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Eur Arch Otorhinolaryngol (2005) 262 : 353355 DOI 10.

1007/s00405-004-0813-1

MISCELLANEOUS

Kemal Grr Derya mit Talas Cengiz zcan

An unusual presentation of neck dermoid cyst

Received: 16 January 2004 / Accepted: 6 May 2004 / Published online: 17 July 2004 Springer-Verlag 2004

Abstract Congenital masses are the most common noninflammatory neck lesions in children. Although usually present at birth, they can appear at any age. Dermoid cysts are benign lesions of congenital origin usually presenting as a midline neck mass. They rarely appear in the lateral region of the neck. Lateral cervical dermoid cyst is presented as a rare, unusual case, and differential diagnosis and management are discussed in light of recent literature. Keywords Dermoid cyst Head and neck Branchial region Congenital cysts Cervical malformation.

Case report
A 16-year-old male patient referred to our ENT department with the complaint of a left lateral cervical mass. His medical history revealed pain and gradually increasing swelling during upper respiratory infections for the last 3 years. There was no history of drainage originating from the mass, hoarseness, disphagia or odinophagia over a period of 2 years. His grandmother and uncle had undergone neck cyst extirpation. Otorhinolaryngological examination revealed a painless and fluctuating mass anterior to the upper 1/3 of the sternocleidomastoid muscle (SCM). Contrast-enhanced computed tomography (CT) showed a cystic mass in the anterio-medial region of the SCM and postero-inferior to the posterior belly of the digastric muscle, having a lipoid density. The mass extended to the parapharyngeal space superomedially, and the ipsilateral submandibular gland was displaced anteriorly. Also, two distinct signal densities were noted, and the possible diagnosis was anticipated as a dermoid cyst or lipodermoid tumor (Fig. 1). Under general endotracheal anesthesia, the cervical skin incision was per-

Introduction
Dermoid cysts (DC) are rare, benign teratamatous congenital lesions. They are located in the orbit, the nasal dorsum, floor of the mouth, infratemporal fossa and anterior side of the neck [1, 2, 3]. Only 17% of all DC affect the head and neck region, while 23% of those are seen in the floor of the mouth [4, 5]. Dermoid cysts, like teratomas, consist of embryonal germ layers. Teratomas contain all three germ layers, including the ectoderm, mesoderm and endoderm, while DCs contain only mesoderm and ectoderm [6]. Mesodermal elements, which include hair follicles and sweat glands in the wall of the dermoid cysts, differentiate them from simple epidermoid cysts [6, 7]. They rarely appear in the lateral region [8]. The purpose of this report is to present and discuss a rare case of lateral cervical dermoid cyst.

K. Grr D. . Talas C. zcan Department of Otolaryngology, University of Mersin, Mersin, Turkey K. Grr () Fatih mah, Babil cad Girne sitesi, D blok, No:1, 33170 Mezitli Mersin, Turkey Tel.: +90-324-3374325, Fax: +90-324-3374305, e-mail: kgorur@mersin.edu.tr

Fig. 1 Two distinct signal densities (fluid and solid elements) appeared in the cervical CT of the patient

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Fig. 2 Segment of cyst wall displaying keratinizing squamous epithelial lining closely associated with mature sebaceous gland lobules (H&E; 100) formed parallel to the skin lines. After division of the platysmal muscle, the mass was exposed. The mass was seen inferior to the posterior digastric muscle during surgical dissection. It was well encapsulated and adhered to the surrounding structures. During the dissection of the mass from the surrounding structures, the cyst was ruptured and its contents (hair follicles, sebaceous glands and adipose matrix) drained. The wall of the cyst was removed totally, and the postoperative period was uneventful. Pathologic examination reported a dermoid cyst, and the histopathological detail of it is demonstrated in Fig. 2. The postoperative period was uneventful, and no recurrence was noted in the follow-up period for 3 years.

Discussion
Dermoid cysts are defined as masses histopathologically lined by keratinized, stratified, squamous epithelium [7]. They commonly appear along embryonic fusion rests [6, 7]. Like other congenital neck masses, DCs are usually noticed during the first few years of life, but if asymptomatic, may not be observed until much later [9]. In the head neck region, they are commonly located in the dorsum of the nose, orbita, floor of the mouth and anterior aspect of the neck [4, 9]. DCs may represent in cystic or solid forms [10]. While cystic DCs are filled with a cheesy, keratinous material, solid dermoid tumors are generally pedunculated and consist of connective tissue and thick fibro-fatty matrix [11]. The cystic mass of our case was filled with epidermal appendages and cheesy liquid material.

