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.

Incidental finding of thyroglossal duct cyst in a neonate during endotracheal intubation: a case report.

BACKGROUND: Thyroglossal Duct Cyst (TDC) is a common lesion of the midline neck, originating from an incomplete involution of the thyroglossal duct. It is typically observed in pre-scholar patients and surgery is the treatment of choice to prevent infections. Here reported a case of incidental diagnosis in a newborn patient. CASE PRESENTATION: a 3-week-old male baby was admitted to our hospital for weight loss and projectile vomits after breastfeeding. After a diagnosis of hypertrophic pyloric stenosis, the baby underwent pyloromyotomy. During the endotracheal tube placement, the anesthetist noticed the presence of a midline neck mass. The suspect of TDC was confirmed by an intraoperative ultrasound, so, despite the age of the patient, we proceeded with the excision of the lesion according to Sistrunk's procedure to avoid future complications and anesthesia. CONCLUSIONS: even if TDC is a common lesion of pediatric patients, anecdotical neonatal cases were described in the literature, all of them symptomatic. An accurate physical examination and ultrasound are essential diagnostic tools to distinguish TDC from other middle neck lesions, particularly ectopic thyroidal tissue. Sistrunk's procedure is the most effective surgical approach. When diagnosis is made in a newborn, we suggest postponing surgery, unless the baby requires general anesthesia for other surgical procedures, such as in our case.

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.

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.

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.

Treatment of thyroglossal cyst using Koempel's technique: initial experience. / Tratamiento del quiste tirogloso mediante técnica de Koempel. Experiencia inicial.

INTRODUCTION: In spite of being inaccurate in terms of suprahyoid dissection, Sistrunk's procedure is the gold-standard technique in the treatment of thyroglossal cyst. Since it was first described in 2014, the modifications introduced by Koempel have allowed for a more reproducible suprahyoid approach. We present our initial experience with this technique. METHODS: A retrospective study of patients with thyroglossal cyst undergoing Koempel's technique in our institution from 2021 to 2022 was carried out. Demographic, clinical, and histological data was collected. RESULTS: In the study period, 5 patients -3 girls and 2 boys- underwent surgery. Median age and weight were 5 years (2-6) and 16 kg (14-25), respectively. All patients had suffered from previous infections, with 60% having cutaneous fistulization. In 2 patients, surgery was indicated following Sistrunk's procedure as a result of recurrence. Median operating time was 77 minutes (57-110), with the genioglossal muscle plane being identified in the 5 patients. No immediate complications were recorded, and diagnosis of thyroglossal cyst was histologically confirmed in all cases. One of the formerly recurrent patients had recurrence following surgery, but it was subclinical and incidentally diagnosed at control ultrasonography. The remaining patients had no recurrences after a median 8-month (1-12) follow-up period. CONCLUSIONS: Koempel's technique allows for a safe and reproducible approach of the suprahyoid segment. It is an attractive option in complicated cases as a result of previous infection or recurrence.

INTRODUCCION: La intervención de Sistrunk es el gold-standard en el tratamiento del quiste tirogloso pese a su imprecisión en cuanto a la disección suprahioidea. Tras su descripción en 2014, las modificaciones introducidas por Koempel, han permitido un abordaje suprahioideo más reproducible. Presentamos nuestra experiencia inicial con esta técnica. METODOS: Estudio retrospectivo de los pacientes con quiste tirogloso intervenidos mediante técnica de Koempel en nuestro centro en el periodo 2021-2022, recogiéndose datos demográficos, clínicos e histológicos. RESULTADOS: Durante el periodo de estudio se intervinieron 5 pacientes (3 mujeres/2 varones), con una mediana de edad y peso de 5 años (2-6) y 16 kg (14-25) respectivamente. Todos los casos habían sufrido infecciones previas presentando fistulización cutánea el 60%. En 2 de los pacientes se indicó la cirugía por recidiva tras intervención de Sistrunk. La mediana del tiempo quirúrgico fue de 77minutos (57-110) identificándose el plano del músculo geniogloso en los 5 pacientes. No hubo complicaciones inmediatas y el diagnóstico de quiste tirogloso se confirmó histológicamente en todos los casos. Uno de los pacientes del grupo con recidiva previa, presentó recidiva tras la intervención, siendo esta subclínica y diagnosticada incidentalmente tras ecografía de control. El resto de los pacientes no presentó ninguna recurrencia tras un seguimiento mediana de 8 meses (1-12). CONCLUSIONES: La técnica de Koempel permite un abordaje seguro y reproducible del segmento suprahioideo siendo una opción atractiva en casos complicados por infección o recidiva previa.

