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Tongue mass that turned out to be an adenoid cystic carcinoma 

Tongue mass that turned out to be an adenoid cystic carcinoma 

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Unusual presentations of thyroid neoplasms have been reported from time to time. Four such cases of bizarre presentations of thyroid malignancies seen in the ENT Department of ESIC Hospital, K.K. Nagar, Chennai, India are presented. These cases highlight certain important issues concerning the diagnosis and management. A review of the literature on...

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Context 1
... with thyroid malignancies present in one or more of the following ways: as a solitary nodule, enlargement of an existing nodule, cervical lymph node metastases, vocal cord palsy and pressure effects on the trachea and esophagus. Malignancies arising from ectopic thyroid tissue found along thyroglossal tract, lateral cervical regions, branchial cyst, trachea and myocardium have been reported in the literature [3, 6, 7, 9, 10, 16]. Four such unusual case sce- narios are presented. A 57-year-old woman presented with complaints of change of voice and dysphagia for 4 months and diffi culty in breathing for 15 days. Examination showed a fi rm mass in the midline from the level of the 2nd tracheal ring extending below the suprasternal notch. Few small lymph nodes were palpable in the posterior triangle on the left side. Routine hematological examination and chest X-ray were normal. An X-ray neck showed compression of lower tracheal air space. Barium swallow showed an extrinsic compression of the esophagus. A computerized tomogram (CT) scan of the neck showed a mass arising from the thyroid gland completely encircling the trachea extending up to the level of carina (Fig. 1). Thyroid function tests showed a marked reduction of TSH levels. Reduced uptake of isotope with patchy distribution was seen on thyroid TC 99 scan. Fine needle aspiration cytology (FNAC) of the mass and of the lymph nodes was not contributory. Flexible laryngo- tracheo-bronchoscopy showed bilateral vocal palsy and a bulge in the posterior wall of the trachea. Bronchi were normal. Tracheostomy was performed for stridor under local anesthesia due to diffi culty in intubation. During surgery it was seen that the entire neck was fi lled with a hard woody mass. Trachea was identifi ed with diffi culty after excision of the surrounding mass. The patient expired after two days due to suspected pulmonary embolism. A diagnosis of anaplastic carcinoma of thyroid was made on histological examination. A 62-year-old man presented with complaints of change of voice, neck swelling for six months and hemoptysis for fi fteen days. Examination showed a single hard fi xed Lymph node of the posterior triangle on the left side. No thyromegaly was noted. Routine hematological examination and X-ray of the chest were normal. X-ray of the soft tissues of the neck showed an ill-defi ned soft tissue shadow in the trachea. Direct Laryngoscopy done under local anesthesia revealed left vocal cord palsy. Flexible tracheo-bronchoscopy done under local anesthesia showed a mass arising from the trachea almost completely occluding the lumen. During the procedure, the patient developed severe stridor, necessitating a tracheostomy which was also done under local anesthesia. On opening the trachea, a soft mass fi lling the lumen was seen. Debulking of the mass was done to facilitate the insertion of tracheotomy tube. Histopathology of the mass showed papillary carcinoma of the thyroid. FNAC of the lymph node showed metastatic papillary carcinoma. A 45-year-old woman presented with increasing diffi culty of swallowing and change of voice of one year’s duration and swelling in the tongue for six months. Examination showed a large smooth mass arising from the posterior one third of the tongue on the right side and was found to be crossing the midline (Fig. 2). The mobility of the tongue was not impaired and the larynx was normal. All routine investigations and X-rays were normal and thyroid functions were within normal limits. The mass did not concentrate the radioactive isotope during thyroid scan. FNAC of the mass showed a picture of papillary carcinoma of thyroid. The mass was excised by lateral pharyngotomy approach and the diagnosis was confi rmed histologically (Fig. 9). A preliminary tracheostomy was done and the patient anes- thetized through the cuffed tracheostomy tube. A 63-year-old woman presented with a swelling on the dorsum of tongue for three years, a right-sided neck mass for six months and increasing dyspnea for 15 days. Examination showed a large, fi rm, circumscribed mass arising from the dorsum of the tongue at the junction of anterior 2/3rd and posterior 1/3rd (Fig. 3). Tongue mobility was not impaired and the larynx and hypopharynx were normal. Examination of the neck showed a 4 cm by 4 cm mobile level III node on the right side. Thyroid gland was normal. Other than the presence of diabetes, routine blood and urine examination were normal. X-ray chest showed the presence of multiple metastases on both sides (Fig. 4). FNAC of the neck mass showed a papillary carcinoma. Open