Mucoepidermoid Carcinoma of the Thyroglossal Duct: Two Rare Entities in a Unique Case Report.

BACKGROUND: Mucoepidermoid Carcinoma (MEC) of the thyroid represents less than 0.5% of all thyroid neoplasms. Thyroglossal duct cyst carcinoma is a rare condition with only approximately 300 cases reported. CASE REPORT: A 34-year-old pregnant woman at 37 weeks gestation presented to an endocrinological center for primary autoimmune hypothyroidism. The thyroid ultrasound revealed a pseudonodular pattern. The patient was followed up after two years. She reported a full-term delivery without complications. A new thyroid ultrasound was performed, showing a cystic lesion in the median suprathyroid area, measuring 6 x 9 x 10 mm, not previously reported. After 4 months, the suprathyroid cystic lesion was confirmed by thyroid ultrasound, measuring 6 x 11 x 12 mm. The patient was referred for fine-needle aspiration cytology. Cytological examination showed lymphocytes, red blood cells, and some epithelial aggregates with large cytoplasm and nuclear polymetrism with oxyphilic aspects. The patient underwent the Sistrunk procedure for the suprathyroid lesion. The histological examination revealed lymphocytic thyroiditis in heterotopic thyroid tissue with solid cell nest, epidermoid epithelium, and mucus-secreting cells suggestive of low-grade mucoepidermoid carcinoma. The immunohistochemistry study was positive, exhibiting thyroid transcription factor 1 and cytokeratin-19. No positivity was observed for thyroglobulin, calcitonin, galectin-3, and Hector Battifora mesothelial antigen 1. The recent follow-up examination, 13 months after the surgery, has been found negative for disease recurrence. CONCLUSION: This is the first case of an MEC occurring within a thyroglossal duct. Considering the age of the patient, the histological diagnosis, and the absence of thyroid nodules and metastasis, we decided on the Sistrunk procedure without total thyroidectomy.

Comprehensive analysis and treatment of intraoral thyroglossal duct cysts with intraoperative techniques: a 7-case series review in a tertiary care center.

OBJECTIVE: Intraoral thyroglossal duct cyst is a relatively rare clinical disease. This article reviews the diagnosis and treatment process of 7 patients and explores the clinical characteristics of diagnosis and treatment of intraoral thyroglossal duct cyst in combination with past literature reports. METHOD: A retrospective analysis was conducted on 7 cases of intraoral thyroglossal duct cyst admitted to the Otolaryngology ward of Dalian Municipal Central Hospital from January 2017 to January 2024. The cases were recorded in terms of gender, age, symptoms, physical signs, radiological examinations, surgical methods, and postoperative complications. All cases were followed up, and the latest follow-up results were recorded. RESULTS: Among the 7 cases, 6 patients underwent laryngoscopic and radiological examinations before surgery, and 1 child was found to have a cyst during surgery. All cases were diagnosed with intraoral thyroglossal duct cyst and treated with plasma radiofrequency surgery. None of the patients had postoperative complications, and no recurrence was found in the six-month follow-up after discharge. CONCLUSION: Intraoral thyroglossal duct cyst is rare in clinical practice. It is important to pay attention to its differential diagnosis clinically, and careful review of images is required before surgery. Cryoablation with low-temperature plasma radiofrequency is not only minimally invasive and has a quick recovery but also has few complications and a low recurrence rate. It is a safe and effective treatment method that is worthy of clinical promotion. LEVEL OF EVIDENCE: Level 3.

Modified Sistrunk Procedure in 391 Pediatric Thyroglossal Duct Cyst Cases: Clinical Analysis of Efficacy and Recurrence Risk.

