Zhang et al.: Lingual Thyroglossal Duct Cyst with Recurrence after Cystectomy

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1 The Laryngoscope VC 2011 The American Laryngological, Rhinological and Otological Society, Inc. Lingual Thyroglossal Duct Cyst with Recurrence After Cystectomy or Marsupialization Under Endoscopy: Diagnosis and Modified Sistrunk Surgery Li-Chun Zhang, MD; Tian-Yu Zhang, MD, PhD; Yan Sha, MD; Yue-xin Lin, MD; Qi Chen, MD Objective: To explore the radiologic characteristics of lingual thyroglossal duct cyst relevant to diagnosis. In addition, to probe the feasibility of modified Sistrunk surgery for patients who experience recurrence after cystectomy or marsupialization under endoscopy. Study Design: Retrospective. Methods: Seven patients, three male and four female, who had been diagnosed with lingual thyroglossal duct cysts, had received cystectomy or marsupialization under endoscopy between one and eight times with cyst recurrence soon after every operation. All of these cases were evaluated by CT before proceeding with the latest modified Sistrunk surgery. During surgery, we routinely used methylene blue to trace the cysts, and then we used an enlarged Sistrunk procedure for such cases. Results: The CT images demonstrated that there were cysts located posteriorly to the tongue that had a close relationship with the hyoid bone and the foramen cecum. In addition, the results of the pathological examinations were consistent with thyroglossal duct cysts. During the postoperative follow up of 18 to 31 months, the patients did not show any symptoms or signs of recurrence. The results of their most recent examinations, two by laryngoscopy and five by CT scan, did not reveal any abnormality in their pars laryngea pharyngis. Conclusions: CT could differentiate lingual thyroglossal duct cysts from the other cysts occurring at the lingual root. For those patients with lingual thyroglossal duct cysts that recur after cystectomy or marsupialization under endoscopy, the modified Sistrunk operation could be a suitable treatment option. Key Words: Lingual thyroglossal duct cyst, recurrence, cystectomy, marsupialization, Sistrunk surgery. Level of Evidence: 4. Laryngoscope, 121: , 2011 From the Departments of Otorhinolaryngology (L.-C.Z., T.-Y.Z., Y.X.L., Q.C.); and Radiology (Y.S.), Shanghai Eye, Ear, Nose & Throat Hospital, Fudan University (formerly Shanghai Medical University), Shanghai, People s Republic of China. Editor s Note: This Manuscript was accepted for publication May 3, The authors have no financial disclosures for this article. The authors has not conflicts of intersts to disclose. Send correspondence to Tian-Yu Zhang, Department of Otorhinolaryngology, Shanghai Eye, Ear, Nose & Throat Hospital, Fudan University (formerly Shanghai Medical University), 83 Fenyang Rd, Xuhui Disctrict, Shanghai, P.R. China. ty.zhang2006@yahoo. com.cn DOI: /lary INTRODUCTION Thyroglossal duct cysts (TDCs), which originate from persisting embryogenic epithelial tissues, are often considered to be one of the most common congenital entities. Most cases are found in pediatric patients, whereas some are detected in adulthood. 1 Lin et al. 2 once reported the frequently detected cases are from children less than 5 years old, as well as adults ranging from 21 to 30 and 41 to 50 years old. The typical patient presents with a mobile, painless mass in the anterior midline of the neck, whereas in some cases, a fistula is present. 3 When the cyst becomes infected, the healing process is difficult. Currently, the primary treatment for TDC is the removal of the mass by the Sistrunk surgery. However, if there some of the abnormal tissue remains, even a tiny amount of epithelial tissue, the cyst will recur soon after the operation. Therefore, it is very important to correctly diagnose these cases to determine the precise locations and ranges of the masses before surgery. It is not difficult for us to diagnose a typical TDC and to determine its location and range; however, if the mass is atypical, such as a lingual cyst, it is likely to be misdiagnosed. 4 8 In this study, we analyzed seven such cases. These cases had been misdiagnosed as epiglottic or lingual cysts and experienced immediate recurrence of the masses after complete excision or marsupialization under endoscopy. We wished to gain a better understanding of the characteristics of lingual TDCs. In addition, we summarize our own personal experiences with the modified Sistrunk surgery for the treatment of lingual TDCs that recur after surgery under endoscopy. MATERIALS AND METHODS Patients From September 2008 to January 2010, we retrospectively analyzed seven patients who were confirmed as having lingual TDCs by us. Among them, three were male and four were female. The patients were 4 to 52 years old, with a median age of 24 years old. All cases had undergone cystectomy or marsupialization under endoscopy between 1 and 8 times before the 1888

