The Laryngoscope C 2012 The American Laryngological, V Rhinological and Otological Society, Inc. Thyroglossal Duct Cysts in Adults Treated by Ethanol Sclerotherapy: A Pilot Study of a Nonsurgical Technique Tam-Lin Chow, FRCS (Edin), FHKAM (Surgery); Chi-Yee Choi, FRCS (Edin), FHKAM (Surgery); Joyce Yee-Hing Hui, FRCR, FHKAM (Radiology) Objectives/Hypothesis: To investigate the effect of ethanol sclerotherapy on the thyroglossal duct cyst (TDC). Study Design: Prospective case series. Methods: Patients with primary TDC were enrolled. The volume of the TDC was calculated using the following formula: length width height p/6. Under sonographic guidance, ethanol was slowly instilled into the TDC cavity after the cyst fluid was aspirated. The procedure was performed in an outpatient setting. Results: A total of eight patients were recruited for this study, but two of them did not receive sclerotherapy. One patient refused treatment after obtaining initial consent, and another patient was not treated due to a technical issue. The median follow-up duration was 21 months. The median cyst volume was 3.5 mL. Of the six patients given sclerotherapy, recurrent TDC occurred in one patient. As expected, the TDC persisted in the two patients who had not undergone sclerotherapy. Two patients experienced moderate pain after the procedure that was well controlled with oral analgesics. No major complications arose, and no patient needed hospitalization because of treatment complications. Conclusions: Percutaneous ethanol sclerotherapy is an effective minimally invasive modality of therapy for TDC. Further studies with longer follow-up are warranted. Key Words: Ethanol, sclerotherapy, thyroglossal duct cyst. Level of Evidence: 4 Laryngoscope, 122:1262–1264, 2012 INTRODUCTION Thyroglossal duct cyst (TDC) is a congenital problem caused by the persistent embryonal thyroglossal duct remnant. The patients can present with asymptomatic upper anterior neck lumps, which sometimes might be complicated with infection. The classical treatment is the Sistrunk operation,1 in which the TDC, middle onethird of the hyoid bone, and part of the tongue base around the foremen caecum are removed. The recurrence rate is about 3%. The Sistrunk operation has stood the test of time and remained the operation of choice for TDC.2,3 A recent anatomic study discovered that the thyroglossal duct arborizes with the formation of multiple side tracts,4 which can account for the uncommon recurrence after the classic Sistrunk operation. Therefore, the extended Sistrunk operation, in which a central core of soft tissue is dissected and removed at the anterior neck compartment, from the thyroid gland to tongue base in addition to the hyoid bone and fibrous tract, has been From the Department of Surgery (T.-L.C., C.-Y.C.) and Department of Diagnostic Radiology and Organ Imaging (J.Y.-H.H.), United Christian Hospital, Kowloon, Hong Kong. Editor’s Note: This Manuscript was accepted for publication January 30, 2012. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Dr. Tam-Lin Chow, Department of Surgery, United Christian Hospital, 130 Hip Wo Street, Kowloon, Hong Kong. E-mail: [email protected] DOI: 10.1002/lary.23254 Laryngoscope 122: June 2012 1262 advocated to prevent recurrence. Kim et al.5 and Patel et al.6 reported encouraging outcomes by adopting the extended approach for patients with a prior failed Sistrunk procedure. However, we should be cautious in applying it as the primary surgery for TDC to balance against the increased morbidity of the extended resection.5 Minimally invasive intervention had been successfully practiced for different benign head and neck cystic lesions (e.g., branchial cyst, plunging ranula, lymphangioma, and TDC).7,8 The lesions were treated by percutaneous sclerotherapy with OK-432. Because OK432 is not available in our locality, we investigated the effect of sclerotherapy of the TDC with ethanol. MATERIALS AND METHODS The diagnosis of TDC relies on the unequivocal tongue tug sign of the anterior midline neck cystic nodule. All adult patients with primary TDC were invited to participate in this study after obtaining informed consent. This