Objectives: The study aims to retrospectively summarize the clinical features of pediatric thyroglossal duct cyst (TGDC), investigate the efficacy of the modified Sistrunk (mSis) procedure, and analyze the recurrence risks. Methods: The clinical data of 391 children with TGDC admitted to Beijing Children's Hospital affiliated Capital Medical University and Baoding Children's Hospital from March 2012 to December 2021 were retrospectively analyzed. All patients underwent cervical ultrasound for preoperative evaluation. Twenty cases had magnetic resonance imaging and 8 cases had computed tomography for further evaluation. All patients underwent the standard mSis procedure, and clinical manifestations information, surgical information, complications, and prognosis were analyzed. Results: Among the 391 TGDC cases, 118 (30.2%) had a history of recurrent neck infection and 36 (9.2%) had undergone previous neck cyst and fistula resection surgeries, initially diagnosed as neck cyst (22 cases), TGDC (12 cases), or branchial fistula (2 cases), with only 6 cases having undergone partial hyoid bone resection in the previous operation. During the 15 to 156 months of follow-up, 10 children experienced local wound infection, but no other complications were reported. The recurrence rate was 2.30%, and the recurrence time ranged from 0.5 to 34 (average, 7.2) months post surgery. In the Poisson regression model examining factors related to recurrence, the P values of the 3 factors were <.05: clearness of the lesion boundary, surgical history, and maximum diameter and the relative risk (RR) values corresponding to the 3 risk factors, such as Exp (B), were 27.918, 10.054, and 6.606, respectively. Conclusions: The mSis procedure demonstrated safety and efficacy with fewer complications and a low recurrence rate of 2.30% in the study. Furthermore, the indistinct lesion boundary, surgical history, and large lesion diameter (>2 cm) were independent risk factors for recurrence in pediatric TGDC.Level of Evidence: IV.

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.

Surgical Removal of Rheumatoid Nodule That Presented as a Thyroglossal Duct Cyst: A Case Report.

Rheumatoid nodules are extraarticular complications in rheumatoid arthritis (RA). This report details how a 63-year-old female patient with a history of lung and kidney cancer presented to the otolaryngology clinic with left submandibular region neck pain and a lesion that looked like a thyroglossal duct cyst on imaging. The patient also had accompanying joint pain. After the patient underwent a full workup including positron emission tomography and computed tomography scans, she underwent surgery to remove the suspected thyroglossal duct cyst via a Sistrunk procedure. Pathology indicated that it was a rheumatoid nodule. The patient was never diagnosed with RA until after removal of the nodule was done. The patient ended up being evaluated by rheumatology and has subsequently been treated for RA. This rare case highlights the importance of interdepartmental communication and assessment of all patient symptoms on examination.

A Rare Recurrence of a Thyroglossal Duct Cyst Accompanied by Several Postoperative Complications in an Eight-Year-Old Boy: A Case Report.

A thyroglossal duct cyst (TGDC) is a fluid-filled mass in the neck resulting from the persistence of a duct from fetal development that typically regresses spontaneously. When it persists, it is most often removed in a surgical procedure known as a Sistrunk operation. This case study presents the intriguing case of an eight-year-old boy who presented to an otolaryngology clinic with both a recurrence of his TGDC, as well as several postoperative complications, after the Sistrunk operation was performed. After the initial procedure resulted in an incomplete removal of the TGDC, the patient was referred to Interventional Radiology for sclerotherapy. After several rounds of this treatment technique the cyst remnants still persisted along with their associated symptoms. Due to the very low likelihood of a recurrence being observed after surgical removal with subsequent sclerotherapy, the reappearance of the cyst raised several clinical questions. This report underscores the significance of a thorough evaluation and consideration of unique presentations when confronted with recurrent TGDCs.

Transoral endoscopic vestibular approach Sistrunk procedure (TEVAS)-case report and scoping review.

Background: Thyroglossal duct cyst (TGDC) is the most common congenital neck mass among the pediatric population. Less than 10% of the cases occur in adults. The standard of care for TGDC is surgical treatment with the Sistrunk procedure via a traditional transverse cervicotomy. This technique involves the resection of the cyst with its tract and the central portion of the hyoid bone body to avoid recurrence. The transoral vestibular approach has gained popularity as an alternative approach to open neck surgery in order to eliminate the transcervical scar associated with these procedures. Methods: We describe a case of an endoscopic Sistrunk procedure performed by the transoral vestibular approach. A scoping review of the transoral endoscopic vestibular approach Sistrunk procedure (TEVAS) was performed. The PubMed, Medline, Cochrane, Lilacs, Scielo, Mary Ann Libert and Scopus databases were systematically searched by using a Medical Subject Heading (MeSH)-optimized search strategy. The selection of papers followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines after setting inclusion and exclusion criteria. Results: The case was successful and without complications. Five studies were included in the final analysis for this review. Conclusions: This novel approach to the Sistrunk procedure is an effective alternative way to treat TGDC in selected patients who are motivated to avoid a visible neck scar.

