Thyroglossal Duct Cyst Coexisting with a Fistulous Tract a Rare Case Report and Literature Review

Journal of Pediatric Surgery Case Reports 28 (2018) 59–61

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Journal of Pediatric Surgery Case Reports

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Thyroglossal duct cyst coexisting with a ﬁstulous tract: A rare case report T and literature review

Ramin Salimnejada, Arash Hasirbaf Momtazb, Ziba Ghoreishic, Majid Shokoohia, ∗ Daniyal Bolandhemmatd, Mohammad Amin Dolatkhaha, Hamed Shooreia, , Seyyed Mostafa Ghavamie a Department of Anatomical Sciences, Faculty of Medicine, Tabriz University of Medical Sciences, Tabriz, Iran b Department of Anatomical Sciences, Faculty of Medicine, Tarbiat Modares University, Tehran, Iran c Department of Nursing, Faculty of Paramedical, Mashhad University of Medical Sciences, Mashhad, Iran d Department of Medical Sciences, Faculty of Medicine, Birjand University of Medical Sciences, Birjand, Iran e Department of Radiology, Faculty of Medicine, Tabriz University of Medical Sciences, Tabriz, Iran

ARTICLE INFO ABSTRACT

Keywords: An 11-year-old boy was admitted to the hospital complaining of odorous discharge from an opening in the Thyroglossal duct cyst anterior region of the left side of his neck. During physical examinations, we understood that his cyst moved with Thyroglossal fistula deglutition and protrusion of the tongue. In order to get a correct diagnosis of the patient problem, fistulography Fistulography with contrast agent meglumine was done from the patient neck in two projections, anterior-posterior and anterior-posterior-oblique. By information obtained of physical examinations and his neck radiographs, thyroglossal duct cyst coexisting with a fistulous tract was diagnosed. In adolescents and adults, thyroglossal duct cyst is one the most common causes of midline congenital cyst formation in the neck that may appear anywhere between the base of the tongue and the suprasternal region. Thyroglossal duct cyst develops from a persistence of any portion of the thyroglossal tract in the embryonic period. After birthday, infection of cyst can sometimes lead to fistula formation in the neck. Both of them, thyroglossal duct cyst and thyroglossal fistula, are usually diagnosed by imaging techniques.

1. Introduction Occasionally, a portion of the thyroglossal duct may remain that forms an enclosed thyroglossal duct cyst (TGDC) and/or a thyroglossal fistula Embryologically, at the late of the 4th week of gestation, the first of (TGF) which has a communication with the surface of the neck [1,2]. the body's endocrine glands, thyroid gland or primordium, develops as The diagnosis of TGDC and TGF is usually made by a clinical history, a solid mass of endoderm epithelial cells proliferating at the apex of the physical examination, and imaging findings. There are several radi- foramen cecum. Foramen cecum is a site between 2 important struc- ologic procedures such as computerized tomography scan (CT), mag- tures; the tuberculum impar or median tongue bud which develops netic resonance imaging (MRI), medical ultrasound (Sonography), and from the first pharyngeal arch, and copula which develops from the X-ray imaging. One of the X-ray imaging techniques is fistulography second pharyngeal arch. These structures—tuberculum impar and co- that can show the course of the tract. Therefore, we present a case of an pula—are involved in forming the tongue. The thyroid primordium 11-year-old boy with thyroglossal duct cyst and a fistulous tract that subsequently descends from the foramen cecum on an S-shaped path diagnosed by physical examinations and imaging findings. anterior to the trachea. During the migration, there is a connection between foramen cecum and thyroid gland which forms the thyr- 2. Case report oglossal duct (TGD). At the end of the 5th week of gestation, the thyroglossal duct breaks down and begins to atrophy and finally, the An 11-year-old boy was admitted to the otolaryngology outdoor foramen cecum will normally be the only remnant of the thyroglossal with a complaint of odorous discharge from an orifice on his neck. The duct in adults. At the 7th week of gestation, the isolated thyroid gland patient presented a history of an opening located at the left lower reaches to the final position, inferior to the cricoid cartilage. anterior border of the sternocleidomastoid muscle for two years and

