Ectopic lingual thyroid with subclinical hypothyroidism in children.

OBJECTIVES: Lingual thyroid is a rare condition that affects approximately 1 in 100,000 individuals. Although it is usually detected in the pediatric population through newborn screening tests or evaluation of congenital hypothyroidism, there are cases in which it remains undetected until adulthood or until symptoms arise because of glandular enlargement. The possible symptoms of lingual thyroid include foreign body sensation in the throat, dysphagia, dyspnea, and hemorrhage. Several cases of lingual thyroid are asymptomatic and accompanied by subclinical hypothyroidism. Herein, we present three cases of lingual thyroid treated with thyroid hormone suppressive therapy. CASE PRESENTATION: The three patients sought medical attention because of a sore throat or foreign body sensation in the throat. Their newborn screening tests and developmental histories were normal. These patients exhibited subclinical hypothyroidism and were treated with hormone suppression therapy. CONCLUSIONS: Patients with lingual thyroid frequently exhibit subclinical hypothyroidism. Hormone treatment may help to reduce the size of the ectopic thyroid and improve symptoms. If an increase in size is noted during follow-up or symptoms do not improve, surgical treatments may be considered.

Lingual thyroid in a 16-year-old girl.

Lingual thyroid is a rare congenital disorder displaying ectopic thyroid tissue at the base of the tongue. This is the most common location for ectopic thyroid tissue and is usually the only thyroid tissue present. This is a case report of a 16-year-old female who presented with nasal congestion. Fiberoptic laryngoscopy showed swelling at the base of the tongue and an ultrasound examination of the neck was without visible thyroid tissue. A 99mTc-pertechnetate scintigraphy confirmed the clinical diagnosis. As the patient was euthyroid and without symptoms active surveillance was planned.

A surgical partial excision for the treatment of lingual ectopic thyroid: a case report.

The lingual ectopic thyroid is a sporadic case. Based on the medical records of Dr. Soetomo General Academic Hospital, Surabaya, they only found one case of ectopic thyroid at least in the last ten years. There is no consensus in the literature about the best therapeutic strategy in managing ectopic thyroid. A 20-year-old female patient with a diagnosis of ectopic lingual thyroid. She has complained of lumps at the base of her tongue since she was ten years old. She performed a partial excision of the tumor with a transoral approach. Partial excision of the lingual ectopic thyroid results in an airway free from obstruction, leaving the rest of the thyroid tissue functioning so that the patient does not require lifelong hormone treatment but has the potential for hypertrophy to recur. The transoral approach provides post-operative results that maintain aesthetic function and reduce morbidity and hospitalization. Partial excision of lingual ectopic thyroid gives good results.

Does surgical management still play a role in the management of ectopic lingual thyroid: Institutional experience and systematic review of the literature.

OBJECTIVE: Management of ectopic lingual thyroid (ELT) must balance the morbidity of disease with the morbidity of treatment. We investigate clinical outcomes associated with modern treatment options and analyze the role of transoral surgery in the treatment algorithm for ELT. METHODS: This was a retrospective chart review of ELT patients treated at a tertiary care center from 1/1/1979 to 12/31/2019. In addition, a systematic review of the literature from 1979 to 2021 for reports of ELT was performed. Symptoms defined as high-risk were dysphagia, dysphonia, dyspnea, neck swelling, bleeding, and obstructive sleep apnea (OSA). RESULTS: 36 patients within the institutional cohort (IC) and 224 cases in the systematic review (SRC) met criteria. The most common presenting symptoms for both cohorts were dysphagia, globus sensation, and dysphonia. One third of each cohort were hypothyroid, while 3% (n = 1) and 9% (n = 21) of the IC and SRC, respectively, had clinical suspicion of malignancy at presentation. 27% (n = 10) of the IC and 55% (n = 121) of the SRC underwent surgical therapy. There was a 4% (3/72) transoral bleed rate for all patients undergoing transoral surgery. Other reported complications were minor. There were no tracheostomies, and no deaths. Among observed, medically treated, and surgically treated patients, symptoms improved during follow up for 43% or 68% in the IC and SRC, respectively. Following surgery, symptoms improved or resolved for 86% or 93% in the IC and SRC, respectively. CONCLUSIONS: Asymptomatic ELTs with no concern for malignancy can be managed with observation. Patients with mild symptoms or hypothyroidism may trial thyroid suppressive therapy or RAI. RAI can be considered for patients with high-risk symptoms. For patients with symptoms resistant to conservative therapy, concern for malignancy or high-risk symptoms not conducive to RAI, surgery should be considered. Transoral approaches offer acceptable morbidity, and most patients experience resolution of symptoms following this approach.