Papillary Thyroid Carcinoma within a Thyroglossal Cyst: A Rare Case Report with Review of Literature.

Thyroglossal cyst (TGC) is the most common congenital anomaly of the thyroid gland and is found in approximately 7% of general population. It represents cystic degeneration of a remnant of the thyroglossal duct that failed to involute during gestation. Malignancy occurring in TGC is rare entity, accounting only for 1% of all thyroglossal cysts. We are presenting such a rare case of papillary thyroid carcinoma arising in a thyroglossal cyst. 30-year-old female presented with a painless, gradually progressive swelling below the chin extending to left side. On examination, swelling was noted in submental region and measuring 4 × 3 × 1 cm, firm in consistency and moves with deglutition and movement of tongue. On radiological investigations, lobulated cystic lesion in midline with thin septations was seen, suggestive of thyroglossal cyst. The thyroid of the patient was normal. Patient underwent Sistrunk procedure. On histopathology, papillary thyroid carcinoma in thyroglossal cyst was seen and it was invading cyst wall and superficial skeletal muscle. The 1st case of thyroglossal duct carcinoma was reported by Brentano in 1911. Till now 300 cases have been reported in literature. Carcinomas occurring in thyroglossal cyst are extremely rare, (< 1% cases). The most frequent histological type is papillary pattern followed by mixed(papillary and follicular), squamous cell, Hürthle cell, follicular and anaplastic variety. The etiology of thyroglossal duct carcinoma is unknown and neither good clinical history nor examination can lead to a preoperative diagnosis. Diagnosis is often incidental on histopathology as in present case. Thyroglossal duct carcinoma is a rare condition that comes as a surprise to both the patient and surgeon and should be considered in patients presenting with cystic midline neck masses.

Malignant degeneration of thyroglossal duct cysts: Clinical aspects, imaging findings and management: Case series.

INTRODUCTION AND IMPORTANCE: Thyroglossal duct cysts (TDCs) are congenital anomalies that can rarely undergo malignant transformation, with approximately 1 % of operated TDCs demonstrating malignant degeneration. Therapeutic management and follow-up methods are still controversial subjects. CASE SERIES PRESENTATION: We report 3 cases of a papillary carcinoma of thyroid occurring in a thyroglossal duct cyst. The diagnosis was suspected preoperatively in 2 patients, hence the indication of frozen section examination. All patients underwent Sistrunk procedure associated with total thyroidectomy and central neck dissection. Concomitant papillary thyroid carcinoma was found in 2 patients. A complementary treatment by radioactive iodine 131 with frenetic hormone therapy was undertaken in all the cases. The follow-up showed no recurrence. CLINICAL DISCUSSION: The clinical presentation of thyroglossal duct cyst carcinoma is often nonspecific, posing challenges in early diagnosis. The chosen treatment strategy, involving the Sistrunk procedure, total thyroidectomy, and central neck dissection, along with adjuvant therapy using radioactive iodine 131 and frenetic hormone therapy, was effective in preventing recurrence. CONCLUSION: The management of degenerated TDC is based on a multidisciplinary approach. Papillary carcinomas of TDC generally have a good prognosis.

Carcinoma arising from thyroglossal duct remnants.

