Skip to main content
NCBI home page
Journal of Brown Hospital Medicine logoLink to Journal of Brown Hospital Medicine
. 2024 Jan 1;3(1):91542. doi: 10.56305/001c.91542

Dual Ectopic Thyroid: A Case Report and Review of the Literature

Alina Fernández-Genao 1, Dahiana Collado 2, Noemí Acevedo 1, Pamela Collado 3, José Arturo Restituyo Rosario 4, Cleiry García 5
PMCID: PMC11864399  PMID: 40027387

Abstract

Ectopic thyroid tissue is an uncommon anomaly, and the presence of dual ectopic thyroid tissues in a single patient is exceedingly rare. We describe a 69-year-old woman who presented with dyspeptic symptoms, asthenia, aphonia, and chronic fatigue. The patient was eventually found to have a multinodular goiter, cervical goiter, and mediastinal goiter. The patient underwent radical thyroidectomy and dissection, followed by treatment with levothyroxine. This report underscores the diagnostic challenges in complex presentations of ectopic thyroid tissue.

Keywords: endocrinology, thyroid diseases, thyroid dysgenesis, thyroid gland, ectopic thyroid

BACKGROUND

During embryonic development, the thyroid gland descends along the thyroglossal duct from the blind foramen to its anatomical position in the anterior portion of the trachea.1 Occasionally, errors occur during gland migration, resulting in what is known as ectopic thyroid tissue.2 A disruption at the beginning of migration results in thyroid tissue at the base of the tongue, the most common location (90%).1,3 Similarly, incomplete migration results in high cervical thyroid tissue, while excessive migration may result in mediastinal (<1%)4 or pericardiac1 thyroid tissue. Other locations include the palatine tonsils, axilla, descending aorta, gastrointestinal system, adrenal gland, and reproductive system.2 We present a woman in the Dominican Republic with dual ectopic thyroid that was incidentally discovered.

CASE PRESENTATION

A 69-year-old woman with a known history of hypertension, type 2 diabetes, and untreated thyroid goiter diagnosed seven years ago was evaluated by the gastroenterology department due to a history of dyspeptic symptoms, asthenia, aphonia, and chronic fatigue. The patient was referred to the internal medicine department for pre-anesthesia evaluation before undergoing an upper gastrointestinal endoscopy. Physical examination showed a symmetrical, cylindrical neck with a mass with irregular borders in the anterior region. Routine laboratory tests and chest radiographs were indicated for pre-procedural evaluation. The chest radiograph showed tracheal deviation to the right. Therefore, other imaging studies were conducted. Ultrasonography findings revealed a multinodular thyroid goiter with cystic degenerations inside. The right lobe was 1.7 cm x 1.2 cm x 1.0 cm with a volume of 1.14 mL and a left lobe of 3.6 cm x 3.5 cm x 2.7 cm with a volume of 18.8 mL. Computed tomography (CT) scan of the neck and chest showed an intrathoracic thyroid goiter with associated calcifications, which caused the trachea and esophagus to be displaced to the right.

Laboratory work revealed a TSH level of 0.350 mlU/L (range: 0.3-4.2 mlU/L) and free T4 of 20.47 pmol/L (range: 12-22 pmol/L). Endocrinology referral for a biopsy of the mass was initially considered. However, due to the intrathoracic location of the mass and the presenting complaints, referral to the surgery department was prioritized. The patient underwent radical thyroidectomy and dissection (Figure 1) with subsequent biopsy of the mass, which yielded the following diagnoses: 1) benign follicular hyperplasia (multinodular goiter), 2) ectopic thyroid tissue with associated benign follicular hyperplasia (cervical goiter) (Figure 2A), 3) ectopic thyroid tissue with focal adenomatous changes within a context of benign follicular hyperplasia (mediastinal goiter) (Figure 2B). During follow-up, the patient’s dyspeptic symptoms had resolved, and the thyroid profile showed a TSH level of 6.90 mlU/L (range: 0.3-4.2 mlU/L). The patient was started on levothyroxine.

