Evaluation of preoperative cutaneous fistula as a risk factor for recurrence of thyroglossal duct cyst in children.

The aim of this study is to investigate the risk factors for recurrence after thyroglossal duct cyst (TGDC) surgery, differentiating between infections with and without a cutaneous fistula. This is a retrospective analysis of all paediatric TGDC in a tertiary care centre with at least 2 years of postoperative follow-up. One hundred and thirty-one patients met the inclusion criteria of the study. A multivariate analysis was performed to analyse the main risk factors for recurrence (presence of a fistula, infection, age). 116/131 patients were managed primarily in our institution; 15 patients had previously been operated on in another centre. The recurrence rate was 4.3% (5/116) when the patient was operated on in our institution at the first operation, and overall, recurrence of TGDC occurred in 20/131 (15.3%) patients. Age was not a risk factor for recurrence (p = 0.596). Two or more episodes of preoperative TGDC infection were a statistically significant risk factor in univariate analysis (p = 0.021) but not in multivariate analysis adjusted for age and the presence of a cutaneous fistula (p = 0.385). In multivariate analysis, cutaneous fistula formation was an independent risk factor for recurrence when adjusted for age and preoperative TGDC infection (Hazard ratio = 5.35; p = 0.011). CONCLUSIONS: A preoperative cutaneous fistula was a critical and independent risk factor for recurrence of operated TGDC, whereas age and TGDC infection were not identified as risk factors for recurrence after surgery. This information should be given to patients and parents before surgery. WHAT IS KNOWN: • The risk factors for recurrence after thyroglossal duct cyst surgery described in the literature are preoperative infection and young age, but this is not supported by strong evidence. • The role of cutaneous fistula formation is unclear. WHAT IS NEW: • The main risk factor for recurrence of TGDC is the presence of a preoperative cutaneous fistula, with an estimated hazard ratio of 4.95 (p = 0.016) in multivariate analysis. • The presence of two preoperative infections was also associated with a greater risk of recurrence in univariate analysis; age and gender were not associated with an increased risk of recurrence.

Atypical thyroglossal duct cyst with intra-laryngeal and para-glottic extension.

Thyroglossal duct cysts (TDC) are the most common congenital neck masses. Although they are anatomically closely related to the larynx, intra-laryngeal extension is very rare. We present a case, review the literature and discuss the challenges of intra-laryngeal TDC. A 55-year-old man presented with a neck mass associated with dysphagia. Computer Tomography neck scan showed a midline cyst extending to the pre-epiglottic space with partial obliteration of the right pyriform sinus and narrowing of the larynx. The cyst was excised en-bloc via Sistrunk procedure. Intra-laryngeal TDC are surgically challenging due to risk of perforation into the aerodigestive tract.

[Papillary Carcinoma Arising in Thyroglossal Duct Cyst in the Right Lateral Neck].

A 47-year-old woman was admitted to our institution with the chief complaint of a right cervical mass. Imaging examination findings showed a cystic mass of 25mm with a nodular lesion in the right cervical region. Therefore, we performed extirpation of the right cervical cystic mass to allow diagnosis of the lesion. The histopathological findings showed a partial thyroid tissue on the cyst wall covered with glandular epithelium or metaplastic squamous epithelium, and tumor cells proliferating in the papillary form. Considering the histopathological evidence of the characteristic epithelium of the thyroglossal duct cyst, the potential carcinogenesis from the remnant thyroid tissues, and the absence of primary tumor in the thyroid gland, the patient was diagnosed with thyroid papillary carcinoma arising from the thyroglossal duct cyst in the right lateral cervical region. We found recurrence of the right cervical lymph node at 1 year and 5 months after the initial operation. Thus, we performed dissection of the right cervical lymph nodes. Two years and 10 months after the operation, neither recurrence nor metastasis have been observed. It was suggested that, thyroid papillary carcinoma arising from the thyroglossal duct cyst should be taken into consideration when a lateral cervical mass lesion is found.

Papillary Carcinoma Within a Thyroglossal Duct Cyst in a 17-Year-Old Child.

Thyroglossal duct cyst is a frequent event; however, papillary carcinoma within a thyroglossal duct cyst is rare, particularly in children. A 17-year-old girl presented with an asymptomatic mid-submental mass for the last 2 months. The diagnosis of thyroglossal duct cyst was made based on physical examination and computed tomography finding. After performance of Sistrunk procedure, an incidental papillary carcinoma within the thyroglossal duct cyst was observed on pathology. We reviewed the pediatric cases of thyroglossal duct carcinoma, and then decided not to perform a concurrent thyroidectomy. We will continue close follow-up for future thyroid involvement.

Thyroglossal Duct Cyst Carcinoma in Child and Adult. Two Case Reports.