Airway management in patients with lingual thyroid: a case report and review of the literature.

PURPOSE: To review the management of patients with lingual thyroid (LT) causing upper airway obstruction and to suggest a diagnostic and therapeutic workflow. METHODS: A PubMed review of published cases from January 1980 up to December 2020 of LT causing upper airway obstruction. We selected cases of confirmed LTs that presented with non-state-dependent airway obstruction. An illustrative case report is presented. RESULTS: Twenty-one articles fulfilling the inclusion criteria were found, reporting 24 cases (7 neonatal, 2 pediatric and 15 adults). The main presenting symptoms was dyspnea with increased work of breathing, followed by dysphagia and stridor most commonly in neonates. At least one imaging modality was performed in all patients. Thyroid function was altered in half the patients and normal in the other half. The LT was the only thyroid tissue in all cases except 2. Altogether, 5/24 patients required tracheostomies and two-thirds of the patients underwent surgical resection of the LT (mostly transoral). Also 2/3 of the patients received thyroid replacement therapy. After a median follow-up of 17 months, airway symptoms had fully resolved for all patients but one. CONCLUSION: While rare, ectopic LTs should be considered in the differential diagnosis of stridor, dyspnea and airway obstruction. In neonates, concomitant presence of hypothyroidism on neonatal screening and airway obstruction should prompt the search for a LT. Early identification and thyroid replacement therapy seem to significantly relieve symptoms of upper airway obstruction, but severe obstruction and concomitant airway lesions may require more definitive management approaches.

Is Transoral Robotic Surgery the Best Surgical Treatment for Lingual Thyroid? A Case-Report and Literature Review.

OBJECTIVES: To highlight the specific outcomes of the current surgical procedures for lingual thyroid excision, for benign and malignant lesions. METHODS: We carried out a systematic review of surgical treatments of lingual thyroid, according to the PRISMA method. We conducted our literature search in PubMed and Ovid. Data was collected concerning patient demographics, tumor characteristics, types of surgery performed, and specific intra- and postoperative outcomes of each procedure. Surgical procedures were classified in 4 categories: transcervical approaches, "invasive" transoral approaches (transmandibular and/or tongue splitting), "non-invasive" transoral approaches, and transoral robotic surgery. We detailed the transoral robotic surgical technique through a case report, along with a surgical video. RESULTS: Of 373 peer-reviewed articles found, 40 provided adequate information on surgical management and outcomes for patients with lingual thyroid. "Non-invasive" transoral approaches and transoral robotic surgeries required significantly fewer tracheostomies than "invasive" transoral and transcervical approaches (P < .001), while there was no statistical difference in the rate of surgical complications between each procedure. CONCLUSIONS: Transoral robotic surgery appears to be a feasible, effective, and fast solution for lingual thyroid excision, with excellent short- and long-term surgical outcomes.

Tiroides Ectópica Lingual: caso Clínico / Lingual Ectopic Thyroid: a clinical case

La tiroides ectópica lingual es una patología muy poco frecuente, producida por la detención en el descenso normal de la glándula durante el desarrollo embrio-nario. La localización lingual de tejido tiroideo es la más común entre las tiroides ectópicas o aberrantes. Esta enfermedad puede ser asintomática pero, cuan-do los signos y síntomas están presentes, guardan estrecha correlación con la localización de la lesión y son proporcionales a su tamaño. El diagnóstico debe realizarse clínicamente y con el complemento de es-tudios por imágenes y endocrinológicos. En los aná-lisis de laboratorio se debe incluir dosaje de las hor-monas TSH, T4 libre y T3, vinculadas con la función tiroidea. Las biopsias deben evitarse ya que causan desequilibrio en la producción hormonal de la glándu-la y peligro de profusas hemorragias. En este artículo se desarrolla una descripción de las generalidades de la tiroides ectópica lingual, y se presenta un caso clínico de un niño con un tumor lingual, que fue deri-vado por su médica pediatra a cirugía para realizar una biopsia. Asimismo, se comenta la importancia que tiene para el odontólogo conocer esta patología a fin de poder evitar sus posibles complicaciones (AU)

