Research progress of ectopic thyroid cancer in thyroglossal duct cyst: A case report and literature review.

RATIONALE: Thyroglossal duct carcinoma, a rare clinical condition characterized by ectopic thyroid adenocarcinoma within thyroglossal duct cysts (TGDCs), typically confirmed through intraoperative rapid pathology, this condition generally has a favorable prognosis. Nevertheless, comprehensive treatment guidelines across all disease stages are lacking, the purpose of this study is to report 1 case of the disease and propose the treatment plan for each stage of the disease. PATIENT CONCERNS: A patient presented with thyroid swelling, classified as C-TIRADS 4A following a physical examination. Preoperative thyroid puncture identified papillary thyroid carcinoma, and genetic testing revealed a BRAF gene exon 15-point mutation. Ancillary tests showed a slightly decreased thyroid stimulating hormone (TSH) level (0.172) with no other significant abnormalities. DIAGNOSES: Preoperative fine-needle aspiration cytology (FNAC) confirmed right-side thyroid cancer. Intraoperative exploration uncovered a TGDC and intraoperative rapid pathology confirmed thyroglossal duct carcinoma. INTERVENTIONS: A Sistrunk operation and ipsilateral thyroidectomy were performed. OUTCOMES: Postoperative recovery was satisfactory. LESSONS: Thyroglossal duct carcinoma is a rare disease affecting the neck. Due to limited clinical cases and the favorable prognosis associated with this condition, there is currently no established set of diagnostic and treatment guidelines. According to tumor size, lymph node metastasis, thyroid status and other factors, the corresponding treatment methods were established for each stage of thyroglossal duct cancer, which laid the foundation for the subsequent treatment development of this disease.

Does thyroglossal duct arborization play a role in the post-surgical outcome of Sistrunk procedure in children?

PURPOSE: The purpose of the present study is to analyze thyroglossal duct cyst (TGDC) histopathological features, with focus on "arborization", in a cohort of pediatric patients who underwent surgical removal, and evaluate a possible correlation with clinical recurrences. METHODS: A retrospective analysis of all patients who underwent surgical resection for TGDC at the division of Pediatric Surgery of the University of Pisa from 2015 to 2020 was performed; for each patient, the following data were recorded: age, sex, clinical presentation, localization, size of the lesion, diagnostic tools, histopathological features, perioperative complications, recurrence and follow-up. RESULTS: With respect to arborization, following histopathological analysis 25/30 patients (83.3%) presented thyroglossal duct branching. After a median follow-up of 3.5 years, only 2 out of 30 patients (6.7%), one male and one female, respectively aged 4 y.o. and 6 y.o., presented recurrence within one year from first surgery. CONCLUSION: Surgery for TGDC remains a challenge for pediatric surgeons, while arborization was present in most of our cases which underwent surgery. With respect to the role of arborization, our study did not highlight sufficient conclusive data regarding their role in recurrence: instead, it showed wide resection as satisfactory, being the arborization present in most of the cases at histopathology.

What is the annual risk of infection in congenital midline neck cysts in children? Thyroglossal duct cysts versus dermoid cysts.

INTRODUCTION: Midline neck lumps in children are mostly found to be thyroglossal duct cysts or dermoid cysts. Thyroglossal duct cysts often have an associated sinus tract which may connect all the way to the foramen caecum on the tongue, while dermoids have no such connection. This study aims to estimate the annual infection risk for midline neck cysts based on our patient series, and to see if this differs between thyroglossal duct cysts and dermoid cysts. METHODS: All children seen at the Royal Hospital for Children, Glasgow who underwent surgical excision of a midline neck cyst between 1st January 2017 and 31st December 2021 were identified. In those whose cyst had been infected prior to surgical excision, the age at which the first episode of infection occurred was recorded and used to calculate a survival curve. RESULTS: We identified 53 children (29 male, 24 female) aged 1-16 years (median 4) at the time of surgical excision. There were 26 thyroglossal and 24 dermoid cysts, plus 2 with indeterminate histology and 1 lymph node. Of the 24 dermoids, 4 suffered infection prior to surgery (17%), and 2 of these recurred after surgery (8%). Of the 26 thyroglossal cysts, 16 suffered infection prior to surgery (62%) and 5 of these recurred (19%). 78% of thyroglossal and dermoid cysts had at least 1 episode of infection by age 10 years. DISCUSSION: In a child with a congenital midline neck cyst that has never been infected, deferring surgery for a year comes with a 7.8% risk that the cyst will get infected.