Thyroglossal Duct Cyst Infection Caused by Neisseria gonorrhoeae: An Unusual Complication of Pharyngeal Gonorrhea.

ABSTRACT: Neisseria gonorrhoeae is a human obligate pathogen whose clinical expression of disease ranges from localized genital infection to involvement of extragenital sites such as the conjunctiva and throat. We describe the second case of a thyroglossal duct abscess due to N. gonorrhoeae, an uncommon complication of pharyngeal gonococcal infection. The fortuitous occurrence in the same individual of these 2 conditions that both exhibit an occult clinical presentation likely accounts for rarity of this infection. We discuss the pertinent gonococcal and host factors that underlie the clinical manifestations of this infection. A particular focus is the fundamental role that the binding of the gonococcal opacity-associated protein to the ubiquitous human carcinoembryonic cell adhesion molecule plays in the pathogenesis of pharyngeal gonorrhea.

Cáncer Papilar en Quiste Tirogloso. Caso Clínico y Revisión de la literatura / Papillary Cancer in Tirogloso Cyst. Case report and review of literature

INTRODUCCIÓN: Los quistes tiroglosos son las lesiones más comunes de la línea media cervical y se ha descrito el carcinoma papilar de tiroides en el 1%. Debido a su baja incidencia no existe un consenso acerca del tratamiento óptimo. Caso clínico: Paciente mujer de 34 años de edad consulta por aumento de volumen cervical doloroso y se evidencia nódulo doloroso en región cervical media. Ecografía de tiroides visualiza una lesión quística compleja. Se completa el estudio con tomografía computada del cuello con contraste que evidencia quiste del conducto tirogloso con compromiso inflamatorio-infeccioso, por lo que se decide cirugía. Biopsia evidencia cáncer papilar de 0.25 cm en quiste del conducto tirogloso, con bordes quirúrgicos negativos. Por bajo riesgo se decide control imagenológico estricto. DISCUSIÓN: Los quistes del conducto tirogloso comprenden las lesiones cervicales congénitas más frecuentes. Se presentan como masas indolentes y asintomáticas. El diagnóstico es confirmado mediante ecografía y la tomografía es utilizada para ampliar el estudio. La aparición de cáncer tiroideo en estos quistes es poco común, y generalmente son indistinguibles de las lesiones benignas en el preoperatorio. En relación al manejo del cáncer papilar en quiste del conducto tirogloso no existe un consenso de su tratamiento óptimo. Para los casos de bajo riesgo se sugiere control anual con TSH y ecografía tiroidea. Para aquellos pacientes de alto riesgo se sugiere tiroidectomía total y ablación de los restos tiroides con yodo radioactivo, con control anual con niveles de tiroglobulina. El pronóstico es excelente, con tasas de remisión que superan el 95%. CONCLUSIONES: Los carcinomas en quistes de conducto tirogloso son poco comunes y en la mayoría de los casos son lesiones diagnosticadas de manera incidental después de la resección quirúrgica. Para definir necesidad de tiroidectomía, debe realizarse estudio individualizado por un equipo multidisciplinario con amplia experiencia.

INTRODUCTION: Thyroglossal cysts are the most common affection of the cervical midline. Papillary carcinoma has been described in 1% of this cysts. Due to its low incidence a consensus on the optimal treatment does not exist. Clinical case: A 34 year old female with no relevant past medical history, presented with a painful cervical mass of many weeks of appearance. The thyroid ecography showed a complex cystic lesion and the cervical computed tomography with contrast evidenced a cyst of the thyroglossal duct with inflammatory and infectious findings. Surgery with no incidents was performed. Biopsy reported a 0.25 cm papillary cancer in the thyroglossal duct cyst, with negativa surgical margins. Strict follow up with imaging studies was decided. DISCUSSION: the thyroglossal duct cyst are the most common congenital cervical affections. Classically, they present as indolente, asyntomatic masses on the cervical midline. The diagnosis is confirmed with ecography and computed tomography is used to extent evaluation. Thyroid cancer in thyroglossal duct cyst is uncommon and generally indistinguishable from benign lesions in the preoperative phase. A consensus regarding the optimal management of this patients does not exist. For low risk cases, an anual control with THS and thyroid ecography is suggested. For patients with high risk a Sistrunk with total thyroidectomy and radioactive ablation of thyroids remnants is recommend. Follow up with anual thyroglubin levels should be performed. The prognostic is excellent, with more than 95% remission rates. CONCLUSSIONS: Thyroglossal duct cyst carcinomas are rare. In most cases, diagnosis is made incidentally after surgical resection. To decide wheter thyroidectomy is necessary each case should be analyzed individually by a multidisciplinary team with vast experience.