Lingual thyroid is a rare disorder produced by a failure in the descent of thyroid gland to its normal position during embryological development. Lingual localization of thyroid tissue is the most common among the ectopic or aberrant thyroids. This condition can be asymptomatic, although when symptoms take place, they are connected to the lesion location and depend on its size. Diagnosis should be made clinically and complemented with imaging and endocrine studies. Laboratory analysis must include dosage of TSH, free T4 and T3, thyroid function-linked hormones. Due to the possible imbalance in the gland hormone production and the risk of massive bleeding, biopsy should be avoided. In this article, a brief description of lingual ectopic thyroid generalities is developed and a clinical case of a 7-years old child is provided. Additionally, dentistry importance of knowing this condition is commented, in order to prevent its possible complications (AU)

Lingual thyroid with severe hypothyroidism: A case report.

INTRODUCTION: Ectopic thyroid tissue presenting at the base of the tongue, called lingual thyroid, is a clinical rarity. Clinical presentation varies depending upon either the severity of regional symptoms associated with the enlargement of gland size, or the features related to thyroid dysfunction. PATIENT CONCERNS: We reported a case of a 29-year-old female who presented with symptoms of easy fatigue and depression for 3 months. DIAGNOSIS: After a series of diagnostic workup, the lingual thyroid with severe hypothyroidism was diagnosed. INTERVENTION AND OUTCOME: She received conservative treatment with thyroid hormone replacement and the symptoms improved significantly. LESSONS: Lingual thyroid is a rare entity that needs careful diagnostic workup including clinical examination, biochemical tests, imaging methods such as ultrasonography, scintigraphy, computed tomography, magnetic resonance imaging, and fine-needle aspiration cytology to plan the management. Lingual thyroid with hypothyroidism and no neck regional symptoms can be conservatively treated and requires regular follow-up for the prevention of potential risk of malignant transformation.

Transoral robotic resection of a lingual thyroid: a novel treatment for obstructive sleep apnoea.

A 34-year-old woman with a history of congenital hypothyroidism and 15 years of obstructive sleep apnoea was admitted with a left submandibular swelling secondary to a dental infection. A CT scan of the neck identified an incidental 27 mm tongue base mass and the absence of any cervical thyroid tissue. This mass was not observable on examination of the oropharynx but was seen on fine nasendoscopy while thyroid function tests showed good thyroid stimulating hormone suppression. Her acute dental infection was treated and, following multidisciplinary team discussion, she was diagnosed with an ectopic lingual thyroid. She was offered different management options including no intervention and radio-iodide treatment but opted for transoral robotic resection. The lesion was resected en bloc with clear margins and histology confirmed lingual thyroid tissue. Since the procedure, she has remained free of sleep apnoea with a significantly improved quality of life.

Enlarged hemorrhagic lingual thyroid managed with transoral robotic surgery.

PURPOSE: Lingual thyroid glands are rare embryologic variants of undescended tissue centered in the base of the tongue. Despite notable size, many lingual thyroids can be asymptomatic, though intervention is warranted for progressive or emergent symptoms. We report a rare manifestation of a hemorrhagic lingual thyroid addressed with both interventional radiology and robotic techniques. METHODS: A previously asymptomatic 41-year old female presented to the emergency department with massive hematemesis after significant binge drinking and vomiting. Emergent intubation was performed and gastroenterology workup ruled out Mallory-Weiss tears or ruptured esophageal varices. Fiberoptic laryngoscopy and imaging revealed a bleeding tongue base mass concerning for malignancy. RESULTS: Aberrant feeding vessels with identifiable blush were embolized by Neurointerventional Radiology and the patient underwent tracheostomy for airway protection. Lingual thyroid was confirmed by biopsy and the mass was definitively resected via transoral robotic surgery. The patient had no further bleeding events and was decannulated uneventfully. CONCLUSIONS: Lingual thyroid glands can present with life-threatening hematemesis and obstruction that may masquerade as entities of vascular or neoplastic origin. Management encompasses multidisciplinary diagnostic confirmation, airway protection, and minimally invasive resection that minimizes functional morbidity.

Thyroid tissue outside the thyroid gland: Differential diagnosis and associated diagnostic challenges.