latest surgery, with a total of 21 times and median of 3 times. The interval between each surgery and the following recurrence ranged from 20 to 90 days, with a median of 60 days.

2 latest surgery, with a total of 21 times and median of 3 times. The interval between each surgery and the following recurrence ranged from 20 to 90 days, with a median of 60 days. Of these seven cases, five had been misdiagnosed as epiglottis cysts, and the remaining two cases had been diagnosed as cysts of the lingual root. Examination Before the modified Sistrunk surgery, all seven patients were examined using a Siemens Sensation 10 CT (Forchheim, Germany). Transverse CT scans were acquired in the routine mode with 120 kv, 240 ma from above the hard palate to the inferior border of the thyroid gland. The images had a serial 1- mm thickness images with a 1-mm intersection gap, a field of the view mm, and a acquisition matrix. Of the seven cases, five cases received intravenous iopamidol, with total volume of 75 ml and an injection speed of 1.5 ml/sec, during the examination. All images were imported into the 3D reconstructive type in the vendor workstation (Wizard; Siemens, Forchheim, Germany) after examination to reconstruct the sagittal images for the observation of the relationship between the cysts and the hyoid bone as well as the foramen cecum. Surgical Procedure To facilitate the tracheal intubation and to locate the cyst intraoperatively, we first removed all of the cyst fluid using a needle under the direct laryngoscopy and then injected methylene blue (3 5 ml) into the capsule after the administration of routine intravenous and inhaled anesthesia. After that, we cleared the pharyngeal residue or spillage of methylene blue and performed the endotracheal intubation. When all of these steps were complete, we did the conventionally disinfection and covered the area with surgical towels and then incised the skin on the midline of the anterior neck at the level of the hyoid bone. After the exposure of surgical field, we isolated the hyoid bone, cut off its middle part, upwardly segregated the whole thyroglossal duct to the cyst, and then excised the entire cyst with middle part of the hyoid bone and the mucosa attached. After the operation, we used shame-type sutures for the mucosal stump and the pharyngeal submucosal tissues and layered sutures for the tongue muscle and the subcutaneous tissue. After that, we placed a negative pressure drainage tube in the surgical site and did the dressed the incision without compression. After surgery, the patient was fed nasally and observed for 1 day in the ICU. We prescribed inhalation therapy with antibiotics. During surgery we found that the cysts originated from the hyoid bone and formed a close relationship with the hyoid bone. The examination of the biopsy specimen after surgery demonstrated that all of the cases were coincident with TDC. RESULTS Clinical Manifestation Among all of the patients, five cases complained of pharyngeal foreign body sensation, one case complained of symptoms of upper respiratory airway obstruction such as sudden laryngeal stridor and vague pronunciation after a cold, and one case reported snoring after a cold, as observed by his parents. All cases demonstrated prominent cystic masses near the foramen cecum during indirect laryngoscope examination. Of the seven cases, four cases had an epiglottis that was pushed backward (Fig. 1). They all had smaller cysts and experienced Fig. 1. Endoscope image showing a cyst located to the right of the midline in the posterior of the tongue (asterisk). relief of the symptoms after the aspiration of the cyst fluid (Table I). CT Examination Of the seven patients, four underwent an CT scan, two underwent an un CT scan, and one underwent both an un and an CT scan. The images of all cases showed cystic masses, with the CT value ranging from 19 to 35 HU. The images of the five cases in which CT scan was used showed that the cystic walls were but that the cysts themselves were un after the contrast agent injection. Among the seven cases, three were unicystic, and the remaining four were multicystic, with internal separations; these separations were also after the injection of the contrast agent. On the axial images, we found that the cysts were mainly located at the posterior one-third of the tongue and protruding the surface of the tongue, which both have a close relationship with the foramen cecum. On their middle sagittal images, we found that all the cysts formed a close relationship with the hyoid bone, and in four cases, the epiglottis pushed to the posterior. All if the cases pharyngolaryngeal cavities diminished clearly (Figures 2 3). Follow-up After Surgery All seven patients accepted and underwent the modified Sistrunk surgery, with the removal of the cysts and their covering mucosa, their thyroglossal duct, and the middle parts of the hyoid bones. All of the removed cysts contained methylene blue, which is indicative of the close connection with the hyoid bones. Among all of the cases, there was one patient whose cystic root originated from the lateral surface of the hyoid bone. None of the cases has experienced recurrent symptoms, with normal respiratory sounds, pronunciation, and the absence of pharyngeal foreign body sensation during the postoperative follow-up period of months (Fig. 4). The results of the pathology examination for all cases showed the cystic wall enclosing the respiratory epithelium, which is consistent with TDC. 1889

