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.

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.

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.

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.

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.

Transoral videolaryngoscopic surgery for papillary carcinoma arising in lingual thyroid.

Carcinoma arising in lingual thyroid is an extremely rare entity accounting for only 1% of all reported ectopic thyroids. Here, we report a case of carcinoma arising in lingual thyroid, which has been successfully managed by transoral resection and bilateral neck dissections. A lingual mass 4-cm in diameter with calcification was incidentally detected by computed tomography at medical check-up. No thyroid tissue was observed in normal position. Ultrasound examination showed bilateral multiple lymphadenopathies. Fine needle aspiration biopsy from lymph node in his right neck was diagnosed as Class III and thyroglobulin level of the specimen was 459ng/ml. Due to the difficulty in performing FNA of the lingual masses, right neck dissection was performed in advance for diagnostic purpose. Pathological examination showed existence of large and small follicular thyroid tissues in several lymph nodes, suggesting lymph node metastasis from thyroid carcinoma. Two months after the initial surgery, video-assisted transoral resection of lingual thyroid with simultaneous left neck dissection was performed. Postoperative course was uneventful. Papillary carcinoma was found in the lingual thyroid and thyroid tissues were also found in left cervical lymph nodes. Video-assisted transoral resection was useful for the treatment of thyroid cancer arising in lingual thyroid.

Transposition of lingual thyroid gland to the submandibular region by transoral approach.

The purpose of this case report is to demonstrate surgical technique of only functional but symptomatic lingual thyroid gland transposition to submandibular region by transoral approach without mandibulotomy and tongue-splitting. A 37-year-old female patient was admitted to our hospital with dysphagia and apnea symptoms. Physical examination revealed 3cm×3cm lingual thyroid gland was detected at the tongue base. The patient was euthyroid and thyroid gland was not detected in the neck. Under general anesthesia, right submandibular gland excision and transposition of lingual thyroid tissue to submandibular region with dorsal lingual artery axis flap were performed by transoral approach. Thyroid hormones remained normal in the postoperative period. In conclusion transoral transposition of lingual thyroid to submandibular region as a flap without mandibulotomy is a minimally invasive and function preserving alternative approach. Besides preserving thyroid functions, this transoral surgical technique can be preferred by patients who avoid skin incision for esthetic concerns.

A young woman with oligomenorrohea and dysphagia.

A 35-year-old house wife attended our clinic with oligomenorrhea for the last three months. She also complained of diffuse body ache, joint pain and decreased bowel movements. Very often she experienced choking sensation in the throat and had difficulty in swallowing solid food. On examination, goitre was absent but a globular pink colour mass was detected at the base of the tongue.

A 51-Year-Old Woman With Hypoacusia and Increased Respiratory Effort in the Supine Position and OSA.

A 51-year-old woman with a personal history of vitiligo, normal thyroid hormone studies, a simple hysterectomy for multiple uterine myomas at age 35 years, and childhood adenotonsillectomy was seen for progressive hearing loss. She reported mild asthenia, cold intolerance, mild dysphagia with frequent choking while eating and drinking, and a progressive increase in inspiratory effort, especially in the supine position. Her partner described a progressively worsening history of snoring and witnessed apneic episodes, mostly in the supine position. Mild to moderate daytime sleepiness was also present.

Differentiated thyroid carcinoma in lingual thyroid.

The lingual thyroid is the most common form of thyroid ectopy. The ectopic tissue may display any disease affecting the thyroid, including malignancies, which have an estimated incidence of less than 1%. To date only 51 cases of lingual thyroid cancer were reported. Analogously to what observed in orthotopic thyroid, papillary carcinoma is the predominant histotype in lingual thyroid carcinoma. The higher frequency of lingual follicular thyroid carcinoma previously reported is possibly related to histological misclassification in some early reports, prior to the standardization of histological typing of differentiated thyroid carcinomas. Nonetheless, the frequency of the follicular histotype is not negligible, accounting for about one-third of the reported cases. Both natural history and prognosis of lingual thyroid carcinoma are poorly known, likely because of the rarity of the disease and the heterogeneity in the therapeutic approach. However, among the cases more recently reported, surgical excision of the mass, either alone or followed by radioiodine ablation, is the first-line approach, with only two cases treated by radioiodine alone. The nonsignificant rate of neoplastic transformation in lingual thyroid should encourage efforts to obtain a widely accepted consensus for the management of this rare condition, along with standardization of either diagnostic or therapeutic handling of malignancies arising in ectopic thyroid.

A large lingual thyroid extending to the epiglottis.

A 22-year-old man presented to our department with a mass on the base of his tongue. He had a cavernous voice causing a moderate speech disorder, and he had some difficulty in swallowing. He had severe problems with sleep, associated with apnoea. In a fibreoptic laryngoscopic examination, a large 4×5 cm vascular mass was detected extending from the base of the patient's tongue to his epiglottis. It covered the epiglottis. Thyroid scintigraphy showed only thyroid tissue on the base of the tongue. Surgery was initiated transorally under the guidance of a rigid endoscope, but as the mass continued extended to the epiglottis, a transhyoid approach was taken. At a 3-month follow-up, the patient was symptom free. Electrocautery-assisted resection under the guidance of a rigid endoscope can reliably be used in surgery of a lingual thyroid. However, a transhyoid approach provides a better view and also helps in achieving haemostasis.

Acute neonatal respiratory distress caused by a lingual thyroid: the role of nasendoscopy and medical treatment.

BACKGROUND: A lingual thyroid is a known cause of oropharyngeal obstruction in the neonate. It can be asymptomatic, or present as stridor, dysphonia, dysphagia or dyspnoea with faltering growth. The therapeutic options include surgical resection. CASE REPORT: A 6-day-old female neonate, born at 36 weeks gestation, presented with stridulous breathing and poor feeding. Although the cause was initially thought to be laryngomalacia, nasendoscopy revealed a lingual thyroid. The baby had deranged thyroid function, as detected on neonatal screening, but this result was not available until a later date. Despite being symptomatic, the patient was managed medically; thyroxine therapy was associated with resolution of the respiratory symptoms. CONCLUSION: Nasendoscopy provides valuable information about an ectopic thyroid gland. Thyroid replacement therapy may help to suppress the size of the ectopic gland and ultimately prevent an unnecessary surgical procedure.

Transoral robotic excision of ectopic lingual thyroid: Case series and literature review.

BACKGROUND: Surgical excision of an ectopic lingual thyroid has traditionally been associated with significant morbidity and has therefore been reserved for patients with severe obstructive symptoms or suspected malignancy. Transoral robotic surgery (TORS) has provided a minimally invasive approach to completely and safely excise ectopic lingual thyroid. METHODS: Three index cases were identified from the detailed clinical database of TORS patients. A systematic review of the management of ectopic lingual thyroid in the English literature was performed. RESULTS: TORS-assisted excision of a lingual thyroid gland was successfully performed in 3 patients with excellent functional outcomes CONCLUSION: TORS-assisted excision of an ectopic lingual thyroid is a safe and feasible treatment modality with minimal morbidity, and, in experienced hands, should be offered as a valid treatment for this pathology.