The presence of thyroid tissue outside of the thyroid gland may occur in various clinical settings and anatomic locations and includes both benign and malignant differential diagnoses. Some of these entities include thyroglossal duct cyst, lingual thyroid, parasitic nodule, thyroid tissue within a lymph node and struma ovarii. In routine daily practice, these entities do pose diagnostic challenges for the pathologists. Differential diagnostic considerations depend largely on the location of lesion and the histologic features. A definitive diagnosis may remain unclear in some cases while knowledge is still evolving in others i.e., incidentally detected bland appearing thyroid follicles in a lateral neck lymph node. This article aims to elaborate on the various entities characterized by thyroid tissue outside of the thyroid gland, both benign and malignant, and the relevant differential diagnostic considerations.

Obstructive lingual thyroid; suprahyoid intracervical surgical procedure - A case report. / Tiroides lingual obstructivo, intervención quirúrgica vía transcervical suprahioidea: reporte de un caso.

INTRODUCTION: The treatment of lingual thyroid is controversial and should be individualized. Options include hormonal replacement, surgery in the presence of bleeding and obstruction of the airway and the digestive tract, and radioisotope therapy. CLINICAL CASE: 8-year-old girl presenting with discomfort when swallowing. A pink, well-vascularized mass, not painful or ulcerated, protruding from the base of the tongue and virtually closing the whole oropharynx, was observed. Absence of thyroid tissue in its normal position was reported by the ultrasound department. Cervical computed axial tomography confirmed the diagnosis and the presence of pharyngeal obstruction. Thyroid hormone replacement was established. As a result of dysphagia symptom progression, surgery was indicated. Thyroid removal was performed by means of a cervicotomy, with re-implantation of thyroid tissue laminas. The postoperative course was uneventful and replacement treatment was maintained, with an excellent clinical status four years later.

INTRODUCCION: El tratamiento de la tiroides lingual es controvertido y debe individualizarse. Las opciones incluyen el reemplazo hormonal, cirugía en presencia de hemorragia y obstrucción de la vía aérea o digestiva, y la terapia con radioisótopos. CASO CLINICO: Niña de 8 años de edad, con molestias a la deglución. Se observa masa rosada, muy vascularizada, no dolorosa ni ulcerada, que protruye desde la base de la lengua y cierra prácticamente toda la orofaringe. Ecografía informa ausencia de tejido tiroideo en su posición normal. Tomografía axial computarizada cervical comprueba el diagnóstico y la obstrucción faríngea. Se indicó tratamiento sustitutivo de las hormonas tiroideas. Ante la progresión de los síntomas de disfagia, se indicó cirugía. Se describe la exéresis tiroidea por vía cervical, suprahioidea, con reimplante de láminas de tejido tiroideo. Evolucionó sin complicaciones y se mantiene tratamiento sustitutivo, con excelente estado clínico después de cuatro años.

A case report of coexistence of ectopic lingual thyroid with hypoplastic normal thyroid gland.

Ectopic lingual thyroid along with a normally located thyroid gland is an uncommon condition caused by an aberrant descent of thyroid during embryogenesis. It is more common among females and expresses during puberty, pregnancy and menopause. It is mostly associated with hypothyroidism. Patient usually presents with complaints of dysphagia, dysphonia and suffocation. Treatment of choice depends upon the primary complaint of the patient. We present the case of a young female who underwent tracheostomy to relieve respiratory tract obstruction during puberty and was later diagnosed as a case of ectopic lingual thyroid by radioactive iodine uptake and CT scan imaging. She had an associated hypothyroidism; patient was then put on thyroxine and after making her euthyroid she was operated by transoral route and her ectopic lingual thyroid was removed. She was discharged on a maintenance dose of thyroxin.

Resección transoral endoscópica de tiroides lingual / Endoscopic transoral resection of lingual thyroid

La presencia de tejido tiroideo ectópico en la base de la lengua es muy infrecuente, y la mayoría de los pacientes tienen hipotiroidismo. La indicación de tratamiento depende de la presencia o no de síntomas; la cirugía es la primera elección. Diversas técnicas quirúrgicas han sido descriptas, pero para nosotros el abordaje transoral con endoscopios constituye la mejor opción, por la buena exposición y la mínima morbilidad que produce. Se describe el caso clínico de una mujer que consultó por odinofagia, con diagnóstico de tiroides lingual y que fue tratada con éxito mediante un abordaje transoral con asistencia de endoscopios. (AU)

