Intralaryngeal thyroglossal duct cyst: implications for the migratory pathway of the thyroglossal duct

An atypically located thyroglossal duct cyst in a 42-year-old man is described. A purely intralaryngeal thyroglossal duct cyst is extremely rare and can mimic other laryngeal lesions. This case demonstrates that thyroglossal duct cyst is a possible cause of intralaryngeal swellings and would have significant implications for the manner in which they are managed.     

Atypical thyroglossal duct cyst

Thyroglossal duct cysts most frequently present in childhood as a painless midline swelling of the neck. Uncommonly, these embryonal remnant cysts present clinically in adult life. The majority of adults with thyroglossal duct cysts present with a swelling at the level of the thyrohyoid membrane. On rare occasions, a thyroglossal duct cyst can present with more sinister symptoms, such as hoarseness, dysphagia and dyspnoea. On these occasions, the pre-operative clinical and investigative diagnosis remains in doubt until histology is available.     

Atypical thyroglossal duct anomalies

OBJECTIVE: Thyroglossal duct (TD) anomalies are generally considered to be a distinct clinical entity. However occasional cases do not conform to the standard picture and are complex. This study aimed to scrutinise consecutive cases of TD anomaly managed surgically over 7 years at a tertiary teaching hospital and analyse those with atypical features. METHODS: This is a retrospective study of 62 children managed for a clinical diagnosis of TD anomaly from 1998 to 2005. Their medical records were reviewed for clinical presentation, investigations, operative findings, histopathology, management and outcome. After a clinical diagnosis, they had individualized investigations (USG, MRI, radioiodine scan, FNA) prior to the Sistrunk operation. RESULTS: 5/62 cases had atypical features. 2/5 were adolescents with a short history while 3 were below 5 years of age with onset since infancy. 4/5 were females. 1/5 had a Sistrunk operation earlier, 2/5 had a redo surgery prior to cure. 3/5 had a grossly identifiable tract at surgery, one with a midline course and two deviating laterally to open at the left pharyngeal wall. The final histological diagnosis was a mixed thyroglossal-dermoid cyst (case 1), TD anomalies with aberrant pharyngeal communication (cases 2 and 3), isthmic thyroglossal 'cold' cyst (case 4), and a tuberculous cyst (case 5). Postoperatively, Cases 2 and 3 had wound infections that were managed conservatively while case 5 developed an incisional sinus that healed after a month of anti-tuberculous chemotherapy. Cases 1 and 4 had an uneventful recovery. At an average f/u of 2 years, all are asymptomatic and well. CONCLUSIONS: 5-10% of apparent TD anomalies may present with atypical clinicopathologic characteristics. Atypical cases and late presentations may warrant additional investigations to establish the diagnosis and tailor management.     

Vagaries of thyroglossal duct cysts

The thyroglossal duct cyst is the most common congenital cyst found in the neck, classically presenting as a paramidline mass in a healthy young adult. Fifty-three cases managed at the Brigham and Women's Hospital and Boston's Beth Israel Hospital over the past 10 years were reviewed. The unique presentation of papillary adenocarcinoma in TGDCs and findings in the elderly population are discussed. These cases represent both diagnostic and therapeutic challenges. The existing literature is correlated with our data, providing a treatise on management.     

Lingual thyroglossal duct cysts--a review

Objectives

Lingual thyroglossal duct cysts (TGDC) are rare and liable to be missed in a cursory clinical examination. This study aimed to report the details of lingual TGDC from the authors’ series and review existing literature on the entity.

Methods

A 12 year retrospective survey of all cases of thyroglossal duct anomalies managed at a tertiary teaching hospital was conducted to identify those with lingual TGDC. Their clinical presentation, investigations, diagnosis and management were analysed. Case series from anecdotal published English literature were critically reviewed with particular regard to diagnosis and management.

