“Extended” Sistrunk procedure in the treatment of recurrent thyroglossal duct cysts: A 10-year experience

Objective

We report a 10-year experience of children with recurrent thyroglossal duct cysts (TGDCs) who have been treated using the “extended” Sistrunk procedure.

Methods

We performed a retrospective review of TGDC surgery from 2004 to 2013. Sistrunk operation was the procedure of choice in all patients. Seven children had TGDC recurrence. All of them underwent “extended” Sistrunk procedure. Follow up ranged from 6 months to 8 years.

Results

There were no gender differences, all recurrences presented within 12 months follow-up in the same location of the primary cyst. Five out of 7 (71%) patients have been treated for preoperative and 2/7 (29%) for postoperative infection at the time of primary surgery. Pathological examination of the surgical specimens showed a single tract in 2 children (29%) and multiple tracts in 5 (71%). We did not observe postoperative complications or further recurrences.

Conclusion

Our experience suggest that recurrent TGDCs are equally common in both sexes, develop in the same location of the primary cyst and recur more commonly after perioperative infections. The “extended” Sistrunk procedure is highly effective and safe in treating recurrent TGDCs also if multiple duct tracts are detected.     

Recurrent thyroglossal duct cysts: a 23-year experience and a new method for management

OBJECTIVES: We present an experience in the management of primary and recurrent thyroglossal duct cysts (TGDCs) and describe a novel method for recurrent TGDC removal. METHODS: We performed a retrospective review of TGDC surgery at Children's Hospital in Seattle from 1980 to 2003. The surgical techniques for primary and recurrent TGDCs and the factors associated with TGDC recurrence were evaluated and analyzed. RESULTS: During the study period, 231 patients underwent 296 TGDC surgeries. Thirty-four of the 231 patients (15%) underwent a total of 88 procedures for recurrent TGDCs. Successful procedures used for secondary TGDC management included central neck dissection with directed base of tongue (BOT) excision in 6 of 9 patients (67%), secondary Sistrunk operation with limited BOT resection in 12 of 27 patients (44%), revision Sistrunk operation with BOT dissection in 7 of 11 patients (64%), and suture-guided transhyoid pharyngotomy in 8 of 8 patients (100%). Ten of the 231 patients (4%) had initial TGDC incision and drainage and then underwent a total of 21 procedures, excluding the incision and drainage. The factors associated with TGDC recurrence were inaccurate initial diagnosis (17 of 34 or 50%), infection (5 of 34 or 15%), unusual TGDC presentation (5 of 34 or 15%), and lack of BOT musculature removal (7 of 34 or 20%). The level of surgeon training affected the surgical outcome. CONCLUSIONS: Successful TGDC treatment requires consideration of factors associated with recurrence. Recurrent TGDCs can be treated by several methods, including suture-guided transhyoid pharyngotomy.     

Doxycycline: A sclerosant agent to treat a case of recurrent thyroglossal duct cyst

Thyroglossal duct cyst (TGDC) is one of the most common congenital anterior midline neck masses in children. Sistrunk operation is considered as the cornerstone in TGDC removal. Nevertheless, 5% to 7 % of patients have been reported to show a recurrence even after adequate resection. This necessitates at times multiple resections with potential complications. One interesting emerging technique is sclerotherapy of TGDC remnants post resection. We present here a case of a child who had a successfully treated recurrent TGDC using sclerotherapy with doxycycline.     

Risk factors for thyroglossal duct remnants after Sistrunk procedure in a pediatric population

To evaluate recurrence after surgery for thyroglossal duct cyst (TDC) we performed a retrospective chart review. Seventy four patients between 0.5 and 8.5 years of age presenting with a midline neck cyst underwent a Sistrunk procedure for a preoperative diagnosis of TDC. Fifty-seven had histologically confirmed TDC (mean age of the population: 4+/-1.5 years, mean follow-up: 6 years and 8 months). Recurrence occurred in 15% of the cases of histologically confirmed TDC. Four individual risk factors have been identified: number of infection before surgery [more than 2 episodes (P<0.05)]; preliminary surgical procedure (P<0.05); age [less than 2 years (P<0.05)] and multicystic lesion on histopathology (P<0.01). The two first factors being correlated, the risk of relapse might be lowered by a wide excision performed before any infection in children over 2 years.     