Thyroglossal duct cyst carcinoma is rarely mentioned in literature representing only 0,7-1,6% of cases with these embryonic remnants. Two patients with thyroid duct cyst carcinoma, a 14-year-old girl and a 44-year-old man operated on in our department, both diagnosed postoperatively are described. In the first one a classical Sistrunk operation was performed removing a 3 cm asymptomatic mass, a milimetric papillary carcinoma being incidentally discovered at paraffin section pathology. The second case presented a 4 cm cyst with benign clinical and ultrasonic features excepting a 8 mm nodule in the right thyroid lobe. Cytology was inconclusive such as a Sistrunk procedure together with a right thyroid lobectomy were done. Pathology revealed a limited carcinomatous focus in the cyst wall but also a papillary thyroid microcarcinoma. Both cases was thereby diagnosed only after microscopic examination. Conservative approach adopted due to "œinnocent" clinical appearance, subcentimetric size and absence of any risk factor in the two cases achieved stable, verified over time healing confirming the favorable prognosis of this rare pathology.

Thyroglossal duct cyst accompanied by laryngomalacia and pulmonary sequestration.

A 2-month-old full-term female infant developed nasal stridor, which progressed to respiratory distress and poor sucking ability. Direct pharyngoscopy showed laryngomalacia and a midline cystic mass in the lingual region. The mass pressed on the epiglottis, causing dyspnea. Computed tomography incidentally revealed extralobar pulmonary sequestration. Direct deroofing of the lingual cyst and plication of the epiglottis were performed at 3 months of age, and the patient recovered from the respiratory distress. Histopathology of the cystic mass showed a thyroglossal duct cyst. Thoracoscopic resection of the pulmonary sequestration was then done at 17 months of age. Thyroglossal duct cysts in the lingual region may cause destabilization of the epiglottis and laryngomalacia, resulting in acquired respiratory obstruction. The combination of thyroglossal duct cyst, laryngomalacia, and pulmonary sequestration is rare; therefore, reports must be accumulated in order to explore the embryological origins of such cases.

Endoscope-assisted concurrent resection of thyroglossal duct cysts and benign thyroid nodules via a small submental incisions.

The objective of this study was to evaluate the technique of endoscope-assisted concurrent resection of thyroglossal duct cysts and benign thyroid nodules via a small submental incision. Endoscope-assisted thyroglossal duct cyst excision and subtotal thyroidectomy were performed via a small submental incision in six patients with thyroglossal duct cysts and benign nodular thyroid lesions. The thyroglossal duct cysts and thyroid lesions were completely excised. The incisions were 3-4 cm in length, and the mean operation time was 84 ± 11 min. The mean volume of blood loss was 19 ± 5 mL. Postoperative pathological examination findings confirmed the diagnosis of thyroglossal duct cyst in all patients. The thyroid lesions were nodular goiter in three patients and thyroid adenoma in three patients. No postoperative complications such as hematoma, pharyngeal fistula, hypocalcemia, recurrent laryngeal nerve paralysis, or dysphagia were observed. No changes in the planned surgical procedure were required, and no recurrent lesions were detected during follow-up. In conclusion, patients with thyroglossal duct cysts and benign thyroid lesions can be treated by endoscopic excision via a small submental incision, thereby avoiding large neck incisions and resulting in better postoperative cosmetic outcomes.

Clinical Characteristics and Prognosis of Infants and Children <3 Years of Age With Thyroglossal Duct Cyst Complicated With a Parapharyngeal Mass: A Case Series Study.

Thyroglossal duct cysts (TGDCs) are congenital and developmental abnormalities in infants and young children. This retrospective case series study examined the characteristics of 7 patients <3 years (mean age, 1.9 years) with TGDC complicated with a parapharyngeal mass treated at one hospital between January 2019 and 2022. Four patients had a painless mass around the neck, 2 had a painless mass associated with snoring, and 1 presented repeated swelling and pain. B-ultrasound suggested 6 cases of TGDC and 1 possible lymphangioma. All patients were treated with Sistrunk surgery to remove the TGDC. Six patients had no cyst recurrence during follow-up (6 months to 2 years). In conclusion, TGDC complicated with a parapharyngeal mass has complex and variable clinical manifestations. Completely removing the cyst while sparing thyroid cartilage and surrounding vascular and neuroanatomical structures is important to avoid complications. The patients are likely to be free from recurrence after surgery.

Symptomatic Lingual Thyroglossal Duct Cyst in Children: A Laryngomalacia Phenotype.