Figure 1. 1) Product of total thyroidectomy. The right lobe measured 4.5 cm x 3 cm x 2 cm, the isthmus 2 cm x 0.7 cm, and the left lobe 6 cm x 3.5 cm x 3 cm. 2) Retrosternal irregular grayish-yellowish soft tissue formation measuring 7 cm x 5 cm x 2.5 cm. 3) Soft tissue in the left lateral neck measuring 2.5 cm x 2 cm x 1 cm.

Figure 1.

Open in a new tab

Figure 2A. Cervical tissue (in the left lateral neck) histology.

Figure 2A.

Open in a new tab

Thyroid tissue in which the proliferated follicular structures are observed, formed by cells lacking atypia and colloid content

Figure 2B. Retrosternal soft tissue histology.

Figure 2B.

Open in a new tab

Thyroid tissue in which follicular structures of variable size are appreciated, some conglomerate, others dilated, with eosinophilic content of colloid appearance, lined by cubic cells without atypia. In some areas, there is fibrous and colloid degeneration.

DISCUSSION

Thyroid ectopia cases occur in 1 per 100,000 to 300,000 persons in the general population, with a female-to-male ratio of 3-4:13. Likewise, the prevalence within the population with thyroid disease is 1 per 4,000 to 8,000 persons.3 Although in most cases (47%) it is asymptomatic, it can present with dysphagia, dysphonia, hypernasality, cough, foreign body sensation, hemoptysis, and stridor, among others.2,5 Treatment is dependent on the patient’s presentation. Asymptomatic euthyroid patients do not usually require treatment and are kept under observation. For hypothyroid patients, treatment is with thyroid hormone replacement.3 Surgical removal of the gland is the preferred approach in cases of hemorrhage, ulceration, obstructive symptoms, uncontrolled hyperthyroidism, and malignant neoplasia.2 In most cases, the diagnosis of malignancy is reached after excision and biopsy of the mass. While rarely documented, there exists a less than 1% estimated risk of malignant transformation from ectopic thyroid tissue, with papillary carcinoma being the most prevalent subtype.2,5 Other complications associated with thyroid ectopia include benign neoplasia, thyroiditis, and hypothyroidism.2

Based on a literature review of 25 cases,6–31 31 patients were identified to have ectopic thyroid tissue. Of the 31 patients, 22 (70.97%) were female, and 21 (67.76%) were 18 years or younger, with a median age of 14, ranging from 4 to 75 years. While most cases are typically identified through neonatal screening, it is noteworthy that in the Dominican Republic, thyroid studies are not routinely done as part of neonatal screening protocols due to their unavailability unless specifically requested by parents, particularly in private healthcare settings. It is essential to consider that ectopic thyroid tissue may undergo enlargement during periods of stress, potentially leading to symptomatic presentations later in life, as observed in this case.2

Regarding the location of the ectopic tissue, 26 (83.87%) of the patients had ectopia at the base of the tongue/lingual area. Other common locations included the sub-hyoid area (n=15, 48.39%), supra-hyoid (n=6, 19.35%), and the area anterior to the thyroid notch, submental region, and pre-hyoid with 9.68% each. The least frequent locations were the liver, the pre-tracheal area, the submandibular area, and the right paramedian line, as well as more unspecific locations, such as the floor of the mouth and neck, present in one patient each. During thyroid examination, the most common finding was swelling of the anterior line of the neck (n=23, 74.19%), followed by absent thyroid tissue in its usual location on palpation (n=4, 12.9%), lateral neck mass (n=1, 3.23%), whereas three (9.68%) of patients had a regular thyroid exam. Among the associated symptoms, five (16.13%) patients reported dysphagia, three (9.68%) odynophagia, and two (3.23%) reported hoarseness, cough, and dysphonia. Other symptoms listed included foreign body sensation, pain, and unspecific symptoms of hypo/hyperthyroidism. Regarding the chemical evaluation of thyroid function, TSH was normal in eight (25.81%) and elevated in twenty-one (67.74%) patients. Among the cases that reported fine needle aspiration (n=12, 38.71%), the findings included follicular cells (n=7, 58.33%) and colloid (n=3, 25%).