Thyroglossal duct cysts: presentation and management in children versus adults

OBJECTIVES: To determine if the clinical presentation of thyroglossal duct cysts (TGDC) varies between children and adults and whether this knowledge helps optimize the surgical management. METHODS: We retrospectively identified all patients with TGDC managed in our department between 1992 and 2002. We reviewed the patients' charts and recorded their gender, age at diagnosis, clinical presentation, radiologic imaging, surgical management, post-operative complications, and recurrence rate and compared the variables between the children and adults. RESULTS: Twenty-one children and 41 adults were treated for TGDC. Of the children, 57% were male and 43% were female, whereas 49% of the adults were male and 51% were female (P = 0.53). The average age was 6 +/- 5 years in children and 45 +/- 16 years in adults, which demonstrates a bimodal distribution. Forty-three percent of children and 42% of adults presented with an infected neck mass (P > 0.99). Among our patients, 96% of the adults and 100% of the children underwent a Sistrunk operation. Four children developed a wound infection that resolved with antibiotics. One adult developed a haematoma and another developed a wound seroma. There was one recurrence among adults and one among children, both of whom were treated with a second Sistrunk procedure. CONCLUSIONS: There appears to be a bimodal distribution for age at presentation of TGDC. Since the differential diagnosis among adults is broader, the opportunity for misdiagnosis is greater. However, once the correct diagnosis is made, the surgical management and post-operative outcome between adults and children is the same.     

Endoscopic management of intralingual thyroglossal duct cysts: Case series and systematic review

ObjectiveTo discuss our institutional experience with endoscopic management of intralingual thyroglossal duct cyst (TGDC) and review cases in the published literature in a systematic review.MethodsPediatric patients with intralingual TGDC treated with endoscopic surgery at our institution from 2009-2019 were identified. Metrics from our case series were then compared to those in the literature in a systematic review to assess pooled outcomes of endoscopic or transoral management. Patient demographics, age of presentation, presenting symptomatology, size of cyst on imaging, type of surgery, and post-operative outcomes were assessed.ResultsWe identified 5 institutional cases of intralingual TGDC and 48 cases of intralingual TGDC described in the literature. The average age of presentation was 20.36 months. 69.8% (N=37) of patients presented with at least one respiratory symptom, 22.6% (N=12) presented with dysphagia, 9.4% (N=5) presented with an identified mass in the oropharynx, and 15.1% (N=8) had the cyst discovered as an incidental finding. Three patients required revision surgeries due to prior incomplete TGDC excisions and one patient experienced a recurrence >6 months after primary excision requiring a second procedure. Our data pooled with published case series in systematic review confirms that endoscopic or transoral management are excellent options for definitive management of intralingual TGDC.ConclusionsIntralingual TDGC is a potentially life-threatening variant of TGDC. Our results pooled with published series in a systematic review suggest that endoscopic or transoral management of intralingual TGDC are excellent minimally invasive treatments with a low risk of recurrence. Postoperative surveillance up to one year is recommended.     

The thyroglossal duct cyst and respiratory failure in neonates: Case report and review of an unusual entity

Thyroglossal duct cysts are unusual neck lesions in neonates. Most cysts are first noticed in preschool-aged children as a small midline neck swelling, and can become infected causing inflammation, erythema, and external drainage. In this patient population respiratory symptoms are frequently part of the initial presentation, and respiratory failure due to cyst mass effect is often fatal in newborns. The case presented here is unusual in terms of age at presentation (4 days), type of infecting bacteria (GBS), rapid cyst enlargement, and prominent respiratory symptoms (previously unreported in cysts inferior to hyoid bone). Although rare, TGDCs should be included in the differential diagnosis of congenital neck masses or unexplained respiratory compromise in neonates, especially when the presentation includes positional, intermittent, or progressively worsening obstructive respiratory symptoms. As this case illustrates, infection of these cysts is common but does not always manifest with visible neck inflammation and swelling. With rapid diagnosis the potentially fatal complications of TGDCs can be avoided in neonatal patients.     