Ectopic Salivary Glands - a Differential Diagnosis to a Thyroglossal Duct Cyst.

Background: Midline developmental neck lesions primarily consist of thyroglossal duct remnants. Their recurrence is uncommon following thorough resection, which includes hyoid removal (the Sistrunk procedure). Case report: A 3-year-old girl presented with mucoid secretion drainage and swelling in the anterior mid-neck region, clinically resembling a thyroglossal duct remnant. Following an initial Sistrunk procedure, the lesion recurred, prompting a subsequent resection. Histological analysis revealed a mucocele alongside acinar and mucous ectopic salivary glands. Conclusions: The ectopic salivary gland can manifest along the midline of the neck and may clinically resemble the signs and symptoms of a thyroglossal duct cyst. Importantly, it can exhibit recurrence post-surgery, even following hyoid resection.

[Clinical analysis of gasless transoral vestibular robotic resection of thyroglossal duct cysts].

Objective:This study aimed to explore the safety and feasibility of gasless transoral vestibular robotic resection of thyroglossal duct cysts. Methods:The clinical data of patients who underwent gasless transoral vestibular robotic resection of thyroglossal duct cysts at the Department of otolaryngology, Sun Yat-sen Memorial Hospital, Sun yat-sen university from September 2020 to May 2022 were analyzed. The operative time, blood loss, postoperative complications, postoperative pain score, postoperative aesthetic score, and recurrence were prospectively evaluated. Results:All patients completed the operation successfully and no case conversed to an open operation. The operation time was 104.00（95.00, 131.25） minutes, and the surgical blood loss was 15.00（10.00, 16.25） mL. The drainage volume was（59.71±9.20） mL. Postoperative pathology was consistent with thyroglossal duct cysts. There was no local reswelling, subcutaneous hematoma, emphysema, skin flap necrosis, infection and other complications. The postoperative hospital stay was 3.00（2.00, 3.00） days. Six patients had mild sensory abnormalities of the lower lip 3 months after surgery, and all patients were satisfied with the cosmetic outcomes. No recurrence was found during the 5-26 months follow-up. Conclusion:gasless transoral vestibular robotic resection of thyroglossal duct cysts is safe and feasible, with hidden postoperative scars and good cosmetic outcomes. It can provide a new choice for patients with thyroglossal duct cysts.

Thyroglossal duct cyst carcinoma case series-Management strategy and outcomes.

OBJECTIVE: To review the clinical presentation, diagnosis, pathology and management strategies in a modern cohort of patients with thyroglossal duct cyst carcinoma. STUDY DESIGN: Retrospective case series following PROCESS Guidelines. SETTING: Comprehensive cancer centre. METHODS: Data recorded included: gender, age at diagnosis, clinical presentation, thyroid function, diagnostic investigations, cytological results, final histology, staging and follow up status. The risk of malignancy in cytological analysis was stratified according to the Royal College of Pathologists classification in United Kingdom. RESULTS: Twelve patients were included. The majority of patients (66.7%) presented with an isolated thyroglossal duct cyst. Only 4 patients had preoperative cytological suspicion of carcinoma (sensitivity: 33.3%). At the time of presentation all patients were euthyroid. Following diagnosis of malignancy, a total thyroidectomy was performed in all patients, with the exception of 2, who had a thyroglossal duct cyst carcinoma of less than 10mm. Among the 10 patients who underwent total thyroidectomy, 7 (70%) patients had proven carcinoma in the thyroid gland, 3 with deposits of less than 10mm. The average size of the thyroid cancer deposits was 7.2mm (1-20mm). With a mean follow-up of is 44 months (5-120), all patients were alive and free of recurrence at the end of the study period. CONCLUSION: Thyroglossal duct cyst carcinoma is a rare condition and its management should be discussed in a multidisciplinary meeting. As with differentiated thyroid cancer originating in the thyroid gland, it bears extraordinary survival rates. Accordingly, the management of these cancers has shifted towards a more conservative approach although its peculiarities must be taken into account: ease of extracystic invasion and possible different lymph node invasion.