There is still controversy about the etiology of DCs. Three theories have been suggested regarding the explanation of pathogenesis. According to the totipotential rest theory, DCs have been suggested to arise from totipotent cells derived from ectodermal and mesodermal germinal layers. The congenital inclusion theory proposes inclusion of germinal layers into deeper tissues of fusion lines that have failed to undergo complete closure during embryonic life leading to epithelial debris trapping. The acquired implantation theory indicates traumatic events for the implantation of germinal derivatives into deeper tissues [9]. Although there have been some reports about inherited transmission in etiology, definite inheritance has not been clarified [9, 12]. Bratton et al. [9] reported cases of a mother and her identical twin daughters who were all found to have evidence of frontal nasal dermoid cysts. The history of our case revealed that the patients grandmother and uncle had undergone neck cyst extirpation; however, the definite pathological diagnoses of these cysts were unknown. Most cases of DC of the lateral neck are seen in the submandibular triangle between the mylohyoid and the hyoglossus muscle [11, 13]. Many authors feel that DCs of the lateral neck localize in the midline and that they may expand laterally during their growth [11, 14]. We could not identify any tract or stalk projecting toward the midline in our case. Although preoperative clinical examination is important in the diagnosis of DCs, unusual presentation of lateral cervical masses, similar to our case, show that more detailed radiographic studies may be needed for reliable diagnosis. Branchial cleft cysts, thyroglossal duct cysts, plunging ranula, lipomas, lymphomas and squamous cell

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carcinomas should be considered in the differential diagnosis [11, 15, 16]. Lymph nodes and dermoid cysts do not move because they are not attached to the underlying structures [11, 15]. In our case, observed mesodermal appendage fluid levels on CT examination were thought to be diagnostic of DC. Midline DC can be confused with thyroglossal duct cysts. DCs do not elevate with tongue protrusion as thyroglossal duct cysts do. If they raise the floor of the mouth, a DC may be mistaken for ranula [6]. De Mello et al. found that 11 of 71 supposed thyroglossal duct cysts were actually DC on pathologic examination [17]. In order to prevent subsequent infection and malignant degeneration, obtain a pathologic diagnosis or correct a cosmetic deformity, surgical excision is the choice for successful treatment [11, 18, 19].
Acknowledgements The authors would like to thank Dr. Serdar nl for his help in the preparation and description of the histopathological photograph.

References
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6. Burns BV, Axon PR, Phade A (2001) Hairy polyp of the pharynx in association with an ipsilateral branchial sinus: evidence that the hairy polyp is a second branchial arch malformation. J Laryngol Otol 115:145148 7. Bloom D, Carvelho D, Edmonds J, Magit A (2002) Neonatal dermoid cyst of the floor of the mouth extending to the midline neck. Arch Otolaryngol Head Neck Surg 128: 6870 8. Pincus RL (2001) Congenital neck masses. In: Bailey BJ (ed) Head and neck surgery otolaryngology. Lippincott Williams & Wilkins, Philadelphia, pp 932939 9. Bratton C, Suskind DL, Thomas T, Kluka EA (2001) Autosomal dominant familial frontonasal dermoid cysts: a mother and her identical twin daughters. Int J Pedaitr Otorhinolaryngol 57:249253 10. Rahbar R, Shah P, Mulliken JB, Robson CD, Perez-Atayde AR, Proctor MR, Kenna MA, Scott MR, Mc Gill TJ, Healy GB (2003) The presentation and management of nasal dermoid. Arch Otolaryngol Head Neck Surg 129:464471 11. Rosen D, Wirtschafter A, Rao VM, Wilcox TO (1998) Dermoid cyst of the lateral neck: a case report and literature review. Ear Nose Throat J 77:125132 12. Nicollas R, Guelfucci B, Roman S, Triglia JM (2000) Congenital cysts and fistulas of the neck. Int J Pediatr Otorhinolaryngol 55:117124 13. Mc Avoy JM, Zuckerbraum L (1976) Dermoid cysts of the head and neck in children. Arch Otolaryngol 102:529531 14. Leveque H, Saraceno CA, Tang CK, Blanchard CL (1979) Dermoids cysts of the floor of the mouth. Laryngoscope 89:296 305 15. zcan C, nal M, Grr K (2001) Servikal (Plunging) ranula: olgu sunumu. Trk ORL Ars ivi 39:4750 16. zcan C, Grr K, Talas D, Aydn (in press) Intramucular benign lipoma of the sternocleidomastoid muscle: a rare cause of neck mass. Eur Arch Otorhinolaryngol 17. De Mello DE, Lima JA, Liapis H (1987) Midline cervical cysts in children: thyroglossal anomalies. Arch Otolaryngol 113:418420 18. Mc Avoy JM, Zuckerbraum L (1976) Dermoid cysts of the head and neck in children. Arch Otolaryngol 102:529531 19. zcan C, Talas D, Grr K, Pata YS (2002) Congenital neck lesions: retrospective study (in Turkish). ukurova University Journal of Health Sciences 17:1116

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