Thyroglossal duct cyst: a comparison between children and adults

PURPOSE: The purpose of the study was to evaluate the differences in the clinical characteristics of thyroglossal duct cysts (TGDC) between children and adults and to find a method for optimizing management of TGDC. MATERIALS AND METHODS: This study consisted of a retrospective chart review of patients with a diagnosis of TGDC from 1997 to 2002. All records were reviewed for age and sex, season of first visit, diagnostic methods, sizes and locations of cysts, surgical management, and recurrences. Differences between children and adults were analyzed. RESULTS: A total of 84 patients (32 children, 52 adults) were recruited. There were no significant differences in sex in either group. Compared with children, more adult patients had left-sided and infrahyoid cyst locations. The cyst sizes were significantly larger in adults. In this review, 90.4% of adults and 75% of children underwent a Sistrunk operation, whereas the others underwent cyst excision. There was a total of 5 recurrences, 2 in children and 3 in adults. CONCLUSION: Although the recurrence rates between children and adults and between different surgical managements were not significantly different, a Sistrunk procedure is recommended as the main operation of choice, especially in adults in whom a more extended tract resection should be performed.     

Cricoid injury complicating excision of thyroglossal duct cyst

The Sistrunk procedure is the treatment of choice for thyroglossal duct cyst excision (TGDC) due to its low recurrence rate and incidence of minor complications. Given the proximity of TGDC to the airway, injury to the laryngotracheal cartilages during this procedure is possible. We report a case of a 2-year-old girl transferred to our institution who had a cricoid cartilage injury during a Sistrunk procedure. The pattern of injury, its treatment and outcome in our patient, as well as preventative measures are described. To our knowledge, this is the first report describing injury to the cricoid cartilage following this procedure.     

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.     

First-intention surgical treatment of thyroglossal duct cysts in children: apropos of 99 cases]

INTRODUCTION: Thyroglossal duct cyst (TGDcs) is the most common malformation of the neck. The risk of infection and malignant transformation impose its treatment. OBJECTIVES: The purpose of our study were: 1) to specify some points about the symptomatology and preoperative evaluation necessary for TGDcs diagnosis; 2) to analyse the factors who can explain an unsuccessful surgical treatment. PATIENTS AND METHODS: Our study is based on a retrospective review of cases and a review of the literature. From 1981 to 2000, 99 children with a mean age of five years were treated for a TGDcs with a surgical procedure in the Grenoble University Hospital. 3 excision and 96 Sistrunck's procedure were performed. In all cases a histological study was made. RESULTS: Ultrasonography was the most frequent preoperative evaluation. We have had 7 complications: 3 haematoma, 2 abscess and 2 desunited scar. 6 children have had recurrent disease. Among these 6 children, 3 have had an excision and 3 a Sistrunck's procedure. No case of malignant transformation was reported. CONCLUSION: Ultrasonography is the first preoperative evaluation to obtain before surgical treatment of a TGDcs. Sistrunck's procedure is the best surgical treatment with a value of recurrence from 1% to 10%. Areas of surgical failure included breaking of cyst during the dissection, inadequate hyo?d bone resection, an anatomical variation with many ductuli in the base of tongue, inadequate muscles of tongue resection.     

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.     

A 16-year experience in treating thyroglossal duct cysts with a “conservative” Sistrunk approach

Although Sistrunk operation is the standard method to treat thyroglossal duct cyst, the reported recurrence rates after a “classic” or “modified” Sistrunk procedure still varied from 0 to 15.8 %, indicating the existence of some technical uncertainties. While simple cystectomy has been recognized as the most important prognostic factor predicting thyroglossal duct cyst recurrence, whether other clinico-pathological parameters also affect disease recurrence has not been well studied. We retrospectively reviewed the medical records of all patients who underwent thyroglossal duct cyst surgery between June 1998 and June 2014 at our institution. Among the 180 primary patients, 160 patients received a “conservative” Sistrunk operation, while the remaining 20 patients received simple cystectomy only. Five patients (2.8 %, 5/180) had recurrence. Four of them received simple cystectomy while 1 had “conservative” Sistrunk operation. In univariable analysis, age (p = 0.02), history of previous infection (p = 0.004) and the type of resection (p = 0.001) were significantly correlated with disease recurrence. In multivariable analysis, the type of resection turned out to be the most important factor (p = 0.03) related to recurrence. In the most parsimonious model selected by backward elimination, both history of infection (p = 0.048) and the type of resection (p = 0.02) were important predictors of postoperative recurrence. Our results demonstrated that a “conservative” Sistrunk approach could provide a comparably low recurrence rate (0.6 %, 1/160) in dealing with primary thyroglossal dust cysts. Routine dissection of suprahyoid tissue may not be imperative. Overall, the type of resection and history of infection are the most important predictors of recurrence for thyroglossal duct cyst.     

