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raz J Otorhinolaryngol. 2018;84(3):398---399

www.bjorl.org

Brazilian Journal of

OTORHINOLARYNGOLOGY

ASE REPORT

nusual coexisting thyroglossal duct cyst and secondranchial cleft fistula in an adult�

oexistência incomum de cisto do ducto tireoglosso e fístula da segundaenda branquial em adulto

ong Hoon Lee, Tae Mi Yoon, Joon Kyoo Lee ∗, Sang Chul Lim

honnam National University Medical School & Chonnam National University Hwasun Hospital, Department oftolaryngology-Head and Neck Surgery, Hwasun, South Korea

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eceived 12 August 2015; accepted 19 November 2015vailable online 28 February 2016

ntroduction

hyroglossal duct cysts are the most common congenitaleck masses, followed by branchial cleft anomalies.1 Thy-oglossal duct cysts are three times more prevalent thanranchial cleft anomalies.2 However, to our knowledge,oexisting thyroglossal duct cyst and branchial cleft anomalyas been reported only once in the literature.3 Herein, weresent the second case report of coexisting thyroglossaluct cyst and second branchial cleft fistula in a 34 year-oldale.

ase report

34 year-old male presented with recurrent anterior andateral neck mucoid discharge since childhood. The patienteported no relevant medical history except for a bilat-ral tonsillectomy 25 years ago. The physical examinationevealed two external openings on the patient’s neck. One

� Please cite this article as: Lee DH, Yoon TM, Lee JK, Lim SC.

nusual coexisting thyroglossal duct cyst and second branchial cleftstula in an adult. Braz J Otorhinolaryngol. 2018;84:398---9.∗ Corresponding author.

E-mail: joonkyoo@jnu.ac.kr (J.K. Lee).

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ttps://doi.org/10.1016/j.bjorl.2015.11.017808-8694/© 2016 Associacao Brasileira de Otorrinolaringologia e Cirurgpen access article under the CC BY-NC-ND license (http://creativecomm

as located at the hyoid level; the other was locatedlong the anterior border of the lower third of the ster-ocleidomastoid muscle (SCM). No palpable cervical massr subcutaneous tract, and no active inflammation wereresent around the lesions. Computed Tomography (CT) scanf the neck revealed an 8.2 cm elongated rim-enhancing cys-ic lesion along the left anterior neck, extending from thenterior border of the lower third of the SCM to the ton-illar fossa (Fig. 1). CT scan showed no fistulous track oryst at the hyoid level, and no connection between the twoxternal openings. The pharyngoesophagogram showed noeakage from the pharynx.

Based on these observations, the patient was diagnosedith a left second branchial cleft fistula and a likely thy-

oglossal duct cyst. As the first surgical step for the branchialleft fistula, a transverse elliptical incision was made aroundhe external opening. With gentle traction on the fistula, theissection proceeded extending to its cephalic portion. After

stepladder incision, the fistulous tract was dissected to theonsillar fossa, where it was ligated and separated (Fig. 2).e performed the Sistrunk’s operation for the fistula at theyoid level under the working diagnosis of a thyroglossaluct cyst (Fig. 2). Intraoperative pharyngoscopy revealed noharyngeal opening. Pathological examination of the lesions

evealed thyroglossal duct cyst and branchial cleft fistula.he patient’s postoperative course was uneventful. At 14onths follow-up, there has been no recurrence, and theatient remains asymptomatic.

ia Cervico-Facial. Published by Elsevier Editora Ltda. This is anons.org/licenses/by-nc-nd/4.0/).

Thyroglossal duct cyst and branchial cleft fistula

Figure 1 Axial computed tomography scan of the neck showsan 8.2 cm elongated rim-enhancing cystic lesion (arrow) alongthe left anterior neck.

Figure 2 Intraoperative finding shows a dissected left secondbranchial cleft fistula (black arrow) by stepladder incisions, and

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a thyroglossal duct cyst (white arrow) with attached hyoid bone.M, mandible.

Discussion

Thyroglossal duct cysts are the most common cause of con-genital neck masses.1 They usually present as midline neckmasses and can be found at any point between the fora-

men cecum and suprasternal notch.4 They are painless,enlarge slowly, and may be associated with a fistula or sinustract.4 In this case, thyroglossal duct cyst presented as a fis-tula. Branchial cleft anomalies can present as cysts, sinuses,

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artilaginous remnants, or fistula; they account for approxi-ately 30% of all congenital head and neck lesions, with theajority being second brachial cleft anomalies.1,2,5 Over-

ll, cystic lesions are more common than fistulae in patientsith second branchial cleft anomalies.6 The current patientresented with a fistula with a small external opening alonghe anterior border of the lower SCM.

Ultrasound, CT, or, magnetic resonance imaging (MRI)ay be helpful to evaluate both thyroglossal duct cysts

nd branchial cleft anomalies.1,4 The definitive treatmentf thyroglossal duct cysts and branchial cleft anomalies isomplete surgical excision of the entire tract. After diag-ostic CT scan in this case, the authors performed surgicalxcision of the entire tract by the Sistrunk’s operation andtepladder incision.

There were some unique characteristics to this case.irstly, this was the second reported case of unusual coexist-ng thyroglossal duct cyst and second branchial cleft fistulan an adult.3 This finding highlights that thyroglossal ductysts and branchial cleft fistula are encountered in adults.herefore, coexisting thyroglossal duct cysts and branchialleft fistula should be considered in the differential diagno-is of neck lesions, even in adults. Secondly, the patient didot present with a history of infection and previous surgicalrocedure for congenital neck lesions. There is a possibil-ty of an asymptomatic thyroglossal duct cyst and branchialleft fistula occurrence without palpable neck lesions indults. Thirdly, if thyroglossal duct cyst is suspected in andult, the Sistrunk’s operation, removing the central portionf the hyoid, is the optimal choice of therapy not only foresthetic reasons, but also for management of recurrentnfections and the potential danger of malignancy.1,6

onclusion

oexisting thyroglossal duct cysts and branchial cleft fistulahould be considered in the differential diagnosis of neckesions, even in an adult.

onflicts of interest

he authors declare no conflicts of interest.

eferences

. Erikci V, Hosgör M. Management of congenital neck lesions inchildren. J Plast Reconstr Aesthet Surg. 2014;67:e217---22.

. Roback SA, Telander RL. Thyroglossal duct cysts and branchialcleft anomalies. Semin Pediatr Surg. 1994;3:142---6.

. Kyi M, Zin T, Paijan R, Abdullah, Din N. A case of an adultsynchronous thyroglossal cyst and branchial sinus: case report.Internet J Pathol. 2012:13.

. Androulakis M, Johnson JT, Wagner RL. Thyroglossal duct andsecond branchial cleft anomalies in adults. Ear Nose Throat J.1990;69:318---22.

. Ozoleck JA. Selective pathologies of the head and neckin children: a developmental perspective. Adv Anat Pathol.

2009;16:332---58.

. Hong SM, Moon SB. Low-lying thyroglossal duct cyst with lat-eral cervical discharge masquerading as a second branchial cleftfistula: a case report. J Pediatr Surg. 2013;48:429---31.