Papillary Thyroid Carcinoma within Thyroglossal Duct Cyst: Case Series and Literature Review

Abstract Introduction Thyroglossal duct cyst (TGDC) is themost common congenital anomaly of the neck, and approximately 7% of all the adult population presents it. Ectopic thyroid tissue is found in the thyroglossal duct cyst wall in up to 65% of cases. This thyroid tissue has the potential to develop some type ofmalignancy, themost common of which is the papillary carcinoma of the thyroid. There are just over 270 cases of thyroglossal duct cyst malignancy reported in the literature. Objectives We aimed to study our population of patients in order to identify cases with thyroglossal duct cyst malignancy. Methods A retrospective chart review was conducted in the section of otolaryngology/ head and neck surgery at a hospital in Karachi, Pakistan, from January of 2004 to December of 2014 on patients with the diagnosis of thyroglossal duct cyst. Results Fifty-eight patients were diagnosed with TGDC, two of whom also presented with thyroglossal duct cyst carcinoma. Both patients revealed papillary thyroid carcinoma on histopathology. Case 1 had an open biopsy before undergoing definitive surgery. Both patients underwent subsequent total thyroidectomy after Sistrunk procedure, and case 2 had selective neck dissection revealing lymph node metastasis. Conclusion Thyroglossal duct cyst carcinoma is a rare finding that comes as a surprise to both the patient and the surgeon. We report 2 out of 58 cases diagnosed with thyroglossal duct cyst carcinoma.

Experiencia en el manejo de la patología del conducto tirogloso: serie de casos / Experience in thyroglossal duct pathology: clinical case series

La patología del conducto tirogloso constituye la segunda anomalía cervical benigna más frecuente en la infancia. El diagnóstico es principalmente clínico. Sistrunk fue quien describió la técnica quirúrgica considerada de elección para el tratamiento definitivo de esta patología. Estudio descriptivo retrospectivo. Entre junio de 2008 y agosto de 2015 se realizaron 54 procedimientos en 45 pacientes (39 casos primarios). La mediana de edad fue de 4,7 años; 14/39 pacientes (31,1%) presentaron infección preoperatoria. Todos los pacientes fueron evaluados inicialmente por ecografía. Todos fueron tratados mediante la técnica de Sistrunk. La recidiva global fue de 17,8%.

The thyroglossal duct cyst pathology represents the second cause of bening cervical anomalies in childhood. Diagnosis is mainly clinical. Sistrunk (1920) proposed a surgical technique that is still considered the gold standard for definitive treatment of this condition. A retrospective study was made including patients who underwent surgery for thyroglossal duct cyst pathology in our department between June 2008 and August 2015. In this period, we performed 54 procedures in 45 patients (39 primary cases). Median age was 4.7 years; 14/39 patients (31.1%) had pre-operative infection. All patients were studied with neck ultrasound. A Sistrunk's procedure was performed in all cases. The global recurrence rate was 17.8% (8/45).

Carcinoma papilar de tiroides en quiste del conducto tirogloso / Thyroglossal duct cyst with papillary carcinoma