In conclusion, the presented case of dual ectopic thyroid tissue showcases this anomaly’s remarkable rarity and diagnostic challenges. Recognizing the multifaceted nature of ectopic thyroid presentations is crucial for accurate diagnosis and appropriate management, especially in countries with limited or unavailable newborn screening. In such settings, heightened awareness, and expertise in recognizing the different presentations become imperative for healthcare practitioners to navigate the challenges posed by limited screening resources, ultimately contributing to enhanced healthcare delivery, and improved overall public health. This case adds to the limited literature, emphasizing the need for thorough investigation in complex clinical presentations.

Author Contributions

All authors have reviewed the final manuscript prior to submission. All the authors have contributed significantly to the manuscript, per the International Committee of Medical Journal Editors authorship criteria.

  • Substantial contributions to the conception or design of the work or the acquisition, analysis, or interpretation of data for the work; AND

  • Drafting the work or revising it critically for important intellectual content; AND

  • Final approval of the version to be published; AND

  • Agreement to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Disclosures

The authors have no conflicts of interest to disclose.

Corresponding Author

Noemí Acevedo

510 W 45th, Manhattan, NY, 10036

Phone: +1 (829) 876 – 8197

Email: n.acevedo@ce.pucmm.edu.do

References

  1. Ectopic thyroid tissue. Agrawal Y. Jan 17;2019 [2023-9-25];Cancer Therapy Advisor. Accessed September 25, 2023.
  2. F A S. Ectopic thyroid. [2023-9-25].
  3. Ectopic thyroid tissue. [2023-9-25]. https://www.pathologyoutlines.com/topic/thyroidheterotopic.html
  4. Niknejad Mohammadtaghi. Radiopaedia.org. Vol. 6. Radiopaedia.org; Ectopic thyroid: Radiology reference article.https://doi.org/10.53347/rid-163832 [DOI] [Google Scholar]
  5. Noussios George, Anagnostis Panagiotis, Goulis Dimitrios G, Lappas Dimitrios, Natsis Konstantinos. European Journal of Endocrinology. 3. Vol. 165. Oxford University Press (OUP); Ectopic thyroid tissue: Anatomical, clinical, and surgical implications of a rare entity; pp. 375–382.https://doi.org/10.1530/eje-11-0461 [DOI] [PubMed] [Google Scholar]
  6. Dual Ectopic Thyroid Gland in an Elderly Male: Double Trouble? Agrawal Rachana, Varty Sangeeta. 2019Indian Journal of Otolaryngology and Head & Neck Surgery. 71(S1):79–81. doi: 10.1007/s12070-017-1082-9. https://doi.org/10.1007/s12070-017-1082-9 [DOI] [PMC free article] [PubMed] [Google Scholar]
  7. Dual thyroid ectopia. Al-Akeely M.H. 2003Saudi Med J. 24(9):1021–1023. [PubMed] [Google Scholar]
  8. Baik S. H., Choi J. H., Lee H.-M. European Archives of Oto-Rhino-Laryngology. 2. Vol. 259. Springer Science and Business Media LLC; Dual ectopic thyroid; pp. 105–107.https://doi.org/10.1007/s00405-001-0419-9 [DOI] [PubMed] [Google Scholar]
  9. Harisankar ChidambaramNatrajan Balasubramanian. Indian Journal of Nuclear Medicine. 1. Vol. 28. Medknow; Dual ectopic thyroid in the presence of atrophic orthotopic thyroid gland in a patient with acquired hypothyroidism: Evaluation with hybrid Single-Photon Emission Computed Tomography/Computed Tomography; p. 26.https://doi.org/10.4103/0972-3919.116808 [DOI] [PMC free article] [PubMed] [Google Scholar]