Thyroglossal duct cysts (TGDCs) are unusual remnants of the thyroid gland that are rarely observed in association with carcinoma. The present study aimed to showcase the clinical characteristics, diagnosis and management of patients with TGDC carcinoma. It was a single-center study conducted from February 2016 to February 2020. The study involved the retrospective analysis of a series of cases with TGDC carcinoma. A total of 10 patients were included in the study, of whom eight (80%) were females. Their age ranged from 25 to 48 years with a mean age of 39.1 years. A total of five cases (50%) underwent only Sistrunk's procedure, four patients (40%) underwent total thyroidectomy along with Sistrunk's procedure and one patient (10%) was treated by Sistrunk operation and lobectomy. In all of the cases (100%), histopathological examination confirmed papillary thyroid carcinoma in TGDCs. In addition, in the thyroid tissue, 2 cases (20%) also had papillary thyroid carcinoma and 3 cases (30%) had papillary thyroid microcarcinoma. Radioiodine was administered in one patient (10%) with suppressive thyroxine. A three-year follow-up with ultrasound revealed no suspicious lesions in any of the cases. Although rare, carcinoma may develop in the thyroglossal cysts. In this situation, both the thyroid gland and different lymph node compartments should be evaluated for malignancy. Surgical intervention is the cornerstone of management.

Endoscopy-Assisted Thyroglossal Duct Cyst Resection: A Scoping Review.

OBJECTIVES: Thyroglossal duct cysts (TGDCs) are a common congenital mass in the cervical region. As the traditional surgical approach for TGDC removal, the Sistrunk procedure, often leaves a visible neck scar, the demand for improved cosmetic outcomes has increased. Emerging endoscopy-assisted approaches offer promise for addressing cosmetic concerns. We conducted a scoping review to evaluate the feasibility and safety of endoscopy-assisted TGDC surgery. DATA SOURCES: PubMed, Embase, and Cochrane databases. METHODS: Electronic databases were searched from their respective inception dates to January 2023. Data on surgical approach, patient demographics, surgical procedure, and postoperative outcomes were extracted and analyzed. The quality of the studies was assessed using the Joanna Briggs Institute Critical Appraisal Checklist. RESULTS: The literature search yielded nine articles published between 2011 and 2022. Overall, 85 patients in these studies successfully underwent endoscopy-assisted TGDC surgery using various approaches, including areolar, axillo-breast, transoral-vestibular, and transoral-sublingual. The operative time varied across the studies, ranging from 50 to 480 min. TGDC sizes ranged from 1 to 3 cm in diameter. Complications, including infection, skin bruising, and dysarthria, were reported in seven patients (8%). No cases of conversion to open surgery or postoperative recurrences were reported. CONCLUSION: Endoscopy-assisted surgery is a potential alternative for patients seeking TGDC resection with satisfactory aesthetic results while ensuring safety. However, existing evidence is insufficient to support the superior effectiveness of endoscopy-assisted TGDC surgery over the traditional Sistrunk procedure. Laryngoscope, 134:3038-3043, 2024.

Risk-adjusted analysis of perioperative outcomes after the Sistrunk procedure.

Objectives: Thyroglossal duct cyst (TGDC) is the most common pediatric congenital neck mass. The Sistrunk procedure is the standard method of excision and is associated with low rates of recurrence. This study aimed to review our institution's outcomes following the Sistrunk procedure, specifically the rates of wound complications and cyst recurrence. Methods: This was a retrospective case series of pediatric patients undergoing the Sistrunk procedure from June 2009 to April 2021. Results: A total of 273 patients were included. Of these, 139 (53%) patients were male and 181 (66%) were white. The average age at the time of surgery was 7.1 years. The overall cyst recurrence rate was 11%. The most common wound complications were seroma (14%) and surgical site infections (SSIs) (12%). Wound complications were associated with prior history of cyst infection (odds ratio [OR] 1.97, 95% confidence interval [CI] 1.07-3.60, z-test 2.2, p = .03). Pediatric surgery was associated with fewer wound complications (OR 0.18; 95% CI 0.05-0.6, z-test -2.78, p = .005). However, pediatric surgery operated on fewer patients with a history of cyst infection (36% vs. 55%, p = .012). Drain placement and postoperative antibiotics did not affect rates of wound complications. Conclusions: Prior cyst infection is associated with increased rates of postoperative wound complications. Postoperative antibiotics and drain placement did not significantly affect complication rates. Level of Evidence: 4.