Introduction: In about 1-2 percent of cases of thyroglossal cyst may be neoplastic changes, mostly correspond to papillary thyroid carcinoma (75-85 percent). The aim of this paper is to present 9 cases of papillary thyroid carcinoma in thyroglossal duct cyst. Materials and Methods: Data were recorded retrospectively from patients diagnosed with papillary thyroid carcinoma in thyroglossal duct cyst treated at Hospital Clínico Universidad de Chile between 1999 2014. Results: From 142 cases operated for thyroglossal duct cyst, 9 cases of papillary cancer (6.34 percent) were recorded. The average age was 32 years. The average diameter of the lesion was 4.4 cm (SD 2.2 cm). 8 patients underwent total thyroidectomy; a simultaneous thyroid cancer was diagnosed in 3 of them. In 6 cases was added iodine therapy. In only one patient a lymph nodal dissection was performed. We do not observe any surgical complication. A solid component in preoperative ultrasonographic study is suspicious of malignancy. The average follow-up time was 85 months. There is no recurrence or mortality in this group of patients. Conclusions: Although the safest long term management is Sistrunk surgery associated with thyroidectomy and radioiodine in selected cases, these patients must be evaluated by a multidisciplinary group and thyroidectomy should be considered in high surgical volume center, in order to minimize complications.

Introducción: En alrededor de 1-2 por ciento de los casos de quiste tirogloso pueden existir cambios neoplásicos, en su mayoría corresponden a carcinoma papilar de tiroides (75-85 por ciento). El objetivo de este trabajo es presentar 9 casos de carcinoma papilar de tiroides en quiste del conducto tirogloso, en cuanto a su forma de presentación y manejo. Material y Método: Se registraron en forma retrospectiva datos de pacientes condiagnóstico de carcinoma papilar de tiroides en quiste del conducto tirogloso atendidos en el Hospital Clínico de la Universidad de Chile entre 1999 2014. Resultados: De 142 casos de pacientes operados por quiste del conducto tirogloso, se registraron 9 casos de cáncer papilar (6,34 por ciento). El promedio de edad de los pacientes fue de 32 años. El diámetro promedio de la lesión fue de 4,4 cm (DS 2,2 cm). Del total, 8 pacientes fueron sometidos a tiroidectomía total, se diagnosticó cáncer de tiroides en 3 de ellos, en 6 se asoció tratamiento con radioyodo. En sólo 1 paciente se realizó una disección linfonodal. El tiempo promedio de seguimiento fue de 85 meses; a la fecha la serie no ha presentado recurrencia ni mortalidad. Conclusiones: Si bien el manejo más seguro a largo plazo es la cirugía de Sistrunk, asociado a una tiroidectomía y eventual radioyodo, la resolución quirúrgica con tiroidectomía asociada debe ser considerada cuando la morbilidad no sea mayor que el beneficio teórico.

Carcinoma papilar de quiste de conducto tirogloso sincrónico con carcinoma papilar de tiroides / Synchronous papillary carcinoma of thyroglossal duct cyst and papillary thyroid carcinoma

Presentamos el caso de un paciente diagnosticado de quiste de conducto tirogloso y un nódulo tiroideo ístmico en el que se evidenciaron focos de carcinoma papilar intraquístico concomitantes con carcinoma papilar en el nódulo tiroideo. La indicación terapéutica en estos casos es controvertida, y dependerá en la mayoría de los casos del estudio microscópico de la muestra. En nuestro caso se realizó en un primer tiempo la intervención de Sistrunk asociada a una istmectomía y tras conocer el resultado de ambas muestras se decidió realizar tiroidectomía total.

We report the case of a patient with a thyroglossal duct cyst and a nodule at the thyroid isthmus which showed intracystic papillary carcinoma foci concomitant with papillary carcinoma in the thyroid nodule. The therapeutic indication in these cases is controversial. In our case it was done in a first intervention a Sistrunk technique associated to an isthmectomy and after knowing the results of both samples was decided to perform a total thyroidectomy.

Carcinoma papilar de tiroides en quiste tirogloso / Papillary carcinoma in thyroglossal duct cyst

Background: to report a rare case of papillary carcinoma in thyroglossal duct cyst and discuss its management. Material and Method: a 72 year-old man was clinically diagnosed to have a big thyroglossal duct cyst. Sistrunk's procedure followed by total thyroidectomy on the evidence of papillary cancer in thyroglossal duct cyst. Results: histopathologic examination revealed a papillary carcinoma in the thyroglossal duct cyst and normallity in the thyroid gland. The patient remained free of disease on follow-up. Conclusion: most cancers arising in thyroglossal duct cyst are of low risk, and Sistrunk's procedure with a total thyroidectomy is an adequate treatment for such cancers.

