Case Report


A challenging entity for endocrine surgeons: Ectopic lingual thyroid gland. A case-based update

,  ,  ,  ,  ,  

1 MD, UOC Multifunctional Center of Endocrine Surgery, Cristo Re General Hospital, Rome, Italy

2 MD, Department of General Surgery, University of Rome “Tor Vergata”, Rome, Italy

3 MD, Division of Otorinolaringology, Cristo Re General Hospital, Rome, Italy

4 MD, UOC of Radiology, Section of Nuclear Medicine, Cristo Re General Hospital, Rome, Italy

5 UOC of Radiology, Cristo Re General Hospital, Rome, Italy

6 MD, Unicamillus International Medical University, Rome, Italy

Address correspondence to:

Pietro Princi

Chief, Multifunctional Center of Endocrine Surgery, Cristo Re General Hospital, via delle Calasanziane 25, 00168 Rome,

Italy

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Article ID: 100151Z12ST2025

doi: 10.5348/100151Z12ST2025CR

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How to cite this article

Tempera SE, Nigro C, D’Ottavi LR, Ieria FP, Perrone L, Princi P. A challenging entity for endocrine surgeons: Ectopic lingual thyroid gland. A case-based update. J Case Rep Images Surg 2025;11(1):13–18.

ABSTRACT


Introduction: Lingual thyroid (LT) is a rare embryologic anomaly. Diagnosis is challenging and treatment is still controversial.

Case Report: We report a case of a 46 years old man with occasional sore throat, who underwent an otorhinolaryngology evaluation. Fiber optic flexible indirect laryngoscopy (FIL) highlighted a mass at the base of the tongue. Subsequent surgeon-performed neck ultrasound, computed tomography (CT) scan and thyroid scintiscan confirmed the diagnosis of LT. The patient had never investigated the thyroid before as he was totally asymptomatic. Subsequent blood tests revealed subclinical hypothyroidism. Fine needle aspiration (FNAC) showed benign cytology. After a multidisciplinary discussion, the patient was treated with suppressive doses of thyroxine due to subclinical hypothyroidism. At the follow-up the patient showed a size reduction of the ectopic gland at ultrasound and resolution of the initial symptoms.

Conclusion: A comprehensive pertinent literature review was performed, showing few small series of patients affected by LT. Treatment options proposed for LT include thyroid hormone suppression therapy, radioactive iodine (RAI) ablation, and surgical excision. However, a consensus in management does still not exist so far. Most authors agree that no treatment is required when LT is asymptomatic and the patient is euthyroid. Thus, we proposed an update of the diagnostic and therapeutic algorithm for the management of LT based on the present cases. Further research regarding evaluation, monitoring and follow-up of ectopic thyroid tissue is necessary for better assessment of the workup and management of LT, in particular due to its possible but rare malignant nature.

Keywords: Lingual thyroid, Therapeutic algorithm, Thyroid ectopy, Thyroid surgery

SUPPORTING INFORMATION


Author Contributions

Serena Elisa Tempera - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Casimiro Nigro - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Luigi Raimondo D’Ottavi - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Francesco Pio Ieria - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Luca Perrone - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Pietro Princi - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Guarantor of Submission

The corresponding author is the guarantor of submission.

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Consent Statement

Written informed consent was obtained from the patient for publication of this article.

Data Availability

All relevant data are within the paper and its Supporting Information files.

Conflict of Interest

Authors declare no conflict of interest.

Copyright

© 2025 Serena Elisa Tempera et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.