Journal of Pediatric Surgery Case Reports 30 (2018) 74–76

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Journal of Pediatric Surgery Case Reports

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Sublingual epidermoid cyst in a young girl T

a a b,∗
Nidhi Kumari , Amit Kumar , Anil Kumar
a
Department of Oral and Maxillofacial Surgery, Buddha Institute of Dental Sciences and Hospital, Patna, India
b
Associate Professor, Department of General Surgery (Trauma and Emergency), All India Institute of Medical Sciences, Patna, India

A R T I C L E I N F O A B S T R A C T

Keywords: An epidermoid cyst is a benign cyst that can occur in any part of the body. The most common age group for this
Epidermoid cyst cyst is 20–40 years. It's extremely rare in oral cavity accounting for only 1.6%. The cyst develops from the
Dermoid cyst ectodermal tissue. It represents the most common cutaneous cysts. A 6 years old female patient presented with a
Sublingual complaint of swelling in the floor of the mouth. It measured 4 × 5 cm in size. Surgical excision with primary
Case report
closure was performed. We present this case because of its rare location, mistaken for other entities, like a ranula
and its occurrence at an early age. After excision, an oval-shaped mass of 4 × 5 cm has been described in this
case. The histopathological report confirms the diagnosis of epidermoid cyst. The epidermoid cyst commonly
occurs in middle-aged adult group, especially over the face, scalp & trunk area. Surgical excision is the treatment
of choice in such case. Epidermoid cyst of the oral cavity is an uncommon entity. Although it's common in
middle-aged adult but sometimes it may occur in pediatrics age group.

1. Introduction slightly raised but its movement was not affected by the position of
swelling. On palpation, the swelling was soft to firm, non-tender,
Epidermoid cysts are the benign condition that can occur anywhere smooth, fluctuant and was not associated with discharge. Although
in the body. Most cases have been reported in the ovaries and the submandibular and sublingual gland orifices could not be assessed be-
testicles (80%), with head and neck accounting for 7%. Its occurrence cause they were masked by the swelling. Bilateral milking of the glands
in the oral cavity is rare as epidermoid along with dermoid cysts produced thick mucus saliva suggested the patency of duct. All in-
comprises only 0.01% of all the oral cysts. In the oral cavity dermoid vestigations were within normal limits. Mandibular cross-sectional
cyst is twice common than epidermoid cyst [1]. We report a rare sub- radiograph did not disclose any calcifications. The buccal and lingual
lingual epidermoid cyst in a young girl. Our case report is in accordance cortical plates were normal with no indication of expansion or dec-
with SCARE criteria [12]. ortication. Aspiration done using an 18 gauge needle yielded scanty
white cheesy material. Computed tomography showed a well-defined
2. Case report submucosal oval mass in the floor of mouth in the midline measuring
approximately 4.0 × 3.0 cm in diameter. The underlying tongue
A 6-year-old girl presented with swelling in the floor of mouth just musculature was normal. Fine needle aspiration cytology (FNAC)
beneath the tongue (Fig. 1) for one year. Initially the size of swelling showed few squamous cells with mild hyperchromatic nuclei visible in
was a pea-sized but gradually increased in size. The patient was abso- the background sheets of macrophages, few lymphocytes and abundant
lutely asymptomatic except swelling in her mouth. Her voice, speech, keratin flakes. In view to deal the cyst of the sublingual area, naso-
swallowing and deglutition functions were normal. There was no his- tracheal intubation was performed and the patient was subjected to
tory of trauma in the oral-cavity. Examination of oral cavity revealed a general anaesthesia. Mouth gag heister introduced to open the mouth
solitary, well circumscribed, distinct, dome-shaped, sessile midline and subsequently tongue was lifted up to show the swelling beneath it.
swelling extending from the lingual aspect of the mucogingival junction Before excision cannulation of the Wharton's duct has done in view to
of mandibular anterior teeth up to the mandibular molars bilaterally. protecting it. Excision of the swelling yielded a reddish white smooth
Extraoral examination revealed a swelling in the sub-mental region surfaced oval mass measuring approximately 3 × 2 × 2 cm which was
(Fig. 2). The floor of the mouth revealed the mucosa over the swelling soft in consistency and cystic in nature (Fig. 3). Primary closure and
appeared normal without any secondary changes. The tongue was hemostasis were secured. The patient recovered uneventfully from

∗
Corresponding author. Gen Surgery (Trauma & Emergency), All India Institute of Medical Sciences, Type-5, Block-B, AIIMS Residential Complex, Hydraulic Road, Patna, 801105,
India.
E-mail addresses: [email protected] (N. Kumari), [email protected] (A. Kumar), [email protected] (A. Kumar).

https://doi.org/10.1016/j.epsc.2017.12.007
Received 15 November 2017; Received in revised form 7 December 2017; Accepted 9 December 2017
Available online 27 December 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/).
N. Kumari et al. Journal of Pediatric Surgery Case Reports 30 (2018) 74–76

Fig. 4. Photomicrograph showing stratified squamous epithelium without dermal ap-

Fig. 1. Arrow indicating Sub-lingual cyst.
pendages.

anaesthesia. Post-operative healing period was uneventful. The histo-

pathologic investigation reveals cystic cavity lined by thin, stratified
squamous epithelium without any evidence of dermal appendages and
confirms the diagnosis of the epidermoid cyst (Fig. 4). Patient has now
been on our follow-up for the last 18 months and is doing well. Written
informed consent was obtained from the father of patient (as patient
was minor) for publication of this case report and accompanying
images. A copy of the written consent is available for review by the
Editor-in-Chief of this journal on request.