Results

Of 78 cases of thyroglossal duct anomalies, 3 were lingual TGDC. All were females. One neonate presented with feeding difficulty and was clinically misdiagnosed as a ranula. The two older children presented with a cyst at the foramen caecum. The varied imaging and diagnostic dilemma are presented. The older children had cysts abutting the hyoid and were managed with transoral excision and a Sistrunk procedure; the neonate was managed with transoral excision only. This report also reviews the sparse literature and discusses specific issues in their treatment. The differential diagnoses encompass a wide array of developmental and neoplastic entities. Specific anatomic imaging with USG/CT/MRI and functional evaluation with radionuclide thyroid scan are essential investigative modalities. Besides a classical Sistrunk procedure and simple transoral excision, newer less invasive treatment options including marsupialisation and alcohol ablation have been reported.

Conclusions

In conclusion, the diagnosis and management of lingual TGDC needs to be individualised depending on their presentation and anatomic location. The Sistrunk's procedure is ideal for those in close proximity to the hyoid; however complete cyst excision would suffice in the rest.     

Carcinoma in thyroglossal duct remnants

Two cases of carcinoma arising in thyroglossal duct remnants are reported to add to the previously documented 78 cases in the literature. This rare lesion, which occurs is less than 1% of thyroglossal duct abnormalities, pathologically and clinically is similar to primary tumors arising in the thyroid gland. Papillary adenocarcinomas constitute 75-85% of all cases reported and most are confined to the duct remnant itself with metastasis being infrequently recorded. Therapy consists of wide excision of the tumor-bearing tissue (Sistrunk procedure) in those cases localized to the duct remnant itself. In instances where metastasis or additional lesions are detected in the thyroid gland, thyroid ablation and neck dissections are indicated. Additional modalities such as suppressive thyroid therapy, radioactive iodine, and external radiation have been employed. Distant metastasis is rare and the long-term prognosis is very favorable.     

Endolaryngeal extension of thyroglossal duct cyst

Thyroglossal duct cysts are the most common congenital neck masses that develop during childhood, The masses develop from remnants of thyroglossal ducts, and typically appear as midline neck masses. Endolaryngeal extension of thyroglossal duct cysts has been reported mostly as midline neck swelling. We observed a case of extension of the thyroglossal duct cyst to the supraglottic area without neck swelling. A 50-year-old man presented with a 1-month history of foreign-body sensation in the throat. Fiberscopic and radiologic findings were similar to those associated with a saccular cyst, but its proximity to the hyoid bone raised the possibility of thyroglossal duct cyst. Operation was performed via an external incision to completely remove the cyst. Postoperative fiberscopy revealed that the aryepiglottic fold swelling had disappeared. Diagnosis of thyroglossal duct cyst was confirmed on the basis of pathological findings. In cases in which it is difficult to remove the cyst from the hyoid membrane, the hyoid bone midline portion should be dissected. Thyroglossal duct cysts should be considered in cases with a submucosal tumor in the supraglottic region, and radiological examinations should be performed.     

Invasive thyroglossal duct carcinoma in childhood

Thyroglossal duct carcinoma is a very rare tumor. Extracapsular invasion is even less common and can result in possible extension of the tumor into the surrounding musculature without metastatic lymphadenopathy. A case of a 13-year-old adolescent is presented to illustrate this problem. In this case a Sistrunk operation and total thyroidectomy followed by I131 ablation and thyroid hormone suppression was considered the treatment of choice.     

Papillary carcinoma on thyroglossal duct cyst

Papillary carcinoma ansing on a thryroglosal duct cyst is a rare tumor. Since Ucherman described first case in 1915, only 150 cases have been reported. We present clinic evolution and treatment of a new case and review literature and discussion about tumor origin, adequate diagnosis test and treatment. This should include Sistrunk procedure but further surgery on thyroid gland is not accepted by all authors.     