[Papillary carcinoma in a thyroglossal duct remnant: a case report].

This case report has described a case of papillary carcinoma of thyroglossal duct in a young male. This patient was admitted with a mass in the anterior neck for 2 years. Preoperative Bultrasonography, CT and MR showed a subcutaneous cystic mass with irregular calcification shadow in the central region of the neck without obvious enhancement. Initial diagnosis was thyroglossal duct cyst, and was excised by Sistrunk under general anesthesia. The postoperative pathological examination showed thyroglossal duct cyst combined with thyroid papillary carcinoma, which was confirmed by immunohistochemistry as thyroglossal duct papillary carcinoma.

Avascular midline oropharyngeal anatomy allows for expanded indications for transoral robotic surgery in pediatric patients.

Transoral robotic surgery (TORS) in children is in its infancy, and indications have been primarily limited to lingual tonsillar hypertrophy and superficial mucosal lesions. However, the relatively avascular channel of the midline posterior tongue, vallecula, and posterior hyoid space provides a safe plane of dissection for deep lesions of the tongue and access to structures in the anterior neck. As robotic surgeons gain experience, application of this technology will continue to grow. The method is retrospective case series. We present seven patients who had either a primary (n = 3) or recurrent (n = 4) lingual thyroglossal duct cyst (TGDC) and underwent TORS excision. Four of the seven patients also underwent transoral resection of the central portion of the hyoid bone, while three had central hyoid resection during prior surgery. Two minor complications occurred with no evidence of lesion recurrence after mean follow-up of 19.7 mo. The midline avascular channel of the tongue allows for relatively bloodless surgical access to pathologies of the midline base of tongue and anterior neck. Lingual thyroglossal duct cysts can safely be removed via a TORS approach with evidence of limited recurrence. Robotic technology can provide safe and effective surgical alternatives for children with a variety of pathologies, and we aim to promote the widespread adoption of TORS in pediatric head and neck surgery by sharing our knowledge and clinical experience. Further study and publication are needed to establish safety and efficacy.

Thyroglossal duct diseases: presentation and outcomes.

OBJECTIVE: The incidence of thyroglossal duct diseases in the general population is about 7%. We aimed to demonstrate the clinical presentations and management of thyroglossal duct diseases. METHODS: We conducted a retrospective review of all patients who underwent surgery for histopathologically confirmed thyroglossal duct cyst, sinus, or fistula at a single center. RESULTS: A total of 151 cases were included in this study. There were more female patients (87, 58%) than male patients (64, 42%). The patients' ages ranged from 1 to 63 years old. The most prevalent complaint was painless upper midline neck swelling (93.3%). Most cases were diagnosed as thyroglossal duct cysts (137, 90.7%). Six cases (4%) were associated with carcinoma. All the cases were managed using the modified Sistrunk procedure. There were no procedure-related complications, and five cases of recurrence. CONCLUSIONS: Although thyroglossal duct cyst is the most common neck anomaly in children, it may also present with various characteristics later in life. This condition can be managed successfully without complications and with a low recurrence rate.

A large thyroglossal duct cyst and its management: a case report.

Thyroglossal duct cysts (TGDC) are congenital neck cysts, formed as a result of the failure of the thyroglossal duct to involute during embryogenesis and their mean size is 1.5-2.4 cm. We present a case of a 44-year-old male who presented with a history of a large anterior neck mass measuring 8.7x6x6.4 cm and causing dysphagia and mild dyspnea. After being mistaken for a goiter, a clinical diagnosis of TGDC was made based on history, clinical and radiographic findings. The patient was treated with Sistrunk's procedure. No recurrence was noted on follow-up. Thyroglossal duct cysts are generally well-defined small lesions, but even bigger ones are not linked with severe symptomatology. The larger size at presentation may increase the list of potential diagnoses and lead to diagnostic dilemmas. Every effort should be made to rule out malignancy before surgery. Sistrunk's procedure with dissection of the posterior hyoid space should be the standard of care.