Objetivo: reportar un caso raro de carcinoma papilar en un quiste tirogloso y discutir su manejo. Paciente y Método: un hombre de 72 años a quien se realizó un diagnóstico clínico de un quiste tirogloso, fue llevado a una cirugía de Sistrunk's y luego una tiroidectomía total ante la evidencia de cáncer papilar en el quiste tirogloso. Resultados: el examen histopatológico reveló un carcinoma papilar de tiroides en el quiste tirogloso y la glándula tiroides fue normal, el paciente permaneció libre de enfermedad en su seguimiento. Conclusiones: la mayoría de los cánceres que surgen en quiste del conducto tirogloso son de bajo riesgo, y el procedimiento Sistrunk's con una tiroidectomía total es un tratamiento adecuado para este tipo de cáncer.

Carcinoma papilar en quiste tirogloso: reporte de 4 casos y revisión de la literatura / Papillary carcinoma in a thyroglossal duct cyst: report of 4 cases and review of literature

Introducción: El quiste tirogloso es una de las masas cervicales más frecuentes. En 1 por ciento puede desarrollar un cáncer, siendo el más frecuente el carcinoma papilar tiroideo. Objetivo: Describir la presentación clínica, el diagnóstico, el manejo y la evolución de los casos de carcinoma papilar tiroideo en quiste tirogloso. Material y método: Se revisaron los archivos del Departamento de Patología de la Universidad de Concepción desde 2000 a 2010 analizando las fichas clínicas y el informe de la biopsia. Resultados: Se encontraron 4 casos de carcinoma papilar en quiste tirogloso, todos de sexo femenino con un promedio de edad de 42 años. Todos fueron sometidos a la operación de Sistrunk. Dos casos fueron sometidos a tiroidectomía total y terapia con radioyodo complementaria. Sólo un paciente presentaba un cáncer sincrónico en la tiroides. El seguimiento promedio fue de 4,7 años y no se presentaron recidivas. Conclusiones: Se encontraron 4 casos, se analiza su presentación, diagnóstico, tratamiento y evolución.

Introduction: Thyroglossal duct cyst is one the most frequent cervical masses. Cancer may develop from 1 percent among them, thyroid papillary carcinoma being the most frequent. Aim: To describe the clinical presentation, diagnosis, management and evolution of thyroid papillary carcinoma cases in thyroglossal duct cyst. Material and method: A review was made of the files from the department of pathology of the Universidad de Concepción, since 2000 to 2010, analyzing clinical records and biopsy reports. Results: Four cases of papillary carcinoma in a thyroglossal duct cyst were found, all female with a mean of age of 42 years. All of them underwent Sistrunk procedure. Two cases underwent total thyroidectomy and complementary radioiodine therapy. Only one patient presented synchronic thyroideal cancer. The mean time of follow up consisted of 4,7 years, with no reported recurrence. Conclusions: We found 4 cases, his presentation, diagnosis, treatment and evolution was analyzed.

Carcinoma en quiste tirogloso / Carcinoma in thyroglosal duct cyst

El carcinoma diferenciado de tiroides en quiste tirogloso (CaQT) es una rara entidad. En diferentes series de pacientes operados por quistes tiroglosos su incidencia fue del 0.7 al 1.07%. Luego de la extirpación del quiste por el procedimiento de Sistrunk, no hay consenso sobre la indicación de tiroidectomía total, radioablación y/o terapéutica supresiva con levotiroxina. El objetivo del Departamento de tiroides de SAEM, fue evaluar: formas de presentación, evolución clínica, métodos diagnósticos de utilidad y tratamiento para consensuar futuras conductas. Material y Métodos: Estudio multicéntrico, retrospectivo en 22 pacientes entre 10 a 69 años, 15 mujeres y 7 varones. Resultados: El tamaño de los quistes osciló entre 1 y 8 cm (Mediana= 3.0 cm, Χ ± DS= 3.7 ± 2.2 cm). La mitad de los pacientes presentó crecimiento del quiste en los 6 meses previos a la cirugía. La punción resultó sospechosa en 2/5 quistes y positiva en uno. La ecografía tiroidea evidenció nódulos en 4/13 casos (30%). Se realizó tiroidectomía en 17/22 pacientes (total: 15 y subtotal: 2). La histología del CaQT demostró carcinoma papilar en 21 y carcinoma folicular en uno. Hubo coexistencia de cáncer intratiroideo en el 23.5% de los casos, ninguno multicéntrico. Dos pacientes presentaron metástasis ganglionares y otro tuvo compromiso muscular (ninguno de ellos coexistió con cáncer intratiroideo). Se radioablacionó a 13 pacientes. En 9/11 pacientes la tiroglobulina permaneció indetectable durante el seguimiento (1 a 14 años). Conclusiones: 1) Realizar ecografía de cuello y punción ecoguiada a todo paciente con quiste tirogloso. 2) En caso de CaQT combinar simultáneamente tiroidectomía total y procedimiento de Sistrunk. 3) Evaluar radioablación complementaria y tratamiento supresivo con levotiroxina en cada caso. 4) Efectuar el seguimiento tal como en los carcinomas ortotópicos.