prospective study was approved by our institutional review board. Apart from conventional magnetic resonance imaging sequences, axial 0.7-mm-thick T2-weighted gradient-echo (3DFT-CISS) magnetic resonance examination (Seimens Medical Solutions, Erlangen, Germany) of the neck was performed to rule out the existence of a patent tract in communication with the oropharynx. Sclerotherapy was then arranged on an outpatient basis. For sonography, an iU22 ultrasound scanner (Philips Healthcare, Andover, MA) was performed using a 5 to 12 MHz linear probe. The volume of the TDC was calculated using the following formula: length width height p/6. For Chow et al.: Ethanol Sclerotherapy for TDC TABLE I. Patient Demographics, Clinical Characteristics, and Ethanol Sclerotherapy Outcome. Case Gender Age, yr TDC Size, cm3 Sclerotherapy Follow-Up, mo TDC Relapse Complication 1 Female 44 1.7 Yes 1 21 Nil Nil 2 Female 49 1.7 Nil* 21 NA NA 3 4 Male Female 61 42 9.1 2.0 Yes 1 Yes 1 53 23 Nil Nil Nil Nil 5 Female 37 1.5 Yes 1 17 Nil Nil 6 7 Male Female 48 52 5.5 4.9 Yes 1 Nil† 21 13 Nil NA Nil NA 8 Female 37 9.8 Yes 3 72 Yes Nil *Patient refused sclerotherapy. † Cyst fluid too viscous. TDC ¼ thyroglossal duct cyst; NA ¼ not applicable. a complex cyst, volume was calculated as the volume of the cystic component rather than solid areas. Under sonographic guidance, a 20- to 22-gauge needle was introduced into the TDC without local anesthesia, and the cyst fluid was aspirated as completely as possible. The aspirates were sent to the laboratory for cytology examination. After that, ethanol was slowly instilled into the TDC cavity at a rate of approximately 2 to 5 mL/min. A volume of 50% to 90% of the original volume of aspirated fluid was injected. Injection of ethanol was stopped if ethanol was seen leaking out from the nodule under ultrasound or if the patient complained of pain. Follow-up ultrasonography was arranged at 3 months, 6 months, and 1 year after the procedure. Sclerotherapy could be repeated if considerable residual/recurrent fluid collection was noted. The data were prospectively collected. RESULTS A total of eight patients were recruited for this study (Table I). However, ethanol sclerotherapy was not given to two patients. One patient (case 2) refused the procedure after consent was obtained. In another patient (case 7), the cyst fluid was too viscous to be aspirated, Fig. 1. Preablation ultrasound depicts thyroglossal cyst with some echogenic debris. Laryngoscope 122: June 2012 and thereby sclerotherapy was not carried out due to a technical issue. The median follow-up duration was 21 months (range, 13–72 months) The demographic data and outcome are summarized in Table I. There were six female and two male patients. Their median age was 46 years (range, 37–61 years). The TDCs were asymptomatic in five patients, whereas three patients experienced mild pain. Past infection of the TDC occurred in one patient. The median cyst volume was 3.5 mL (range, 1.7–9.8 mL). For those six patients who had undergone sclerotherapy, one session of treatment was given in five patients, whereas three sessions were required in the remaining patient. No patient suffered from hoarseness or hemorrhage after therapy. Two patients experienced moderate pain after the procedure that was well controlled with oral non-narcotic analgesics (paracetamol) for 2 days. No patient needed hospitalization because of treatment complications. Of the six patients given sclerotherapy, recurrent TDC occurred in one patient. The recurrent nodule was only 1 cm and painless. The patient refused further sclerotherapy or surgical extirpation. As expected, the TDC persisted in the two patients without sclerotherapy. Both of them declined the Sistrunk operation. Of those five patients with successful ablation without recurrence, the cysts vanished completely (Fig. 1 and Fig. 2). Fig. 2. Postablation ultrasound shows complete disappearance of the thyroglossal cyst. Chow et al.: Ethanol Sclerotherapy for TDC 1263 DISCUSSION In general, benign neck cysts are treated by surgical excision. Percutaneous sclerotherapy has gained popularity in the past decade. In 2005, Taguchi et al.9 reported