[Hyoid body morphology of thyroglossal duct cyst: a comparative study based on imaging].

Objective:To provide reference for preoperative diagnosis and treatment of thyroglossal duct cyst by studying the morphological changes of hyoid body. Methods:The CT data（midsagittal image） of congenital thyroglossal duct cyst（TGDC group） diagnosed by pathology and the control group（C group） were collected from January 2016 to October 2021. The differences of hyoid body height（HBH）, hyoid body width（HBW）, hyoid bone thickness（HBT）, HBW/HBH, HBT/HBH between the two groups were compared. HBH, HBW and HBT were analyzed by t-test; The HBW/HBH and HBT/HBH were analyzed by Mann-Whitney U test. Results:Twenty-nine cases were included in the TGDC group and 58 in the C group. The HBH in the TGDC group was（8.93 ±0.22） mm and that in the C group was（8.94±0.12） mm, there was no significant difference between the two groups（P>0.05）. The HBW in the TGDC group and the C group were（5.09±0.21） mm and（4.48±0.11） mm, and the HBT were（3.84±0.12） mm and（3.13±0.08） mm, respectively, the difference between the two groups was statistically significant（P<0.05）. The average rank sum of HBW/HBH in the TGDC group and the C group was 53.95 and 39.03, respectively, and the average rank sum of HBT/HBH was 59.90 and 36.05, respectively, the difference between the two groups was statistically significant（P<0.05）. Conclusion:The morphological changes of hyoid body of thyroglossal duct cyst may be helpful for preoperative diagnosis, and it also suggests that hyoid body resection may reduce the possibility of postoperative recurrence.

Thyroglossal Duct Cyst Papillary Carcinoma With Airway Compromise.

Thyroglossal duct cysts are typically benign and usually asymptomatic. Malignant transformation is uncommon. Intralaryngeal extension is rare and results in dysphonia or dyspnea. There is no literature nowadays reporting the thyroglossal duct cyst carcinoma combining the clinical features of intralaryngeal extension. The authors present a case of progressive hoarseness and midline neck mass for 2 years. The laryngoscope and computed tomography revealed a 6-cm thyroglossal duct cyst containing ectopic thyroid tissue with intralaryngeal extension and causing airway obstruction. Complete excision with Sistrunk operation revealed papillary thyroid carcinoma. The patient resumed normal phonation after the surgery. There was no evidence of tumor recurrence and no hoarseness or dyspnea at 6 months follow up. This is the first reported case of a huge thyroglossal duct cyst carcinoma with intralaryngeal extension causing airway compromise. Complete excision of tumor is essential and vital to the symptom relief. A thyroglossal duct cyst carcinoma with endolaryngeal involvement should be considered in the differential diagnosis when the case has a massive midline neck mass with ectopic thyroid tissue and develops dyspnea or hoarseness concurrently.

[Transoral robotic surgery for treatment of lingual thyroglossal duct cyst].

Objective: To investigate the feasibility, safety and efficacy of transoral robotic surgery (TORS) in the treatment of lingual thyroglossal duct cyst (LTGDC). Methods: The clinical data of 10 patients with LTGDC treated with TORS in Tongji Hospital affiliated to Tongji Medical College of Huazhong University of Science and Technology from May 2017 to November 2020 were analyzed retrospectively,including 6 males and 4 females, aged 5-44 years. The cysts were fully exposed, and resection usually started from the cephalic side of lesions. The range of resection was 3 to 5 mm away from the lesions, and partial hyoid bone was removed if necessary. Intra-operative robotic set-up time,operation time and estimated blood loss,and post-operative local bleeding, dyspnea and recovery time for oral intake were analyzed. SPSS 12.0 software was used for statistical analysis. Results: The cysts in all 10 patients were successfully resected by TORS with da Vinci Si surgical system. The mean robotic set-up and exposure time, operation time, estimated intraoperative blood loss and recovery time for oral intake were (15.5±7.1) min, (17.6±7.4) min, (8.9±6.4)ml and (2.3±2.2)days, respectively. No patient required tracheostomy intra-or post-operatively, and no symptoms of airway obstruction, postoperative bleeding, pharyngeal fistula, hoarseness and neurological impairment occurred after operation. The patients were followed up for 5 to 47 months, with median follow-up time of 17 months, and no recurrence was observed. Conclusion: TORS is safe and feasible for resection of LTGDC, with rapid recovery and low recurrence rate.