  10. Dual ectopic thyroid. Basu S., Nair N. 2006Indian Pediatr. 43(8):741. [PubMed] [Google Scholar]
  11. Dual ectopic thyroid. Bayat M.R., Vawda F., Campbell H. Jul;2005 Clinical Radiology. 60(7):821–825. doi: 10.1016/j.crad.2005.02.016. https://doi.org/10.1016/j.crad.2005.02.016 [DOI] [PubMed] [Google Scholar]
  12. Dual ectopic thyroid: case series and review of the literature. Chawla Madhavi, Kumar Rakesh, Malhotra Arun. Jan;2007 Clin Nucl Med. 32(1):1–5. doi: 10.1097/01.rlu.0000249590.70176.58. https://doi.org/10.1097/01.rlu.0000249590.70176.58 [DOI] [PubMed] [Google Scholar]
  13. Dual ectopic thyroid with normally located thyroid: a case report. Kumar Choudhury Bipul, Kaimal Saikia Uma, Sarma Dipti, Saikia Mihir, Dutta Choudhury Sarojini, Barua Santanu, Dewri Swapna. 2011Journal of Thyroid Research. 2011(159703):1–4. doi: 10.4061/2011/159703. https://doi.org/10.4061/2011/159703 [DOI] [PMC free article] [PubMed] [Google Scholar]
  14. Dual ectopic thyroid: A rare entity. Dual ectopic thyroid tissue. Dabas Aashima, Khadgawat Rajesh, Bal Chander S. Jun;2015 Journal of Paediatrics and Child Health. 51(6):647–648. doi: 10.1111/jpc.12593. https://doi.org/10.1111/jpc.12593 [DOI] [PubMed] [Google Scholar]
  15. Ghanem Nadir, Bley Thorsten, Altehoefer Carsten, Högerle Stefan, Langer Mathias. Thyroid. 5. Vol. 13. Mary Ann Liebert Inc; Ectopic thyroid gland in the porta hepatis and lingua; pp. 503–507.https://doi.org/10.1089/105072503322021188 [DOI] [PubMed] [Google Scholar]
  16. Hazarika Produl, Siddiqui Shahab Ahmed, Pujary Kailesh, Shah Parul, Nayak Deepak Ranjan, Balakrishnan R. The Journal of Laryngology & Otology. 4. Vol. 112. Cambridge University Press (CUP); Dual ectopic thyroid: a report of two cases; pp. 393–395.https://doi.org/10.1017/s0022215100140563 [DOI] [PubMed] [Google Scholar]
  17. Dual thyroid ectopia with a normally located pretracheal thyroid gland: case report and literature review. Huang Ting-Shuo, Chen Huang-Yang. Mar 14;2007 Head & Neck. 29(9):885–888. doi: 10.1002/hed.20604. https://doi.org/10.1002/hed.20604 [DOI] [PubMed] [Google Scholar]
  18. Dual Ectopy of Thyroid Tissue: Intratracheal and in Nasopharynx, Associated with Normally Located Thyroid. Hussain D., Waheed S., Haider G.., et al. 2015J Coll Physicians Surg Pak. 25(Suppl 2):S86–S88. doi: 10.2015/JCPSP.S86S88. https://doi.org/10.2015/JCPSP.S86S88 [DOI] [PubMed] [Google Scholar]
  19. Jha Pankaj S, Rote-Kaginalkar Varsha, Titare Prashant, Jadhav Manoj B. Indian Journal of Radiology and Imaging. 01. Vol. 28. Georg Thieme Verlag KG; Hemiagenesis of thyroid with dual thyroid ectopia: A rare case report; pp. 14–17.https://doi.org/10.4103/ijri.ijri_292_17 [DOI] [PMC free article] [PubMed] [Google Scholar]
  20. A case of dual ectopy thyroid along the thyroglossal tract demonstrated on 99mTc-Pertechnatate hybrid single photon emission computed tomography/computed tomography. Tripathi Madhavi, Agarwal KrishanKant, Karunanithi Sellam, Jain Sachin. 2014Indian Journal of Nuclear Medicine. 29(2):105–107. doi: 10.4103/0972-3919.130300. https://doi.org/10.4103/0972-3919.130300 [DOI] [PMC free article] [PubMed] [Google Scholar]