Foreign body sensation 4 F 21 Foreign body sensation 5* F 23 Foreign body sensation Epiglottic cyst 5 Unicystic Pushed posterior Un Epiglottic cyst 1 Unicystic Pushed posterior Cystic wall Epiglottic

3 TABLE I. The Patients Clinical Histories and CT Examination Results. No. Gender Age Complaint Misdiagnosis Previous Surgical Interventions CT Characteristics Type Epiglottis Enhanced 1 F 4 Vague pronunciation and dyspnea after cold 2* M 25 Foreign body sensation 3 M 30 Foreign body sensation 4 F 21 Foreign body sensation 5* F 23 Foreign body sensation Epiglottic cyst 5 Unicystic Pushed posterior Un Epiglottic cyst 1 Unicystic Pushed posterior Cystic wall Epiglottic cyst 4 Multicystic Pushed posterior Cystic wall and separation Cyst of lingual root 3 Unicystic Unclear Un Epiglottic cyst 2 Multicystic Pushed posterior Cystic wall and separation 6 M 12 Snoring after cold Cyst of lingual root 8 Multicystic Slightly pushed posterior 7 F 52 Foreign body sensation Cystic wall and separation Epiglottic cyst 1 Multicystic Pushed posterior Cystic wall and separation Gender: F, female; M, male. All cysts were located at the posterior of the tongue and formed a close relationship with the hyoid bone and the foramen cecum. *Patient had previously undergone marsupialization under endoscopy. DISCUSSION The thyroglossal duct is the path of the gradual decline of the thyroid primordium during embryonic development. This structure usually originates from the foramen cecum and terminates at the thyroid isthmus. 9 Commonly, the tube is gradually degraded and disappears during development. A TDC can form in anywhere in this path if the embryogenic epithelial tissue remains during this process. The research done by Allard et al. 9 demonstrated that TDC is most frequently detected between the hyoid bone and thyroid cartilage (60.9%), above the hyoid bone (24.1%), at the suprasternal fossa (12.9%) or in the tongue (2.1%). For those uncommon cases with TDC in the tongue, many were misdiagnosed before the surgery; 10,11 some cases were even diagnosed by autopsy In this study, five patients were initially diagnosed with epiglottis cysts at the beginning and underwent cyst resection or marsupialization under endoscopy many times, but the cysts recurred soon after surgery. The remaining two cases were misdiagnosed as cysts of the lingual root; these cysts also recurred after surgery. The main reason for misdiagnosis is that physicians have little knowledge of the clinical and radiologic characteristics of this subtype of TDC. The previous reports have demonstrated that the typical symptoms of TDCs include the absence of anterior midline cystic masses, the sudden occurrence of pharyngeal foreign body sensation, and symptoms of upper respiratory obstruction such as stridor, dyspnea, and dysphagia; these respiratory symptoms are typically worse after colds, which was also true for patients in our group. In all the cases, the cysts recurred soon after Fig. 2. Axial un image (a) and image (b) demonstrating a cyst located at the midline in the posterior of the tongue (arrows). This cyst was unicystic and without any separations. The cystic wall was after injection of the contrast agent. Fig. 3. Sagittal image showing a cyst located near the foramen cecum that was pushing the epiglottis posterior (thin arrow) and formed a close relationship with the hyoid bone and the foramen cecum (bold arrow). In addition, the laryngopharyngeal cavity was diminished. 1890

