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.

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.

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.

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.

TORS excision of lingual thyroid carcinoma: Technique and systematic review.

OBJECTIVE: Clinically significant lingual thyroid tissue has a prevalence of 1/3000-10,000, and in 70% of these individuals, the lingual thyroid is their only thyroid tissue. Malignant transformation is exceedingly rare. Herein, we present a case of lingual thyroid carcinoma with a systematic literature review and description of our treatment technique. DATA SOURCES: PubMed, Ovid. REVIEW METHOD: The primary author performed a search of the literature for reports of lingual thyroid carcinoma or ectopic thyroid carcinoma associated with the tongue. Articles that did not present novel data, presented cases of ectopic thyroid carcinoma outside the tongue, non-malignant cases, non-thyroid carcinomas, or were non-English articles were excluded. Studies were limited to those published in the last 60 years. RESULTS: There are 39 cases reported in the literature. 23 cases occurred in females. Age at diagnosis ranged from 12 to 86; cases were more commonly diagnosed in the second decade of life, then in the 5th and 6th decades of life. Dysphagia, globus sensation, episodes of bleeding, voice changes, and presence of a neck mass were common symptoms at initial presentation. Nearly all patients underwent some form of pre-operative imaging, but practices varied as to the type of imaging. Treatment included surgical excision of the tumor in all but one case that was successfully treated with radioactive iodine therapy alone. CONCLUSIONS: Surgeons should be aware of lingual thyroid, its presentation, workup, and carcinoma treatment. Tumors are amenable to surgical excision, possibly followed by radioactive iodine therapy. Advances in robotic and endoscopic surgery over the past decade now allow for less morbid excisions of lingual thyroid tumors.

LINGUAL THYROID: 35-YEAR EXPERIENCE AT A TERTIARY CARE REFERRAL CENTER.

OBJECTIVE: Lingual thyroid (LT) results from a developmental abnormality due to failure of the thyroid gland to descend to its pretracheal position. Given the low incidence of this disease, standardized management recommendations are lacking. We aimed to describe our institution's experience in LT management and to suggest a practice algorithm. METHODS: We conducted a retrospective review of LT diagnosed at Mayo Clinic, Rochester, Minnesota, between 1976 and 2010. Demographics, clinical presentation, laboratory data, treatment received, and outcomes were collected. RESULTS: We identified 29 patients with LT. Eighty-three percent were female; age at diagnosis ranged from 2 weeks to 68 years. Almost one-third of patients were symptomatic, with the most common symptoms being cough and hoarseness. The diagnosis of LT was incidental in 9 patients (31%). Seventy-two percent of patients developed hypothyroidism. Levothyroxine was the treatment of choice, followed by thyroidectomy. Two asymptomatic euthyroid patients were followed without any intervention. CONCLUSION: Management of patients with LT should be individualized and guided by the patient's symptoms and thyroid hormone status.

[Ectopic thyroid basi-lingual: A case report].

Introduction: The thyroid ectopic gland is a rare anomaly, especially when it's a lingual thyroid. It is characterized by aspecific clinical presentation, causing a diagnostic problem. The diagnosis is based on a combination of imaging techniques as well as histological examination. Case presentation: We are presenting a case of a patient with thyroid basi-lingual treated surgically. Discussion: The low incidence of ectopic lingual thyroid , and their clinical variability requires radiological and isotopic investigations. Conclusion: The diagnosis of this disease is primarily histological. The management of these ectopic thyroid is surgical.

Lingual thyroidectomy: the Mayo Clinic experience with transoral laser microsurgery and transoral robotic surgery.

OBJECTIVES: We report the clinical findings, surgical management, and outcomes for lingual thyroidectomy. METHODS: We performed a retrospective case review of lingual thyroidectomy performed at 3 tertiary-care academic referral centers between 1994 and 2012. RESULTS: Nine patients underwent lingual thyroidectomy for symptoms including globus sensation (6 patients), dysphagia (5 patients), and airway obstruction (5 patients). Before surgery, 3 patients had attempted medical suppressive therapy. Lingual thyroidectomy was performed by transoral laser microsurgery in 4 patients, transoral robotic surgery in 3 patients, transoral surgery without microscopic assistance in 1 patient, and an open approach with a modified Sistrunk procedure in 1 patient. Total thyroidectomy was attained in 7 patients, and subtotal resection in 2. The follow-up averaged 8 months, and all patients reported significant improvement in their symptoms. One patient had a recurrence. Complications included postoperative bleeding and epiglottic perforation in 1 patient and airway obstruction secondary to angioedema in another patient. There was no significant difference in operative times between transoral laser microsurgery (91 ± 16 minutes) and transoral robotic surgery (109 ± 35 minutes). Transoral surgery without microscopic assistance and open resection had longer operative times (206 and 246 minutes, respectively). CONCLUSIONS: Surgical resection of lingual thyroid glands achieves significant improvement in patient symptoms, with low rates of recurrence. We favor a total lingual thyroidectomy approach with use of either a microscope or a robotic endoscope for optical assistance.

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.

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.

Computed tomographic angiography study of the relationship between the lingual artery and lingual markers in patients with obstructive sleep apnoea.

AIM: To determine the relationship between the lingual artery and lingual markers for preoperative evaluation of the lingual artery in patients with obstructive sleep apnoea/hypopnoea syndrome (OSAHS). METHODS: A 16-section computed tomographic angiography (CTA) of the lingual artery was performed in 87 inpatient cases with OSAHS, from December 2007 to May 2009. The course of the lingual artery and the anatomic relationship between the lingual artery and the lingual markers were analyzed using CTA imaging. RESULTS: The course of the lingual artery with the tongue in a resting position was similar to that of the Big Dipper constellation (Plough) in the sagittal view of CTA imaging. The first segment of the lingual artery declined approximately 19.27 ± 5.24 mm, the middle segment of the lingual artery was forward approximately 19.30 ± 6.79 mm, and the ascending segment of the lingual artery rose approximately 52.49 ± 10.98 mm. The entry point where the lingual artery entered into the tongue was adjacent to the tip of the greater horn of the hyoid bone. The relationship between the second segment of the lingual artery and the greater horn of the hyoid bone was relatively steady with the tongue in whatever position. The interval between the bilateral greater horn of the hyoid bone equalled that between the bilateral lingual arteries. CONCLUSIONS: Recognizing some lingual markers in the patients with OSAHS, such as the greater horn of the hyoid bone, foramen cecum, circumvallate papilla, lingual vein and tongue midline, may facilitate the surgeon's ability to define the course of the lingual artery accurately in the treatment of OSAHS.