biopsy of the tongue mass was consistent with adenoid cystic carcinoma. Thyronormalcy was seen on thyroid function tests. Thyroid scintigraphy however, showed the presence of a cold nodule in the right lobe of the thyroid gland (Fig. 5). Ultrasound scan revealed the presence of a partly cystic nodule in the right lobe of the thyroid gland. Benign, self-limiting autoimmune conditions such as Hashimoto’s thyroiditis and Riedel’s thyroiditis present as rapidly enlarging neck masses with signs and symptoms of extrinsic compression of trachea, esophagus and the recurrent laryngeal nerves. These conditions are self-limiting and carry good prognosis. On the other hand, anaplastic carcinomas of the thyroid gland are uncommon represent- ing only about 5% of all thyroid malignancies. These tumors have a very poor prognosis [14]. However, associa- tion of Riedel’s thyroiditis with follicular carcinoma of thyroid has also been reported [1, 2]. Wan et al. (1996) reported two cases of anaplastic thyroid carcinoma that closely mim- icked Riedel’s thyroiditis. Histologically, these tumors were predominated by acellular fi brotic tissue along with atypical spindle cells. The term “paucicellular variant” was used to describe this uncommon variant of anaplastic thyroid carcinoma [2]. The fi rst case in our series was similar to the ones described by Wan et al. This patient also presented with a hard woody mass infi ltrating into and replacing all the neck structures, presenting with signs and symptoms of compression typical of Riedel’s thyroiditis. Histologically, however the tumor consisted of fi brotic tissue along with the presence of malignant cells diagnostic of paucicellular variant of anaplastic thyroid carcinoma (Fig. 6). Lingual thyroid is one of the common sites of ectopic thyroid tissue. Many patients with this condition are asymp- tomatic. The disorder presents during times of growth and increased metabolic activity such as puberty and pregnancy. Obstructive symptoms include foreign body sensation, dysphagia, dysphonia and dyspnea. Both follicular and papillary carcinomas in lingual thyroid have been reported [7, 8]. These studies underline the fact that malignancy must be excluded in all the cases of lingual thyroid since the presence of malignancy completely changes the management of this rare condition irrespective of the presence or absence of a functioning thyroid gland. Our case is an example of this rare entity of a papillary carcinoma in lingual thyroid that was diagnosed by fi ne needle aspiration cytology (FNAC). Excision of this tumor was successfully carried out by a lateral pharyngotomy approach. Although ectopic thyroid tissue is found along the normal path of development, the presence of ectopic thyroid tissue at extrathyroidal sites such as mediastinum and heart has been widely reported [12, 13]. Rare instances of thyroid malignancies from extrathyroidal sites such as myocardium and ovarian dermoid have been reported [3, 4]. Cases of thyroid malignancies presenting as primary tracheal tumors have also been reported. These malignancies are believed to arise from the ectopic thyroid rests from the submucosa of the trachea [5, 6]. In our series, this rare case of papillary carcinoma arising from the trachea presented clinically like a laryngeal neoplasm with cervical metastases and was detected only when the trachea was examined since larynx did not show any evidence of malignancy on endoscopic examination. Enlargement of a part of or the entire thyroid gland was not observed in this case. Airway management was challenging the fi rst three cases. In the fi rst case, the patient developed severe stridor due to bilateral vocal cord palsy. Emergency tracheostomy was done under local anesthesia since endotracheal intubation was not possible. In the second case also, the patient developed severe stridor during fi beroptic bronchoscopy and emergency tracheosomy had to be done under local anesthesia. In the third case, general anesthesia was given through a cuffed tracheostomy tube after a preliminary tracheostomy. The case that presented as a tongue mass and a cervical mass with pulmonary metastases was a diagnostic chal- lenge. While the tongue lesion showed an adenoid cystic carcinoma (Fig. 7), the cytological picture of the neck mass was consistent with papillary carcinoma of thyroid. (Fig. 8). The problem was compounded by the detection of a non- functioning nodule in the right lobe of the thyroid gland. Instances of thyroid neoplasms arising from branchial cysts presenting as lateral ...
Context 2
... 63-year-old woman presented with a swelling on the dorsum of tongue for three years, a right-sided neck mass for six months and increasing dyspnea for 15 days. Examination showed a large, fi rm, circumscribed mass arising from the dorsum of the tongue at the junction of anterior 2/3rd and posterior 1/3rd (Fig. 3). Tongue mobility was not impaired and the larynx and hypopharynx were normal. Examination of the neck showed a 4 cm by 4 cm mobile level III node on the right side. Thyroid gland was normal. Other than the presence of diabetes, routine blood and urine examination were normal. X-ray chest showed the presence of multiple metastases on ...