∗ Corresponding author. Gholgasht Ave, Tabriz University of Medical Sciences, Faculty of Medicine, Tabriz, Iran. E-mail address: [email protected] (H. Shoorei). http://dx.doi.org/10.1016/j.epsc.2017.09.030 Received 24 July 2017; Received in revised form 20 September 2017; Accepted 23 September 2017 Available online 27 September 2017 2213-5766/ © 2017 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/). R. Salimnejad et al. Journal of Pediatric Surgery Case Reports 28 (2018) 59–61

Fig. 1. Anterior-Posterior (AP) fistulography view of the neck. The arrow shows the Fig. 3. Lateral (Lat) fistulography view of the neck. The arrows show the thyroglossal thyroglossal duct cyst and fistulous open to the skin in the midline of the neck. duct cyst and the fistulous tract ascending towards the base of tongue and also the curved arrow shows the position of the hyoid bone.

and also the TGDC position is located in front of the hyoid bone (Figs. 2 and 3). CT scan and MRI due to radiation and cost considerations were not performed. Also, sonography due to its weakness to show the pathway of thyroglossal duct was not done.

3. Discussion

The thyroid gland is one of the endocrine glands that are normally located in the neck. It originates from a diverticulum (foramen cecum) present on the floor of the pharynx [2]. Around the fourth week of development, between the first and second pharyngeal pouches, an endodermal thicken appears. Then, it descends in front of the pharynx, hyoid bone, and laryngeal cartilages as a bilobed diverticulum. The diverticulum reach to its final position in the lower of the neck by the 7th week of gestation [1]. Therefore, during its descending, the pathway of the diverticulum forms the thyroglossal duct which usually disappears by the 5th week of gestation, but, in adults, a normal remnant of the thyroglossal duct on the dorsum of the tongue is known as Fig. 2. Anterior-Posterior-Oblique (APO) fistulography view of the neck. The arrows the foramen cecum [1,3]. However, after birth, a persistence of any show the thyroglossal duct cyst and the fistulous tract ascending towards the base of portion of the duct is considered as thyroglossal duct cyst [1–3].Ina tongue. quite uncommon situation, TGDC may present on the tongue that is named as lingual thyroglossal duct cyst (LTGDC) [4]. Moreover, Van- also told that occasionally has slight pain. In the physical examination, dana G. and Maruti D. reported a rare case of intra-thyroid thyroglossal no swelling around the orifice and no sign of cervical lymphadenopathy duct cyst [5]. TGDC is one of the causes of neck lumps and the most were observed. But, there was some odorous discharge when the patient common anomaly in the development of the thyroid gland which ac- neck squeezed. On the other hand, during physical examination, we counting for 70% of congenital neck anomalies [6,7]. Also, the thyr- understood that his cyst moved with deglutition and protrusion of the oglossal fistula may occur in this abnormally, TGDC. Both conditions tongue. According to these findings by physical examinations, the di- are rarely seen with together, like in our study. Common regions of agnosis of thyroglossal duct cyst with a fistulous tract that commu- TGDC are infrahyoid (25–65%), suprahyoid (15–50%), at the hyoid nicates the TGDC to the surface of the neck was made. Then, to find out level (20–25%), and rarely intralingual (3%) [8,9]. The incidence of that the upper end of fistula is open or not, fistulography with contrast TGD cyst in childhood and adults is 70% and 7%, respectively, also it is agent meglumine (76% compound meglumine diatrizoate) was per- more common in women than in men (1.5:1 ratio) [10,11]. Several formed after cannulating into the orifice of the fistula. The radiography studies have been reported that TGDCs move upward during swal- projections of the patient neck were true anterior-posterior (AP), lowing, deglutition, and tongue protrusion because of their attachment anterior-posterior-oblique (APO), and lateral (Lat). His neck images to the tongue via the tract of thyroid descent [12,13]. Therefore, by demonstrated an abnormally remain of the embryonic thyroglossal duct considering the mentioned fact, the diagnosis of TGDC during the that formed TGDC and a fistulous tract on the left side of the neck. The physical examination can be so simple. In order to diagnose TGD cyst position of the TGDC was unilateral, and near to the midline of the neck and/or fistula, CT scan, MRI, sonography, and X-ray imaging with (Figs. 1 and 2). During fistulography, the patient had no complaint of a contrast are useful. For example, one paper reported that computed bitter taste after the injection of the contrast medium. The oblique and tomography fistulography can be used as a method to diagnose thyr- lateral images showed that the upper end of the fistulous tract is closed oglossal fistula [14]. In addition, several studies have been reported that sonography as an inexpensive and easily available method can

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