OBJECTIVES: Patients with lingual thyroglossal duct cyst (TGDC) can present as symptomatic with obstructive airway and feeding difficulties. METHODS: We present 3 cases of symptomatic lingual TGDC. RESULTS: All 3 patients were diagnosed with laryngomalacia and underwent further concurrent or delayed airway intervention, in addition to cyst removal. CONCLUSIONS: We reason that there is a phenotype of laryngomalacia in the symptomatic lingual thyroglossal duct cyst patients who present with symptoms due to disruption in laryngeal anatomy rather than the cyst itself causing obstructive symptoms. Distinguishing this phenotype, especially in comparison to other pathologies such as vallecular cysts, may better allow for planning of concurrent or delayed airway procedures and overall counseling of parents.

[Gasless submental approach endoscopic removal of thyroglossal cyst].

Objective: To explore the safety and feasibility of gasless submental approach endoscopic removal of thyroglossal cyst. Methods: This work prospectively included the clinical data of 13 patients who underwent the gasless submental approach endoscopic removal of thyroglossal cyst at the Department of Otolaryngology, the First Affiliated Hospital of Chongqing Medical University from August 2021 to February 2022. The operative time, bleeding volumes, postoperative complications, subjective pain levels, satisfaction with incisional scars, neck and facial deformities, and recurrences were prospectively evaluated by visual analogue scale(VAS) score. Results: All of 13 patients were successfully treated using this new technique. The patients had a median age of 38 years, ranging from 24 to 59 years, comprising of 3 males and 10 females. The length of the submental incision was about 3 cm and the median of operation time was 135 minutes. Postoperative complications were minimal. The median volume of blood loss was 10 ml. Surgical site swelling existed in 1 case and dysphagia for more than 1 week occurred in 2 cases. Patients were discharged from the hospital with a median of 3 days after surgery. On a VAS of 0-10 scores, the pain had a median of 2 on the first day after surgery, and the satisfaction with incision scars and neck and facial deformities showed a median of 8 at 6 months after surgery. There were no recurrences during the follow-up period of 9-15 months. Conclusion: Gasless submental approach endoscopic removal of thyroglossal cyst may be a reliable new surgical method that is safe and has cosmetic advantages.

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.

Papillary Thyroid Cancer in Thyroglossal Duct Cysts: A Pictorial Review and Case Series.

ABSTRACT: Thyroglossal duct cyst is the most common congenital neck mass, but the incidence of malignancy within a thyroglossal duct cyst is rare, estimated at 1%. Most cancers arising within thyroglossal duct cysts are incidentally detected after surgical excision. We present the preoperative radiologic findings of 8 patients with papillary thyroid cancer arising within a thyroglossal duct cyst, as evaluated on ultrasound, computed tomography, magnetic resonance imaging, and positron emission tomography scan.

Surgical management of primary thyroid carcinoma arising in thyroglossal duct cyst: an experience of a single institution in Serbia.

Thyroglossal duct cyst (TDC) carcinoma is a comparable rare entity and treatment strategies have not been standardized. Here, we report a favorable outcome of TDC carcinoma patients based on our therapeutic strategy. Twelve patients with TDC carcinoma treated in our department from 1986 to 2012 were enrolled. Ten patients underwent Sistrunk's procedure in other institutions and referred to our institution for re-operation after the diagnosis of TDC carcinoma and the remaining two underwent initial surgery in our institution. Eleven patients were diagnosed as papillary and one as follicular carcinoma originating from TDC. We performed total thyroidectomy for 11, and limited thyroidectomy for one patient. Three patients (25%) had carcinoma lesions in the thyroid. We routinely dissected level I bilaterally and 6 of 11 patients (55%) with papillary carcinoma-type TDC carcinoma had metastasis. Level II/III nodes were biopsied and if positive, we performed level II-IV dissection. Of the 5 patients positive for level II/III, 2 were also positive for level IV. For the 3 patients with synchronous carcinoma in the thyroid, we performed level VI dissection and two had metastasis in this level. To date, 1 patient showed a recurrence to the lung, but none of the patients in our series died of carcinoma. For surgery of TDC carcinoma, Sistrunk's procedure, total thyroidectomy with level I dissection is mandatory. Whether level II-IV dissection is performed depends on pathology of biopsied level II/III nodes. Level VI dissection is also recommended especially when carcinoma lesions are pre/intra operatively detected in the thyroid.

Synchronous carcinoma of thyroglossal duct cyst and native thyroid gland.

The unobliterated portion of embryological thyroglossal duct may present as cystic swelling later in life and may contain functional thyroid follicles. This cyst requires excision along with the entire thyroglossal duct remnant and adjacent portion of hyoid bone. At times, the excised specimen could demonstrate a focus of carcinomatous change inside the cyst wall. Very rarely, this thyroglossal duct cyst carcinoma could be associated with malignancy of native thyroid gland. This case report illustrates an interesting case of synchronous carcinoma of thyroglossal duct cyst and native thyroid gland. It also sheds light on the controversies related to the pathophysiology of such association and the dilemmas surrounding the management of thyroglossal duct cyst carcinoma, with or without concurrent carcinoma of thyroid gland.