3. Discussion

An epidermoid cyst is one of the three congenital developmental

cysts. Others two are the dermoid and teratoid cyst. Both epidermoid
and dermoid cyst arising from ectodermal tissue and differs by the
presence or absence of skin appendages [2,3]. Skin appendages are
present in dermoid cyst but not in the epidermoid cyst. These cysts are
rare, benign lesions found throughout the body, with 7% occurring in
the head and neck area [4] and 1.6% only in the oral cavity. During the
Fig. 2. Arrow indicating swelling over upper part of the neck.
closure of first and second branchial arches at the midline, if entrap-
ment of epithelium remnant occurs, it leads to the formation of epi-
dermoid cyst [5]. The epidermoid cysts are more common in between
15 and 35 years of age with male predilection [6,7]. In our case age of
the female patient was only 6 years with painless swelling in the sub-
lingual area. Zielinski R reported submental epidermoid cyst in a 6 year
old boy [8]. Based on the histopathological picture Meyer also divided
the floor of the mouth cysts into three types. 1. Epidermoid cysts which
are lined with epithelium without skin appendages. 2. Dermoid cysts
where epithelial lined cystic cavity encloses skin appendages such as
hair, hair follicles, sebaceous, and sweat glands. 3. Teratoid cysts in
which in addition to skin appendages also encloses mesodermal deri-
vatives such as bone, muscle, gastrointestinal and respiratory tissue.
Usually, the size of this cyst is in between 1 and 5 cm and rarely it may
be of larger size like > 5 cm [9,10]. The clinical presentation of epi-
dermoid cyst depends upon the size and site of the cyst [7]. Clinical
assessment along with Fine needle aspiration cytology (FNAC), ultra-
sound, computed tomography and magnetic resonance imaging is es-
sential to differentiate an epidermoid cyst from other midline swellings
of oral cavity like thyroglossal duct cysts, plunging ranulas and lipoma.
Although FNAC has been advocated as an essential investigation but not
equivalent to CT and MRI. Treatment of choice for epidermoid cyst is
total surgical excision. Excision may be carried out intraorally as well as
Fig. 3. Excised sub-lingual cyst.
extra oral approach. The main critical step in the removal of mouth
lesion is to protect the Wharton's duct and the best way to protection is

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N. Kumari et al. Journal of Pediatric Surgery Case Reports 30 (2018) 74–76

cannulation of the duct as done in the present case. Recurrences are cyst: a report of two cases. J Laryngol Otol 2006;120(3):E19.
unusual after absolute surgical excision. Reports of malignant trans- [2] Suga K, Muramatsu K, Uchiyama T, Takano N,Shibahara T. Congenital epidermoid
cyst arising in soft palate near uvula: a case report. Bull Tokyo Dent Coll
formation of epidermoid cysts are present in the literature [11]. In our 2010;51:207–11.
case, the histopathological report was epidermoid cyst without any [3] Lin HW, Silver AL, Cunnane ME, Sadow PM, Kieff DA. Lateral dermoid cyst of the
shreds of evidences of malignancy. Although recurrence is rare but floor of mouth.unusual radiologic and pathologic findings. Auris Nasus Larynx
2011;38:650–3.
needs a long-term follow up for an epidermoid cyst. [4] Koca H, Seckin T, Sipahi A, Kazanc A. Epidermoid cyst in the floor of the mouth:
report of a case. Quintessence Int 2007;38:473–7.
4. Conclusion [5] Kandogan T, Koç M, Vardar E, Selek E, Sezgin O. Sublingual epidermoid cyst: a case
report. J Med Case Rep 2007;1:87.
[6] Damle MV, Irani DK, Hiranandani NL. Epidermoid cyst of the floor of the mouth.
Epidermoid cyst of the oral cavity in a young girl is extremely rare. Case report. Bombay Hosp J 2002;44:267–70.
Even in asymptomatic case surgical excision is the ideal treatment. In [7] Hemaraju N, Nanda SK, Medikeri SB. Sub-lingual dermoid cyst. Indian J
Otolaryngol Head Neck Surg 2004;56:218–20.
view of recurrence long-term follow up are recommended.
[8] Zielinski R, Zakrzewska A. Submental epidermoid cyst in children. Open Med
(Wars) 2014 Nov 11;10(1).
Conflict of interests [9] Kalgutkar A, Kini S, Jambhekar N, Das S. Intradiploic primary epithelial inclusion
cyst of the skull. Ann Diagn Pathol 2006;10:20–3.
[10] Kang SG, Kim CH, Cho HK, Park MY, Lee YJ, Cho MK. Two cases of giant epidermal
The authors have no conflicts of interests to disclose. cyst occurring in the neck. Ann Dermatol 2011;23(Suppl 1):S135–8.
[11] López-Ríos F, Rodríguez-Peralto JL, Castaño E, Benito A. Squamous cell carcinoma
Appendix A. Supplementary data arising in a cutaneous epidermal cyst: case report and literature review. Am J
Dermatopathol 1999;21:174–7.
[12] Riaz A Agha, Alexander J Fowler, Alexander Saeta, Ishani Barai, Shivanchan
Supplementary data related to this article can be found at http://dx. Rajmohan, Orgill Dennis P. The SCARE Statement: consensus-based surgical case
doi.org/10.1016/j.epsc.2017.12.007. report guidelines. Int J Surg 2016 October;34:180–6.

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