Citations

Article
Full-text available
Several excellent guidelines and expert opinions on congenital hypothyroidism (CH) are currently available. Nonetheless, these guidelines do not address several issues related to CH in detail. In this article, the authors chose the following seven clinical issues that they felt were especially deserving of closer scrutiny in the hope that drawing attention to them through discussion would help pediatric endocrinologists and promote further interest in the treatment of CH. 1. How high should the levothyroxine (L-T4) dose be for initial treatment of severe and permanent CH? 2. What is the optimal method for monitoring treatment of severe CH? 3. At what level does maternal iodine intake during pregnancy affect fetal and neonatal thyroid function? 4. Does serum thyroglobulin differ between patients with a dual oxidase 2 (DUOX2) variants and those with excess iodine? 5. Who qualifies for a genetic diagnosis? 6. What is the best index for distinguishing transient and permanent CH? 7. Is there any cancer risk associated with CH? The authors discussed these topics and jointly edited the manuscript to improve the understanding of CH and related issues.
Article
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.
Article
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.
Article
Although lingual thyroid is the most common site for ectopic thyroid gland but carcinomas originating from lingual thyroid are extremely rare, accounting only for 1% of all ectopic thyroids. Here we represent a young female with a bleeding mass at the base of her tongue and review the diagnostic approach towards papillary thyroid carcinoma of lingual thyroid. The surgical treatment and follow up are discussed. A combination of radiological studies and histological evaluation should be deployed to investigate suspicious lingual thyroids. The perspective of diagnostic and therapeutic approaches for carcinomas of lingual thyroid is the same as orthotopic thyroid tissue.
Article
Introduction: Lingual thyroid cancer is a rare entity with a paucity of literature guiding methods of surgical treatment. Its location presents anatomic challenges with access and excision. Objective: We present a case of T4aN1b classical variant papillary thyroid carcinoma of the lingual thyroid that was removed without pharyngeal entry. We also present a review of the literature of this rare entity and propose a treatment algorithm to provide safe and oncologic outcomes. Findings: Our review of the literature found 28 case reports of lingual thyroid carcinoma that met search criteria. The trans-cervical/trans-hyoid approach was the most frequently used and provides safe oncologic outcomes. This was followed by the transoral approach and then lateral pharyngotomy. Complications reported across the series include 1 case of pharyngocutaneous fistula associated with mandibulotomy and postoperative respiratory distress requiring reintubation or emergent tracheostomy in 2 patients. Conclusion: The location of lingual thyroid carcinoma can be variable, and surgical management requires knowledge of adjacent involved structures to decrease the risk of dysphagia and airway compromise. In particular, for cases where there is extensive loss to swallowing mechanisms, laryngeal suspension can allow the patient to resume a normal diet after treatment.
Article
Tracheal invasion by differentiated thyroid carcinomas is rare and surgical technique to be performed is still controversial among surgeons. The aim of this article was to review the available literature on the tracheal invasion by differentiated thyroid carcinomas. A literature review on the literature related to tracheal invasion by differentiated thyroid carcinomas (- 2016 2000) was performed in PubMed / MEDLINE. The following keywords and phrases in English were used: thyroid, carcinoma, differentiated thyroid cancer, tracheal invasion and their corresponding translation into Spanish. No randomized clinical trials were found. Most articles were retrospective. Clinical characteristics of this lesion and its diagnostic methods were evaluated: laryngo-tracheoscopy, aspiration cytology, and imaging studies: clinical characteristics of the lesion and diagnostic methods were evaluated. Surgical indications and anesthesiology care were defined. Finally, a critical discussion was made on the diagnostic methods and surgical techniques used and the value and indication of each. The clinical condition, the tracheoscopy, imaging and cytopathology study are essential to establish the preoperative diagnosis. Wherever possible, the circumferential resection of the trachea should be the technique of choice in the treatment of patients with tracheal infiltration by differentiated thyroid carcinomas.
Article
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.