Sistrunk revisited: a 10-year review of revision thyroglossal duct surgery at Toronto's Hospital for Sick Children

The definitive surgical management of thyroglossal duct cysts (TGDC) was elucidated by Sistrunk in 1920. However, the procedure is often poorly performed. We reviewed the charts of 143 patients managed for TGDC at the Hospital for Sick Children. Toronto, between 1978-1988. These patients underwent 214 surgical procedures at HSC and elsewhere for cure. One hundred and five patients needed only one procedure. Thirty-eight patients had recurrent disease. These needed 109 procedures for cure. Inadequate surgery was the direct cause of recurrence. Areas of surgical failure included misdiagnosis, inadequate hyoid bone resection and persistent infra or suprahyoid tract remnants. Representative case samples are outlined with clinical, surgical and pathologic correlation. We recommend performing the complete, classic Sistrunk procedure for all cases of TGDC.     

Thyroglossal duct cyst and sinuses: A 20-year Los Angeles experience and lessons learned

Background

First described in 1920 and later modified in 1928, the Sistrunk procedure substantially reduced the incidence of recurrence of midline neck cysts compared with a local excision or cystectomy. The purpose of this study was to determine if the rate of recurrence was influenced by performing either a ‘classic’ or a ‘modified’ Sistrunk procedure, if the recurrence rate was influenced by the physician's training, how successful we have been in managing patients with a recurrence? Finally, is outpatient surgery safe for Sistrunk procedures?

Methods

We performed a retrospective review of all patients with a thyroglossal duct remnant (TGDR) who were seen at the Children's Hospital Los Angeles (CHLA) from 1990 to 2010. The following data were collected: patient age, gender, presence or absence of a pre-operative infection, imaging studies, type of procedure performed, the attending surgeon's training background, inpatient or outpatient status, and complications.

Results

A total of 128 patients (61% male, 39% female) met the inclusion criteria. The age ranged from 2 months to 14 years (mean of 5.1 years). A total of 137 procedures were performed; 114 (83.2%) for primary and 23 (16.8%) for secondary disease. Complications included post-operative infection (10.9%), recurrence of disease (6.6%), undesirable scar (5.8%), and fistula (2.9%). Surgeons with fellowship-training in pediatric otolaryngology had a recurrence rate of 4.0% and surgeons with fellowship-training in pediatric surgery or pediatric plastic surgery had a recurrence rate of 30.1%. Twenty patients had a ‘classic’ Sistrunk (14.6%) and 117 (85%) had a ‘modified’ procedure. Patients were admitted after surgery in 78 cases (56.9%) and 59 patients (43.1%) had an outpatient (OPD) procedure.

Conclusions

There is no place for cystectomy in the treatment of TGDR. A ‘modified’ Sistrunk procedure is the procedure of choice in both primary and revision cases. Wide local excision of recurrences is required and a ‘classic’ Sistrunk should be considered. Specific training to gain an intimate knowledge of the anatomy in and around the larynx and experience with multiple cases reduces the incidence of recurrence. Outpatient surgery is safe and effective for selected patients who undergo a Sistrunk procedure.     

Impact of age on surgical outcomes and failure rates in patients with thyroglossal duct cysts

PurposeThyroglossal duct cyst (TGDC) may appear in all age groups. The impact of age on surgical outcome has been reported with conflicting results. The aim of the study was to evaluate different risk factors for surgical success according to stratified age groups.MethodsA single center retrospective study. All patients who underwent a Sistrunk procedure between 2004 and 2018 were enrolled. Data included demographics, pre-operative presentation, intra\postoperative complications and surgical failures. Population groups were divided into adults (≥18 years), older children (OC, 3–18 years) and toddlers (<3 years).ResultsA total of 109 patients were included: 55 adults (50.5%), 36 OC (33%) and 18 toddlers (16.5%). The adult group demonstrated a significant lower rate of surgical failures when compared to the toddler (3.6%, vs. 38.9%, P < 0.001, respectively) and the OC group (3.6%, vs 16.7%, P = 0.032, respectively). A borderline significance was found when comparing surgical failure rates among toddlers and OC (38.9% vs.16.7%, P = 0.07, respectively). Post-operative complications were associated with surgical failures among toddlers (P = 0.045) and OC (P = 0.016), but not adults. Pre-operative infection and admission were significantly associated with failure in the OC group.ConclusionsSurgical failures following Sistrunk procedures and their associated risk factors vary between age groups. Familiarity with these rates and associations can contribute to better decision making in managing TGDC patients.     

