go back to reference 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):375–82.CrossRefPubMed 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):375–82.CrossRefPubMed
3.
go back to reference Yoon JS, Won KC, Cho IH, Lee JT, Lee HW. Clinical characteristics of ectopic thyroid in Korea. Thyroid. 2007;17:1117–21.CrossRefPubMed Yoon JS, Won KC, Cho IH, Lee JT, Lee HW. Clinical characteristics of ectopic thyroid in Korea. Thyroid. 2007;17:1117–21.CrossRefPubMed
4.
go back to reference Bhojwani KM, Hegde MC, Alva A, Vishwas KV. Papillary carcinoma in a lingual thyroid: an unusual presentation. Ear Nose Throat J. 2012;91:289–91.PubMed Bhojwani KM, Hegde MC, Alva A, Vishwas KV. Papillary carcinoma in a lingual thyroid: an unusual presentation. Ear Nose Throat J. 2012;91:289–91.PubMed
5.
go back to reference Guerrissi JO. Follicular variant of papillary carcinoma in submandibular ectopic thyroid with no orthotopicthyroid gland. J Craniofac Surg. 2012;23:138–9.CrossRefPubMed Guerrissi JO. Follicular variant of papillary carcinoma in submandibular ectopic thyroid with no orthotopicthyroid gland. J Craniofac Surg. 2012;23:138–9.CrossRefPubMed
6.
go back to reference Berghmans T, Paesmans M, Mascaux C, Martin B. Thyroid transcription factor 1--a new prognostic factor in lung cancer: a meta-analysis. Ann Oncol. 2006;17(11):1673–6.CrossRefPubMed Berghmans T, Paesmans M, Mascaux C, Martin B. Thyroid transcription factor 1--a new prognostic factor in lung cancer: a meta-analysis. Ann Oncol. 2006;17(11):1673–6.CrossRefPubMed
7.
go back to reference Matoso A, Singh K, Jacob R, Greaves WO. Comparison of thyroid transcription factor-1 expression by 2 monoclonal antibodies in pulmonary and nonpulmonary primary tumors. Appl Immunohistochem Mol Morphol. 2010;18(2):142–9.PubMedCentralCrossRefPubMed Matoso A, Singh K, Jacob R, Greaves WO. Comparison of thyroid transcription factor-1 expression by 2 monoclonal antibodies in pulmonary and nonpulmonary primary tumors. Appl Immunohistochem Mol Morphol. 2010;18(2):142–9.PubMedCentralCrossRefPubMed
8.
go back to reference Pardo V, Vono-Toniolo J, Rubio IG, Knobel M. The p.A2215D thyroglobulin gene mutation leads to deficient synthesis and secretion of the mutated protein and congenital hypothyroidism with wide phenotype variation. J Clin Endocrinol Metab. 2009;94(8):2938–44.CrossRefPubMed Pardo V, Vono-Toniolo J, Rubio IG, Knobel M. The p.A2215D thyroglobulin gene mutation leads to deficient synthesis and secretion of the mutated protein and congenital hypothyroidism with wide phenotype variation. J Clin Endocrinol Metab. 2009;94(8):2938–44.CrossRefPubMed
9.
go back to reference Targovnik HM, Citterio CE, Rivolta CM. Thyroglobulin gene mutations in congenital hypothyroidism. Horm Res Paediatr. 2011;75(5):311–21.CrossRefPubMed Targovnik HM, Citterio CE, Rivolta CM. Thyroglobulin gene mutations in congenital hypothyroidism. Horm Res Paediatr. 2011;75(5):311–21.CrossRefPubMed
10.
go back to reference Pichu S, Krishnamoorthy S, Shishkov A, Zhang B, McCue P, Ponnappa BC. Knockdown of Ki-67 by dicer-substrate small interfering RNA sensitizes bladder cancer cells to curcumin-induced tumor inhibition. PLoS One. 2012;7(11):e48567.PubMedCentralCrossRefPubMed Pichu S, Krishnamoorthy S, Shishkov A, Zhang B, McCue P, Ponnappa BC. Knockdown of Ki-67 by dicer-substrate small interfering RNA sensitizes bladder cancer cells to curcumin-induced tumor inhibition. PLoS One. 2012;7(11):e48567.PubMedCentralCrossRefPubMed
11.
go back to reference Cowen D, Troncoso P, Khoo VS, Zagars GK, von Eschenbach AC, Meistrich ML, et al. Ki-67 staining is an independent correlate of biochemical failure in prostate cancer treated with radiotherapy. Clin Cancer Res. 2002;8(5):1148–54.PubMed Cowen D, Troncoso P, Khoo VS, Zagars GK, von Eschenbach AC, Meistrich ML, et al. Ki-67 staining is an independent correlate of biochemical failure in prostate cancer treated with radiotherapy. Clin Cancer Res. 2002;8(5):1148–54.PubMed
13.