The presence of ectopic thyroid tissue at the base of the tongue is very rare, and most patients have hypothyroidism. The indication of treatment depends on the presence or not of symptoms, surgery being the first choice. Various surgical techniques have been described, being for us the transoral approach with endoscopes the best option, due to the good exposure, and minimum morbidity that it produces. The clinical case of a woman who consulted for odynophagia, with a diagnosis of lingual thyroid and who was successfully treated by a transoral approach with endoscopic assistance is described. (AU)

Caso Clínico: Tiroides Lingual Ectópica en Paciente Adulta Hipotiroidea. Diagnóstico Casual por Tomografía Computarizada. / Clinical Case: Ectopic Lingual Thyroid in Adult Hypothyroid Patient. Casual Diagnosis by Computed Tomography.

Introducción: Los casos de tiroides ectópica localizados en la base de la lengua son anormalidades congénitas raras y difíciles de diagnosticar. Razón de presentación del caso. Caso Clínico: El caso corresponde a una mujer de 41 años con tiroides en base de la lengua diagnosticada incidentalmente con tomografía computarizada (TC), con antecedentes de hipotiroidismo y cáncer de mama derecha. Al examen físico de cuello no se palpa glándula tiroidea ni se observa masa o protuberancia en cavidad bucal. Por control del cáncer de mama, se solicita tomografía por emisión de positrones (PET) y ecografía de cuello, reportándose captación del radiofármaco en la región cervical anterior y superior de cuello, y ausencia de tejido glandular tiroideo a nivel habitual, respectivamente. Por cuanto, se realiza TC simple y contrastada observándose a nivel de la raíz de la lengua una imagen nodular hipercaptante que mide 23x20x20 mm, bien definida, contornos regulares, no infiltra tejidos adyacentes, impronta luz de la orofaringe, sin individualizar la glándula tiroides a nivel habitual, corroborando así el diagnóstico de tiroides ectópica lingual. Conclusión: El diagnóstico de tiroides ectópica en paciente adulto hipotiroideo es raro, por lo que debe considerarse la realización de TC si al examen físico y ecográfico no es palpable ni observable

Introduction: Cases of ectopic thyroid located at the base of the tongue are rare and difficult to diagnose congenital abnormalities. Reason for presenting the case. Clinical case: The case corresponds to a 41-year-old woman with thyroid based on the tongue diagnosed incidentally with computed tomography (CT), with a history of hypothyroidism and right breast cancer. On the physical examination of the neck, the thyroid gland is not palpated, and no mass or bump is observed in the oral cavity. For breast cancer control, positron emission tomography (PET) and neck ultrasound are requested, radiopharmaceutical uptake is reported in the anterior and upper cervical neck region, and absence of thyroid glandular tissue at the usual level, respectively. As a simple and contrasted CT scan, a hypercapting nodular image measuring 23x20x20 mm, well defined, regular contours is observed at the root of the tongue, does not infiltrate adjacent tissues, oropharynx light imprint, without individualizing the thyroid gland to usual level, thus corroborating the diagnosis of lingual ectopic thyroid. Conclusion: The diagnosis of ectopic thyroid in an adult hypothyroid patient is rare, so CT should be considered if the physical and ultrasound examination is not palpable or observable.

Congenital Oral Masses: An Anatomic Approach to Diagnosis.

Although congenital oral masses are rare, they are readily detectable during fetal US screening. Most congenital oral masses are benign, but some may cause mechanical airway obstruction, resulting in poor outcomes at delivery. The radiologist's ability to describe these abnormalities and their physiologic sequelae accurately can have a substantial effect on perinatal treatment. Furthermore, despite being rare, congenital oral lesions encountered at screening and at follow up fetal MRI provide the opportunity to make a specific diagnosis by following a simple anatomic approach. This article describes an anatomic algorithm as the framework for accurate diagnosis of congenital oral lesions. The imaging appearance of the most common congenital oral cavity neoplasms is outlined, including vascular anomalies, epulides, choristomas, congenital lingual thyroid anomalies, lingual hamartomas, and epignathi, and other conditions that mimic these at US. Also reviewed are perinatal management of masses that affect the fetal airway and the imaging features key to optimizing delivery outcomes. Online supplemental material is available for this article. ©RSNA, 2019.