one case of neonatal branchial cleft cyst treated by intralesional injection with OK-432. Subsequently, Roh et al.10 reported a series of 12 branchial cleft cysts managed with intracystic injection of OK-432. There was no recurrences in their series. The use of OK-432 sclerotherapy has expanded to plunging ranula, lymphangioma, and TDC.7,8 Akin to OK-432, ethanol can be used as a sclerosing agent for benign cysts. Ethanol sclerotherapy for thyroid cysts is well documented.11–14 The merit of ethanol over OK-432 is the absence of post-treatment fever or flu-like symptoms. Moreover, OK-432 is not licensed in our locality. Therefore, we used ethanol as the sclerosing agent in our study. Percutaneous ethanol sclerotherapy was well tolerated by the patients. Only two patients in our series complained of moderate pain after therapy. The pain was well controlled with a non-narcotic analgesic (paracetamol). There were no complications of hoarseness or bleeding. All of the procedures were performed easily in an outpatient setting. The outcome was very positive in this study. Only one patient suffered a small TDC relapse out of the six patients who had undergone the sclerotherapy. Conversely, the two patients who did not undergo sclerotherapy after enrollment had persistent disease. The recurrent case (case 8) was the first trial case, and the cyst was the biggest (9.8 cm3). Whether the relapse was due to the learning curve or large cyst volume is uncertain due to the small sample size of this study. Further studies with more patients are needed to investigate the parameters for treatment failure. The median follow-up in our series was 21 months, which indicates that the therapeutic effect is sustaining. Likewise, Fukumoto et al.15 reported their experience of ethanol sclerotherapy in two patients with TDC. Both patients were cured with the follow-up duration around 30 months. The instilled ethanol can migrate to the secondary tracts of the TDC, which could explain the effectiveness of sclerotherapy on TDC despite tract arborization. Laryngoscope 122: June 2012 1264 General anesthesia is necessary when performing the Sistrunk operation. The surgical complications include bleeding, infection, and scar formation. The patients undergoing surgical treatment also need hospitalization. Therefore, the advantages of percutaneous ethanol sclerotherapy for TDC are that it is less invasive than surgery, there is minimal pain, it has a lower cost, and it provides a better cosmetic outcome. CONCLUSION Percutaneous ethanol sclerotherapy is an effective minimally invasive modality of therapy for TDC. Further studies with longer follow-up are warranted. BIBLIOGRAPHY 1. Sistrunk WE. Technique of removal of cysts and sinuses of the thyroglossal duct. Ann Surg 1920;71:121–124. 2. Pelausa EO, Forte V. Sistrunk revisited: a 10-year review of revision thyroglossal duct surgery at Toronto’s Hospital for Sick Children. J Otolaryngol 1989;18:325–333. 3. Lin ST, Tseng FY, Hsu CJ, Yeh TH, Chen YS. Thyroglossal duct cyst: a comparison between children and adults. Am J Otolaryngol 2008;29: 83–87. 4. Hoisawa M, Ninomi N, Ito T. Anatomic reconstruction of the thyroglossal duct. J Pediatr Surg 1991;26:766–769. 5. Kim MK, Pawel BR, Isaacson G. 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Treatment of cystic lesions of the thyroid by ethanol instillation. World J Surg 1992;16:958–961. 12. Verde G, Papini E, Pacella CM, et al. Ultrasound guided percutaneous ethanol injection in the treatment of cystic thyroid nodules. Clin Endocrinol 1994;41:719–924. 13. Monzani F, Lippi F, Goletti O, et al. Percutaneous aspiration and ethanol sclerotherapy for thyroid cysts. J Clin Endocrinol Metab 1994;78: 800–802. 14. Bennedbak FN, Hegedus L. Treatment of recurrent thyroid cysts with ethanol: a randomized double-blind controlled trial. J Clin Endocrinol Metab 2003;88:5773–5777. 15. Fukumoto K, Kojima T, Tomonari H, Kontani K, Murai S, Tsujimoto F. Ethanol injection sclerotherapy for Baker’s cyst, thyroglossal cyst, and branchial cleft cyst. Ann Plast Surg 1994;33:615–619. Chow et al.: Ethanol Sclerotherapy for TDC
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