A Rare Case of Thyroglossal Duct Cyst Cancer and Literature Review.

Thyroglossal duct cyst is the most common thyroid developmental abnormality with a prevalence of 7%, but thyroglossal duct cyst cancer is rare. The incidence of thyroglossal duct cyst cancer is about 1%. The diagnosis is limited by low yield on fine-needle aspiration biopsy (FNAB), and most cases are diagnosed after surgery. There is a paucity of data on the utility of thyroglobulin washout for diagnosis of thyroglossal duct cyst cancer, and it has not been mentioned in previous case reports/series. Papillary thyroid cancer is the most common pathology. Preoperative planning is important as the decision about total thyroidectomy with the Sistrunk procedure (excision of the thyroglossal duct cyst, middle part of hyoid bone, and surrounding tissue around the thyroglossal tract) depends on the presence of clinical or radiological thyroid abnormality. Thyroglossal duct cyst cancer has an excellent prognosis. However, owing to a lack of standard of care for this type of thyroid cancer, there is institutional variability in management. We present a case of thyroglossal duct cyst cancer in a man presenting with painless midline neck swelling. Imaging was suspicious for thyroglossal duct cyst cancer. FNAB was benign. The patient underwent the Sistrunk procedure and pathology was positive for papillary thyroid cancer.

Clinical Observation and Analysis of Thyroglossal Duct Cyst Carcinoma: A Report of 5 Cases.

Thyroglossal duct cyst carcinoma (TGDCCa) is a rare condition with only approximately 300 cases reported to date. There is a lack of comprehensive reporting on its clinical manifestations, ultrasound, contrast-enhanced computed tomography, magnetic resonance imaging (MRI) features, immunophenotyping, procedure, and prognosis following modified Sistrunk's procedure. This study aimed to address these gaps by analyzing and summarizing the clinical features of 5 cases of papillary carcinoma arising in thyroglossal duct cysts (TGDC).Five patients with papillary carcinoma in TGDC treated by modified Sistrunk's procedure were included. Their clinical manifestation, physical examination findings, iconography, pathological findings, treatment, and outcomes were analyzed in aiding the diagnosis and treatment of TGDCCa. Immunohistochemistry was used to confirm the papillary carcinoma subtype. The BRAFV600E mutation was detected in 2 patients. No evidence of cancer recurrence, distant metastases, and malignant changes in the thyroid was found after a mean follow-up of 29.8 months.The management of TGDCCa with papillary carcinoma in low-risk patients can be accomplished by performing a modified Sistrunk's procedure along with a regular follow-up imaging of the thyroid and neck. Although postoperative pathological diagnosis is the gold standard for diagnosis, it is equally crucial to comprehend the clinical manifestations and auxiliary diagnostic techniques before surgical intervention.

Thyroglossal duct cyst on the suprasternal region: An extremely unusual location.

INTRODUCTION AND IMPORTANCE: A thyroglossal duct cyst (TGDC) is the most common cause of congenital midline anterior neck mass. It arises as a cystic expansion of a remnant of the thyroglossal duct anywhere between the foramen cecum of tongue and the isthmus of the thyroid. They are found in juxtaposition to the hyoid bone in 85 % of cases. Based on the obtainable information, it has been documented that there exist only two reported instances of a thyroglossal duct cyst occurring on the suprasternal region, which is contemplated as an atypical location for TGDCs. The atypical location of the swelling and difficulty of diagnosis made us report this case. CASE PRESENTATION: This case report describes a 30 years old female patient who presented with a suprasternal swelling which was diagnosed to be a suprasternal TGDC after histopathology and a review of the literature on this topic. CLINICAL DISCUSSION: Thyroglossal duct cysts are usually asymptomatic, but may occasionally be infected by bacteria in the oral cavity, prompting the patient to seek medical care. It is mainly diagnosed at an early age if it is located on common anatomic areas. Later age of presentation or an unusual site like in this case makes the diagnosis difficult. Sistrunk procedure is the treatment of choice. CONCLUSION: Thyroglossal duct cyst at the suprasternal location is a very rare occurrence, but should be considered by the evaluating surgeon when he/she encounters midline neck swellings across all age groups.