Differentiated thyroid carcinoma (DTC) in thyroglosal duct cyst (TGDC) is rare, ranging from 0.7 to 1.07% in different series. After the surgery of choice (Sistrunk procedure) the other alternative treatments such as thyroidectomy (Tx), radioiodine and L-T4 therapy are controversial. OBJECTIVE: to evaluate several and controversial aspects in the largest series of DTC in TGDC reported in the literature. Subjects and methods: retrospective multicentric study: n= 22, aged 10-69 yrs. (15 females and 7 men) who underwent the Sistrunk procedure for TGDC. Results: none of the TGDC was less than 1 cm (median 3.0 cm, Χ±SD= 3.7 ± 2.2cm). In half of them there was an increased cystic size in the last 6 months before surgery. Cyst FNA was suspicious in 2/5 and positive in one, whereas the histological diagnosis of the operated TGCD was papillary cancer in 21 and 1 follicular carcinoma. Thyroid ultrasound (US) (n=13) showed nodules in 30% of the cases. Tx was performed in 17/22 (total: 15, subtotal: 2). Thyroid DTC coexisted in 4/17 (23.5%), and was unilateral in all of them. Lymph node metastases were present in 2 adults and muscle involvement was found in the 10-year old girl. None of these 3 patients had overt thyroid lesions. 131-I therapy was performed in 10 patients. In 9 out of 11 subjects Tg remained undetectable during follow-up (1-14yrs.). Persistent high Tg was present in one case without thyroid DTC. Conclusions: 1) Ultrasonography and FNAB should be performed to every patient with thyroglossal duct cyst 2) In case of TGDC, total Tx and Sistrunk's procedure should be simultaneously combined 3) 131-I therapy and L-T4 suppressive treatment should be evaluated in every case 4) Follow-up as in the DTC.

Aggressive form of thyroglossal duct cyst carcinoma: role of ablative dose of Iodine-131

Ectopic thyroid tissue carcinoma is very rare and has usually good prognosis. It could arise in 1 percent of thyroglossal duct cysts (TDC), the most common nonodontogenic cysts that occur in the neck, which results from a failure in obliterating the embryogenic duct produced during thyroid migration. TDC is most often diagnosed during the childhood but may be discovered later in adult age. In most of the cases reported in the literature, thyroid carcinoma arising in the TDC is limited to the cyst without local extension and its efficient treatment consist of the surgical removal of the cyst by Sistrunk's operation. However, some controversies remain regarding the indication of total thyroidectomy when thyroid investigations are normal. Cases of aggressive thyroid carcinomas of the TCD with metastatic cervical lymph nodes are exceptional, mainly when histological findings of the thyroid gland are normal. We report a case of an aggressive form of a thyroglossal duct cyst carcinoma complicated with several infiltrated cervical lymph nodes but normal thyroid gland. The therapeutic strategy adopted in this case shows the decisive role of the post-operative ablative dose of Iodine-131 both for treatment and staging of aggressive form of thyroid carcinoma arising in thyroglossal duct cyst.

Carcinoma papilar en quiste del conducto tirogloso: reporte de dos casos / Papillary carcinoma in a thyroglossal duct cyst: report of two cases

Thyroglossal duct cyst is the most common congenital anomaly of thyroid gland development. However, papillary carcinoma is described only in 1-2 percent of cases and the clinical appearance is indistinguishable from a benign thyroglossal duct cyst. We report two females aged 15 and 27years consulting for a cervical mass. In both, a solid cystic lesion was found and excised using the Sistrunk procedure. The biopsy disclosed a papillary carcinoma in both. The postoperative evolution of both patients was uneventful.