  21. A Case of Subclinical Hypothyroidism with Lingual and Right Pretracheal Ectopic Thyroid. Kim Min Sun, Kong Young Hwa, Lee Dae-Yeol. Jun 5;2015 Journal of Clinical Research in Pediatric Endocrinology. 7(2):148–150. doi: 10.4274/jcrpe.1791. https://doi.org/10.4274/jcrpe.1791 [DOI] [PMC free article] [PubMed] [Google Scholar]
  22. Kisakol G., Gonen S., Kaya A., Dikbas O., Sari O., Kiresi D., Gungor K., Karakurt F. Journal of Endocrinological Investigation. 9. Vol. 27. Springer Science and Business Media LLC; Dual ectopic thyroid gland with Graves’ disease and unilateral ophthalmopathy: A case report and review of the literature; pp. 874–877.https://doi.org/10.1007/bf03346284 [DOI] [PubMed] [Google Scholar]
  23. Dual ectopic thyroid gland. Matta-Coelho C, Donato S, Carvalho M, et al. Oct 2;2018 BMJ Case Rep. 2018:bcr2018225506. doi: 10.1136/bcr-2018-225506. [DOI] [PMC free article] [PubMed] [Google Scholar]
  24. Dual Ectopic Thyroid: A Case Report. Minichil F., Nour A.S. 2022Ethiop J Health Sci. 32(Spec Iss 1):77–79. doi: 10.4314/ejhs.v32i1.12S. https://doi.org/10.4314/ejhs.v32i1.12S [DOI] [PMC free article] [PubMed] [Google Scholar]
  25. Dual-site thyroid ectopy in a mother and son. Misaki TAKASHI, Koh TOSHIKIYO, Shimbo SHIN'ICHIRO, Kasagi KANJI, Konishi JUNJI. Jan;1992 Thyroid. 2(4):325–327. doi: 10.1089/thy.1992.2.325. https://doi.org/10.1089/thy.1992.2.325 [DOI] [PubMed] [Google Scholar]
  26. Incidental Finding of Dual Ectopic Thyroid on Computed Tomography Angiography. Pierro Antonio, Cilla Savino, Modugno Pietro, Sallustio Giuseppina. Aug 3;2017 J Clin Imaging Sci. 7(30):30. doi: 10.4103/jcis.jcis_21_17. https://doi.org/10.4103/jcis.jcis_21_17 [DOI] [PMC free article] [PubMed] [Google Scholar]
  27. Dual ectopic thyroid with lingual and anterior hyoid ectopic thyroid tissue in a Chinese girl. Dong R., Li K., Liu G.., et al. 2012Saudi Med J. 33(10):1125–1127. [PubMed] [Google Scholar]
  28. Dual ectopic thyroid presenting with an anterior neck mass. Seok Hannah, Oh Su-Jin, Ha Won-Chul, Son Hyun Shik, Sohn Tae-Seo. 2015The Korean Journal of Internal Medicine. 30(1):129–130. doi: 10.3904/kjim.2015.30.1.129. https://doi.org/10.3904/kjim.2015.30.1.129 [DOI] [PMC free article] [PubMed] [Google Scholar]
  29. Dual ectopic thyroid: a case report with review of literature. Sood A., Seam R.K., Gupta M.., et al. 2011Iran J Radiol. 8(1):29–32. [PMC free article] [PubMed] [Google Scholar]
  30. Dual thyroid ectopia-role of thyroid scintigraphy and neck ultrasonography. Sood Ashwani, Jain Tarun Kumar, Meena Ram Singh, Bhatia Anmol, Bhattacharya Anish, Mittal Bhagwant Rai. 2015Indian Journal of Nuclear Medicine. 30(4):338. doi: 10.4103/0972-3919.164023. https://doi.org/10.4103/0972-3919.164023 [DOI] [PMC free article] [PubMed] [Google Scholar]
  31. Ulug Tuncay, Ulubil S. Arif, Alagol Faruk. The Journal of Laryngology & Otology. 7. Vol. 117. Cambridge University Press (CUP); Dual ectopic thyroid: report of a case; pp. 574–576.https://doi.org/10.1258/002221503322113076 [DOI] [PubMed] [Google Scholar]

Articles from Journal of Brown Hospital Medicine are provided here courtesy of Department of Medicine of Alpert Medical School of Brown University

RESOURCES