Surgical approach for thyroglossal duct cyst cancer: a case report of challenging conventional thinking.

This article reports an older woman with a 2-year history of enlarging submental neck mass. Head and neck imaging displayed a group with a solid central component. Additionally, a fine-needle aspiration suggested neoplasia. Thyroglossal duct cyst cancer is sporadic, accounting for ˂1% of all thyroid malignancies. A Sistrunk procedure was followed by a total thyroidectomy and unilateral neck dissections. From histological and immunohistochemical examinations, the incidental finding of carcinoma indicates the diagnosis of thyroid tissue thyroglossal duct cyst carcinoma, as it is the most common site for malignancy in ectopic thyroid tissue. The patient had an excellent recovery without additional treatments after surgery.

Combined stepladder incision and Sistrunk procedure for en bloc resection of supraclavicular thyroglossal duct cysts.

The accurate diagnosis and therapeutic strategies of thyroglossal duct cysts (TGDCs) are challenging for surgeons if the opening with exudate is far from the hyoid bone. A 7-year-old boy presented with a right supraclavicular mass and persistent pus. Ultrasonography and magnetic resonance imaging revealed the fistula to the hyoid bone. Suspecting a TGDC, we combined the stepladder incision technique and the Sistrunk procedure. The first skin incision cut through the opening in a spindle shape, and the second incision was made in the skin just above the hyoid bone. This combined technique allowed en bloc resection of the TGDC and the hyoid bone to the base of the tongue. Cervical masses are commonly encountered in surgical clinics, and even distant openings off the midline must be considered in the differential diagnosis of TGDCs. This treatment strategy is essential for preventing the recurrence of TGDCs and ensures optimal cosmetic outcomes.

Papillary carcinoma on thyroglossal tract cyst: our attitude toward a rare pathology whose management is often controversial.

Malignant degeneration on remnants of the thyroglossal tract is a very rare phenomenon. In our practice setting, we report the management of papillary carcinoma on a thyroglossal tract cyst. This was a 44-year-old female patient with a postoperative diagnosis of papillary carcinoma of the thyroglossal tract with an atypical clinical and ultrasound presentation. She subsequently underwent total thyroidectomy and bilateral recurrent lymph node dissection, with resection of fibrous scar tissue in the previously operated hyoid region. We have discussed our therapeutic attitude to this rare pathology, the management of which is the subject of controversy.

Double Thyroglossal Duct Cyst: A Case Report.

Thyroglossal duct cysts are one of the most common cervical congenital anomalies. They occur along the thyroid migration tract, which extends from the base of the tongue through the midline of the neck to the level of the cricoid cartilage. Thyroglossal duct cysts present as a midline neck mass and are closely associated with the hyoid bone. Here, we describe a case where two cystic structures were found just inferior to the thyroid gland and inferior to the hyoid bone, suggesting a double thyroglossal duct cyst. It is important to diagnose and surgically manage thyroglossal duct cysts as they are associated with complications, such as infection and malignancy.

A rare location of papillary carcinoma: Thyroglossal duct cyst.

The inadequate closure of the thyroglossal tract paves the way for a thyroglossal cyst. Thyroglossal duct cyst (TDC) malignancy is quite rare. A thirty-nineyear-old female patient was admitted to the polyclinic with a complaint of palpable mass in the neck. Findings compatible with TDC were determined in the patient's neck screening and it was considered to be malignant due to irregular margins, apparent vascularization and punctual calcifications. Fine needle aspiration biopsy was unremarkable. TDC was excised by Sistrunk procedure and frozen examination was performed. Total thyroidectomy was performed additionally since the result of the frozen examination was found to be compatible with the primary papillary carcinoma of TDC. If preoperative biopsy does not provide a diagnosis, frozen section study will be beneficial in terms of both providing the early diagnosis and directing the operation strategy during the surgery in clinically or radiologically suspected patients.