4 Fig. 4. An excised cyst from a patient demonstrating that the cyst had a close relationship with the hyoid bone (bold arrow); the inner region of the cyst was stained with methylene blue (narrow arrow). resection. Under laryngoscopy, the cysts located at the middle or unilateral posterior region of the tongue are found. 10,15 In our study, all cases experienced recurrent cysts after every resection or marsupialization under endoscopy and had cystic masses located at the posterior of the tongue. In this study, we determined the main radiologic characteristics to distinguish this subtype TDC from epiglottis cysts and cysts of lingual root; TDCs are distinguished by the relationship between the cysts and the hyoid bone or the foramen cecum. This type of relationship was observed in all seven cases, regardless of the location of the cyst. It is commonly recognized that the Sistrunk surgery is the most effective treatment for TDC. However, for the aforementioned subtype of TDCs, some physicians feel that the Sistrunk procedure will result in many large unnecessary wounds, and therefore, physicians have tried many other surgical techniques, such as cystectomy and marsupialization under endoscopy. In the 1990s, Urao et al. 16 performed cyst marsupialization via the mouth for three children who did not show any recurrence during the 2- to 5-year follow-up period after surgery. Collin et al. 17 also reported a group of 16 such cases who underwent cystectomy under endoscopy, of which 2 (12.4%) experienced recurrence. The other 14 cases did not experience any recurrence during the follow-up period, which was a mean of 3.7 years after surgery. Although these two research studies have demonstrated some success for the treatment of TDC patients, both studies had some limitations. With respect to the investigation done by Uaro ert al., 16 the number of the patients was too small to achieve statistical significance; additionally, all of the cysts were unicystic without any segregations or branches. As for the investigation performed by Collin et al., 17 most of the cases did not have any branches or distributions, and these cysts were not difficult to resect entirely under endoscopy. If a case involves complicated branches originating from the main cyst and long duct connecting with the hyoid bone, it will be very difficult for the physician to incise the cysts under endoscopy. Therefore, for those patients with complicated lingual TDCs, we could first try to perform cystectomy or marsupialization under endoscopy; but if the case showed any recurrence, we should consider using the Sistrunk surgery. In this study, all seven cases were such cases, in which cystectomy or marsupialization under endoscopy was performed before the last Sistrunk procedure and in which recurrences occurred after every endoscopy procedure. To decrease the recurrence rate, and combining the typical characteristics of this subtype of TDC, we improved the accepted Sistrunk surgery in two ways. 1) We routinelyusedmethylenebluetotracethedistributionofthe TDC before surgery, which is very useful to show the range of the cyst. In 1920, Sistrunk used methylene blue in the assessment of those patients whose sinuses communicated with the outside, 18 atechniquethatwasprovento be useful for tracking the range of a cyst. However, for those common cases with TDC located at below the hyoid bone, the use of methylene blue was limited due to the difficulty of the injecting dye into the cyst. In our study, this situation was different. We were able to easily inject dye into the cyst through the oral cavity due to the location of thecystatthesuperiorpart.hence,weuseddyeinall cases. We were able to judge the incised range by determining whether the edge of the incision had been stained by methylene blue, and thus we were able to effectively decrease the rate of recurrence. 2) To further decrease the recurrence rate, we completely incised the mucosa covering the cyst, resulting in a larger incised range of the mucosa than that used by Sistrunk, who incised only the foramen cecum, 18 and we sutured the residual oropharynx cavity mucosa and submucosal tissue. Lingual TDCs are commonly located in the superficial part of the tongue and form a cystic mass that protrudes into the oropharyngeal cavity when it becomes large. The wall of the cyst and the pharyngeal mucosa are closely connected. For patients with this type of TDC, it is hard for a physician to separate the cyst wall from the mucosa, and if the separation is not complete, the cyst will recur. In conclusion, for patients with lingual TDCs, CT examination prior to surgery is useful and can help us to discriminate TDCs from the other cysts by observing the relationship between the cyst and the hyoid bone as well as the foramen cecum. When it comes to the treatment of TDCs, complete resection or marsupialization under endoscopy is the preferred method. However, if the patient experiences any recurrence of the cyst, the modified Sistrunk surgery could be an important remedy. Due to such limitations as the small sample size and the short follow-up time, the modified Sistrunk surgical procedure needs to be evaluated further by multicenter randomized controlled trials. AUTHOR CONTRIBUTOR STATEMENT L.C.Z. and T.Y.Z. equally participated in the conception, design, analysis, and interpretation of data, drafting of the manuscript and revising it for important intellectual content. Y.S., Y.X.L., and Q.C. participated in the data analysis and interpretation, searching for 1891