go back to reference Papi G, Rossi G, Corsello SM, Corrado S. Nodular disease and parafollicular C-cell distribution: results from a prospective and retrospective clinico-pathological study on the thyroid isthmus. Eur J Endocrinol. 2010;162(1):137–43.CrossRefPubMed Papi G, Rossi G, Corsello SM, Corrado S. Nodular disease and parafollicular C-cell distribution: results from a prospective and retrospective clinico-pathological study on the thyroid isthmus. Eur J Endocrinol. 2010;162(1):137–43.CrossRefPubMed
14.
go back to reference Kalan A, Tariq M. Lingual thyroid gland: clinical evaluation and comprehensive management. Ear Nose Throat J. 1999;78:340–1. 345–349.PubMed Kalan A, Tariq M. Lingual thyroid gland: clinical evaluation and comprehensive management. Ear Nose Throat J. 1999;78:340–1. 345–349.PubMed
15.
go back to reference Gopal RA, Acharya SV, Bandgar T, Menon PS, Marfatia H, Shah NS. Clinical profile of ectopic thyroid in Asian Indians: a single-center experience. Endocr Pract. 2009;15:322–5.CrossRefPubMed Gopal RA, Acharya SV, Bandgar T, Menon PS, Marfatia H, Shah NS. Clinical profile of ectopic thyroid in Asian Indians: a single-center experience. Endocr Pract. 2009;15:322–5.CrossRefPubMed
16.
go back to reference Felice MD, Lauro RD. Thyroid development and its disorders: genetic and molecular mechanisms. Endocr Rev. 2004;25:722–46.CrossRefPubMed Felice MD, Lauro RD. Thyroid development and its disorders: genetic and molecular mechanisms. Endocr Rev. 2004;25:722–46.CrossRefPubMed
17.
go back to reference Gillam MP, Kopp P. Genetic regulation of thyroid development. Curr Opin Pediatr. 2001;13(4):358–63.CrossRefPubMed Gillam MP, Kopp P. Genetic regulation of thyroid development. Curr Opin Pediatr. 2001;13(4):358–63.CrossRefPubMed
18.
go back to reference Grasberger H, Ringkananont U, Lefrancois P, Abramowicz M, Vassart G, Refetoff S. Thyroid transcription factor 1 rescues PAX8/p300 synergism impaired by a natural PAX8 paired domain mutation with dominant negative activity. Mol Endocrinol. 2005;19(7):1779–91.CrossRefPubMed Grasberger H, Ringkananont U, Lefrancois P, Abramowicz M, Vassart G, Refetoff S. Thyroid transcription factor 1 rescues PAX8/p300 synergism impaired by a natural PAX8 paired domain mutation with dominant negative activity. Mol Endocrinol. 2005;19(7):1779–91.CrossRefPubMed
19.
go back to reference Amendola E, De Luca P, Macchia PE, Terracciano D, Rosica A, Chiappetta G, et al. A mouse model demonstrates a multigenic origin of congenital hypothyroidism. Endocrinology. 2005;146(12):5038–47.CrossRefPubMed Amendola E, De Luca P, Macchia PE, Terracciano D, Rosica A, Chiappetta G, et al. A mouse model demonstrates a multigenic origin of congenital hypothyroidism. Endocrinology. 2005;146(12):5038–47.CrossRefPubMed
20.
go back to reference Sturniolo G, Violi MA, Galletti B, Baldari S, Campenni A, Vermiqlio F, et al. Differentiated thyroid carcinoma in lingual thyroid. Endocrine. 2015. [Epub ahead of print]. Sturniolo G, Violi MA, Galletti B, Baldari S, Campenni A, Vermiqlio F, et al. Differentiated thyroid carcinoma in lingual thyroid. Endocrine. 2015. [Epub ahead of print].
21.
go back to reference Liu Z, Farley A, Chen L, Kirby BJ, Kovacs CS, Blackburn CC, et al. Thymus-associated parathyroid hormone has two cellular origins with distinct endocrine and immunological functions. PLoS Genet. 2010;6(12):e1001251.PubMedCentralCrossRefPubMed Liu Z, Farley A, Chen L, Kirby BJ, Kovacs CS, Blackburn CC, et al. Thymus-associated parathyroid hormone has two cellular origins with distinct endocrine and immunological functions. PLoS Genet. 2010;6(12):e1001251.PubMedCentralCrossRefPubMed
Metadata
Title
New insight into ectopic thyroid glands between the neck and maxillofacial region from a 42-case study
Authors
Ting Gu
Boren Jiang
Ningjian Wang
Fangzhen Xia
Lizhen Wang
Aichun Gu
Feng Xu
Yongshun Han
Qin Li
Yingli Lu
Publication date
01-12-2015
Publisher
BioMed Central
Published in
BMC Endocrine Disorders / Issue 1/2015
Electronic ISSN: 1472-6823
DOI
https://doi.org/10.1186/s12902-015-0066-6

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