Papillary carcinoma in thyroglossal duct remnants

Summary About 100 cases of carcinoma arising in thyroglossal remnants have been reported in the world literature. Five additional cases were discovered incidentally on histopathological examinations of specimens following Sistrunk's operation for removal of thyroglossal cysts and are now reported. The possibility of preoperative clinical diagnosis and the modalities of treatment are discussed. Offprint requests to: L. Pacheco-Ojeda     

Branched and polycystic thyroglossal duct anomaly

An unusual thyroglossal duct anomaly characterized by a branched tract terminating in two separate cysts is described. The patient presented with a long-standing fistula following previous incomplete excision. Thyroglossal cysts are common congenital anomalies (Moussatos and Baffes, 1963; Allard, 1982), which do not usually present any diagnostic difficulty. However, they may sometimes present with unusual clinico-pathological features. In this report we describe a branching and polycystic thyroglossal duct abnormality occurring in a young woman with a history of previous surgery for a midline cervical mass.     

Cartilage within a thyroglossal duct anomaly

A case of thyroglossal duct anomaly showing an unusual finding of foci of cartilage is presented. Although the concept of choristoma may explain the pathogenesis of cartilage within the present congenital anomaly, the histologic features were more in favor of a metaplastic process. In addition, the history of recurrent bouts of local inflammation supports the latter view.     

甲状舌管残留临床分析

目的　探讨甲状舌管囊肿及瘘的复发因素及治疗情况。方法　回顾性分析我科2010年1月～2013年3月收治的114例甲状舌管囊肿及瘘患者的临床资料。结果　所有病例均经手术治疗,其中113例行Sistrunk术式切除,1例单纯囊肿切除,术后均一期愈合,随访6～46个月,3例复发,其中2例成人再次行Sistrunk扩大根治术,术后未再复发。结论　误诊、非典型的甲状舌管残留、急性感染时行手术、病变组织切除不完全、手术年龄过小和术中囊肿破裂是导致术后复发的重要因素,术前抗感染及术中对舌骨及残端的处理是治疗与防止复发的关键。     

Presentations of thyroglossal duct cysts in adults

The thyroglossal duct cyst is the most common congenital neck mass, resulting from persistence and dilatation of remnants of an epithelial tract formed during migration of the thyroid during embryogenesis. Approximately 7% of the population has thyroglossal duct remnants. Although thyroglossal duct cysts generally present clinically in children, it is important to understand that the lesion can present in adults as well, sometimes much later in life. Incidental carcinoma of the thyroglossal duct cyst is rare, but is more likely to occur in adults than children. Between 1991 and 1998, 11 cases of thyroglossal duct cysts were seen in adult patients at Georgetown University Medical Center, including 2 cases containing papillary carcinoma. This report discusses their clinical presentations and management. Received: 11 September 1998 / Accepted: 23 October 1998     

Endoscope-assisted intra-oral resection of the external thyroglossal duct cyst

Objective

Surgical removal of a thyroglossal duct cyst (TGDC) and its tract is usually accomplished through an external neck incision, including the removal of the middle part of hyoid bone and a block of tissues extending to the foramen cecum. However, this procedure inevitably results in neck scarring. We implemented a modified approach to TGDC removal in a 28-year-old woman through the floor of the mouth using an endoscope system.

Case report

Here, we describe the detailed procedure of the endoscope-assisted intra-oral resection for TGDC. The total operative time was 130 minutes. The patient complained of swelling and pain in the floor of the mouth for 2 days, but did not require any intervention. Follow-up imaging studies confirmed no recurrence (18 months) without any sequelae, and the patient was satisfied with her surgical outcome.

Conclusion

The intra-oral approach through the floor of the mouth is a technically feasible alternative surgical option that allows for complete removal of a TGDC without the neck scar.     

Papillary carcinoma of the thyroglossal duct cyst in childhood

Thyroglossal duct carcinoma is a rare malignancy that is usually diagnosed postoperatively. Approximately 150 cases have been reported in the literature. Eighty-five percent of these were papillary carcinomas. Controversies exist concerning its nature and treatment. In this report, we present an 11-year-old boy with an anterior cervical cystic mass originating in the thyroglossal duct. After a primary Sistrunk procedure, the cyst and tract extending to the foramen caecum at the base of the tongue in continuity with the midportion of the hyoid bone were resected. Histopathologic study demonstrated a papillary carcinoma. After 4 months of follow-up, the patient is asymptomatic without any evidence of recurrence. The clinical and histopathological features and therapeutic options are discussed.