5 publications. All authors have approved the final version of the report. BIBLIOGRAPHY 1. Mondin V, Ferlito A, Muzzi E, et al. Thyroglossal duct cyst: personal experience and literature review. Auris Nasus Larynx 2008;35: Lin ST, Tseng FY, Hsu CJ, et al. Thyroglossal duct cyst: a comparison between children and adults. Am J Otolaryngol 2008;29: Madana J, Yolmo D, Saxena SK, et al. True thyroglossal fistula. Laryngoscope 2009;119: Purdom E, Robitschek J, Littlefield PD, Cable B. Acute airway obstruction from a thyroglossal duct cyst. Otolaryngol Head Neck Surg 2007;136: Nicollas R, Mimouni O, Roman S, Triglia JM. Intralaryngeal manifestation of thyroglossal duct cyst. Otolaryngol Head Neck Surg 2007;137: Fu J, Xue X, Chen L, et al. Lingual thyroglossal duct cyst in newborns: previously misdiagnosed as laryngomalacia. Int J Pediatr Otorhinolaryngol 2008;72: Paez P, Warren WS, Srouji MN. Stridor as the presenting symptom of lingual thyroglossal duct cyst in an infant. Clin Pediatr 1974;13: Kuint J, Horowitz Z, Kugel C, et al. Laryngeal obstruction caused by lingual thyroglossal duct cyst presenting at birth. Am J Perinatol 1997;14: Allard RHB. The thyroglossal cyst. Head Neck Surg 1982;5: Lubben B, Alberty J, Lang-Roth R, et al. Thyroglossal duct cyst causing intralaryngeal obstruction. Otolaryngol Head Neck Surg 2001;125: Sari M, Baylancicek S, Inanli S, et al. Unusual presentation and location of thyroglossal duct cyst in a child. Otolaryngology Head Neck Surg 2007;136: Eom M, Kim YS. Asphyxiating death due to basal lingual cyst (thyroglossal duct cyst) in two-month-old infant is potentially aggravated after central catheterization with forced positional changes. Am J Forensic Med Pathol 2008;29: Kanawaku Y, Funayama M, Sakai J, et al. Sudden infant death: lingual thyroglossal duct cyst versus environmental factors. Forensic Sci Int 2006;156: Byard RW, Bourne AJ, Silver MM. The association of lingual thyroglossal duct remnants with sudden-death in infancy. Int J Pediatr Otorhinolaryngol 1990;20: Brousseau VJ, Solares CA, Xu M, et al. Thyroglossal duct cysts: presentation and management in children versus adults. Int J Pediatr Otorhinolaryngol 2003;67: Urao M, Teitelbaum DH, Miyano T. Lingual thyroglossal duct cyst: a unique surgical approach. J Pediatr Surg 1996;31: Burkart CM, Richter GT, Rutter MJ, et al. Update on endoscopic management of lingual thyroglossal duct cysts. Laryngoscope 2009;119: Sistrunk WE. The surgical treatment of cysts of the thyroglossal tract. Ann Surg 1920;71:

Thyroglossal cyst our experience

Volume 3 Issue 1 2013 ISSN: 2250-0359 Thyroglossal cyst our experience Balasubramanian Thiagarajan 1 Ulaganathan Venkatesan 2 Geetha Ramamoorthy 1 1 Stanley Medical College 2 Meenakshi Medical College