The role of pre-operative infection in the recurrence of thyroglossal duct cyst.

OBJECTIVES: To evaluate the role of pre-operative infection in thyroglossal duct cyst recurrence and post-operative complications of Sistrunk procedure. METHODOLOGY: Medical records of 55 patients undergoing thyroglossal duct cyst excision from 1998 to 2005 were reviewed. The following data was collected: age at operation, gender, surgical technique, presence and length of infection prior to operation, time interval between resolution of infection and operation, recurrence, management of recurrence, and complications. RESULTS: Forty-two patients (76.3%) had no recurrence following Sistrunk procedure, while 13 patients (23.7%) underwent one or more additional procedures due to recurrence. Our results indicate that 61.5% (8 of 13) of those with a failed Sistrunk had pre-operative thyroglossal cyst infection that lasted more than 6 months. In 92.3% of patients with recurrence, the interval between resolution of infection and time of operation was less than one month. CONCLUSION: Effective treatment of thyroglossal duct cyst infection, at least one month pre-operatively, is warranted to reduce post-operative recurrence rate.

Thyroglossal Duct Cyst Occupying Posterior Hyoid Space with Endolaryngeal Extension Presenting After Neck Trauma.

OBJECTIVE: Thyroglossal duct cysts are the most common congenital neck mass. They typically present as a painless midline neck mass in a child or young adult, but may also present later in adulthood when the cyst becomes symptomatic. Thyroglossal duct cysts are most commonly located inferior to the hyoid bone in close relation with the thyrohyoid membrane. Very rarely, they may extend intralaryngeal, occupy the posterior hyoid space, and present with dysphonia and/or dysphagia. To our knowledge, this is the 24th reported case in the literature. METHODS: Case report with a comprehensive review of the literature. RESULTS: The patient was a 43-year-old male experiencing dysphonia and dysphagia following a motor vehicle accident. He was subsequently found to have a large thyroglossal duct cyst with endolaryngeal extension that was previously asymptomatic and undiagnosed. He underwent successful surgical excision which resulted in resolution of symptoms. CONCLUSION: This is the first reported case of a thyroglossal duct cyst in the posterior hyoid space with endolaryngeal extension being diagnosed following a traumatic event. This case illustrates the need to consider thyroglossal duct cyst in the differential diagnosis when working up a post-traumatic intralaryngeal neck mass. A secondary educational objective in this case is to be diligent to consider and rule out laryngeal fracture in the case of a neck mass presenting after trauma as they can easily be missed and present with many overlapping symptoms.

The imperative of the Sistrunk operation: review of 160 thyroglossal tract remnant operations.

OBJECTIVE: Analysis of the pre- and postoperative features, long-term follow-up, and complications. STUDY DESIGN AND METHODS: Case series with chart review of 160 thyroglossal tract remnant excisions over a 20-year period (1988-2007). RESULTS: The mean age of diagnosis was 10.9 +/- 14.2 years with 63.8 percent male predominance. There was a prior history of thyroglossal tract remnant infections in 70 percent of patients, and 30 percent presented with cutaneous fistulas. The majority had ultrasound imaging that identified cysts mainly (66.7%) in the infrahyoid region. Preoperative fine-needle aspirations in 18 patients were benign. On pathological reevaluation, 26.5 percent had thyroid tissue inside the remnant, with one case of papillary carcinoma. After the Sistrunk operation, postoperative complications occurred in 7.5 percent including a 1.9 percent recurrence rate. CONCLUSIONS: All age groups had similar clinical presentations and outcomes. Ultrasound is a reliable and appropriate imaging modality for most patients. Surgery must effectively incorporate the tract and cyst to allow low recurrence rates (<2%). Clinically diagnosed thyroglossal tract remnant may, in fact, be other pathologies in 10 percent of cases. Nevertheless, our recommendation is a Sistrunk procedure for all midline neck lesions suspected to be a thyroglossal tract remnant.

[Papillary carcinoma in thyroglossal duct cyst]. / Rak brodawkowaty w torbieli srodkowej szyi.

Authors present a rare case of the papillary thyroid carcinoma in thyroglossal duct cyst in a 40 year-old woman. Preoperative assessment (palpation and ultrasonography) showed symptoms of typical thyroglossal duct cyst and the final diagnosis was established after histopathological examination of the surgical specimen. Epidemiology, diagnostic methods and treatment of this rare disease were also discussed in the study.