Incidental radiographic findings of thyroglossal duct cysts: Prevalence and management

Objectives/HypothesisTo determine the epidemiology of incidental thyroglossal duct cysts (TGDC) discovered on imaging studies obtained in the head and neck area in children and to discuss subsequent management.MethodsA retrospective chart review was performed at Texas Children's Hospital of all computed tomography (CT) and magnetic resonance (MR) imaging studies obtained in the head and neck region between July 2011 and July 2014. Images obtained for the purpose of evaluating a neck or lingual mass were excluded from the study, as were patients with previously known TGDCs. Data including age, sex, location of TGDC, size, presence of symptoms, referral to Otolaryngology, and intervention were recorded.ResultsA total of 60,663 CT and MR studies of the head, brain, sinus, neck, and C-spine during this time period were reviewed; of these 69 (0.1%) cases contained incidental discovery of probable TGDCs with more males (40) than females (29). Ages ranged from 3 days to 17 years old, with the mean age at 5 years. Locations varied, with majority at base of tongue (83%) followed by hyoid (13%) then infrahyoid straps (4%). Sizes ranged from 2 to 28 mm with average size at 8 mm. 11 of these patients were referred to an Otolaryngologist; 9 were asymptomatic and decision was made to observe, the other two subsequently underwent surgical excision secondary to mass effect and dysphagia with histologic confirmation of diagnosis.ConclusionTGDCs commonly present as an anterior neck mass, however the majority of incidentally discovered TGDCs on imaging are located at the base of tongue. Management of these findings should include referral to an Otolaryngologist for further evaluation with the decision to intervene surgically based on development of clinical symptoms.     

Preoperative sonography in presumed thyroglossal duct cysts

OBJECTIVE: To determine the utility of ultrasonography as a sole diagnostic study in the preoperative preparation of patients with presumed thyroglossal duct cysts. DESIGN: Retrospective chart review. SETTINGS: Children's Memorial Hospital, Chicago, Ill. PATIENTS: Forty-five pediatric patients with midline masses. MAIN OUTCOME MEASURE: Accuracy in the determination of a normally positioned thyroid gland excluding the presence of a solitary ectopic thyroid gland. RESULTS: A retrospective chart review was performed at our institution for the period February 1990 to January 1996. A total of 45 patients with midline masses were identified, 39 of whom had undergone preoperative ultrasonography as their sole diagnostic imaging study. In all 39 patients, both a cyst and a normal thyroid gland were identified. All 39 patients underwent the standard Sistrunk procedure. Thirty-seven patients had pathologically confirmed thyroglossal duct cysts. The remaining 2 had dermoid cysts. There were no cases of postoperative hypothyroidism. CONCLUSIONS: The incidence of ectopic thyroid in the diagnosis of thyroglossal duct cysts has been reported to be as high as 1% to 2%. In our surgical and clinical experience, the actual incidence of solitary ectopic thyroid tissue is substantially lower. Nevertheless, to prevent the inadvertent removal of the only functioning thyroid tissue, with resultant postoperative hypothyroidism and possible medicolegal consequences, we advocate the routine preoperative identification of normal thyroid gland. We recommend ultrasound as an accurate, cost-effective, noninvasive imaging modality in the preoperative evaluation of all patients with neck masses suspicious for thyroglossal duct cyst. Also, it does not require sedation.     

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。     

Thyroglossal duct cyst of the tongue

Thyroglossal duct cysts commonly present as midline neck masses with or without infection. The presentation of thyroglossal duct cyst on the tongue is quite uncommon. The present report reviews the embryology of thyroglossal duct cyst development and describes two cases of lingual thyroglossal duct cyst. They presented in an infant of two months of age with feeding problems and also in an older child four years of age with a mass in the base of the tongue. Both patients were managed by marsupialization of the cyst of the tongue. The complications of tongue and mandible splitting or cervical approaches were avoided and satisfactory control was obtained. It is important to distinguish thyroglossal duct cyst of the tongue from other conditions that can affect this region.