[Ectopic thyroid carcinoma in front of hyoid bone misdiagnosed as thyroglossal cyst：a case report].

A clinical case of ectopic thyroid carcinoma in front of hyoid bone was reported in this paper. The patient, a 17-year-old female, presented with an enlarging neck mass of 1-week history. Physical examination revealed a 3 cm×2 cm neck mass in front of the hyoid bone. Ultrasonographic depicted as a cystic solid mixed echogenic mass with punctate strong echogenicity. CT scan showed a cystic-solid mass in front of the hyoid bone with punctate calcifications. The patient was misdiagnosed as a thyroglossal duct cyst and underwent surgery. The final pathological diagnosis was papillary thyroid carcinoma with cyst formation.

Symptomatic Thyroglossal Duct Cyst Presenting With Synchronous and Localized Tongue Base Extranodal Mantle Cell Lymphoma. A Unique Case Report Demonstrating Lymphoma Colonization of Cyst Wall, Potentially Relating to a Persistent Embryological Foramen Cecum Remnant.

A 77-year-old male presented with a progressively enlarging midline neck mass. On further investigation he was found to have synchronous thyroglossal duct cyst and extranodal mantle cell lymphoma (MCL) localized to the base of tongue. Both pathologies were managed simultaneously with a surgical approach and the patient remained in clinical remission at the time of publication without indication for systemic oncological treatment. Histology revealed primary extranodal nonblastoid MCL forming a base of tongue mass, with colonization of the thyroglossal duct cyst. Lymphoma was also found in the epithelium of a crypt-like tract traversing one of the tongue base tumor sections. This tract was anatomically and histologically consistent with documented descriptions of the foramen cecum. This case report illustrates a previously undescribed temporal, clinical, and histological association between a base of tongue MCL and symptomatic thyroglossal duct cyst. We provide evidence for a potential causal relationship for the presentation of the thyroglossal duct cyst as a result of oropharyngeal MCL, in the absence of clinical and histological evidence of disseminated disease, directly infiltrating from its tongue base origin to the infrahyoid neck region, potentially via an embryologic foramen cecum remnant. We also highlight the crucial role of the histopathologist in multidisciplinary clinicopathological discussion in demonstrating how fundamental embryological and microanatomical relationships can unite apparently separate diseases.

Cancer in thyroglossal duct cysts.

BACKGROUND: Thyroglossal duct cysts (TDC) account for 7% of midline neck swellings. TDC carcinoma (TDCC) is rare, reported in 1% of all TDCs. We aimed to describe the incidence of TDCC, the accuracy of fine needle aspiration cytology (FNAC), and to identify suspicious, predictive ultrasound (USG) features. METHOD: A cross sectional study of TDC patients in the Monash University Endocrine Surgery Database from 2001 to 2021. RESULTS: Eighty-six patients had surgery for TDC, of median age 48 and a female preponderance (62%). Preoperative USG was used in 85% and FNAC in 57%. There were six cases (7%) of TDCC with papillary thyroid cancer (PTC). USG in five TDCC cases showed a solid nodule within the cyst wall. FNAC was undertaken in five TDCC cases from the solid nodule; malignant in one and suspicious for malignancy in two. Two TDCC patients had concurrent total thyroidectomy (TT), and three subsequently had a TT. Multifocal thyroid PTC was found in two patients. One patient had a recurrence in the lateral lymph nodes, nine years after excision of the TDCC and TT. All patients are alive and well with no distant metastases (median follow up 11 years). CONCLUSION: The 7% incidence of TDCC is higher than the usually reported 1% in the literature. We recommend a preoperative USG to evaluate the TDC, thyroid gland and cervical lymph nodes, and an FNAC to target the solid component of the TDC, and favour total thyroidectomy for all patients with TDCC.