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| CASE REPORT |
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| Year : 2021 | Volume
: 23
| Issue : 1 | Page : 92-96 |
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Oral epidermoid cyst: clinicopathological report of a rare case
Mohammed Adam Sheikh Abdullahi1, Abba Bukar Zarami2, Abubakar Kaka Sanda1, Hector Olasoji1
1 Oral and Maxillofacial Surgery Department, Faculty of Dentistry, University of Maiduguri Teaching Hospital, Maiduguri, Nigeria
2 Pathology Department, University of Maiduguri Teaching Hospital, Maiduguri, Nigeria
| Date of Submission | 25-Jun-2020 |
| Date of Decision | 08-Sep-2020 |
| Date of Acceptance | 16-Sep-2020 |
| Date of Web Publication | 28-Apr-2021 |
Correspondence Address:
Dr. Mohammed Adam Sheikh Abdullahi
Oral and Maxillofacial Surgery Department, University of Maiduguri Teaching Hospital Maiduguri, Borno State
Nigeria
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jomt.jomt_42_20
Background: Epidermoid cyst of the oral cavity is very rare, slow-growing, benign neoplasm derived from the remnant of the neural tube. It accounts for less than 0.01% of all cystic lesions of the head and neck. Most of the intraoral cases are reported in the midline and floor of the mouth. Very rare cases are seen involving the tongue, lips, uvula, temporomandibular joint, maxillary, and buccal mucosa. The rare oral lesion is asymptomatic, but large masses present early with difficulty in breathing and swallowing. We report an uncommon case of epidermoid cyst in an infant with failure to thrive due to obstruction and inability to latch breast milk. Case report: A 40-day-old male infant presented with a large oral swelling in the floor of the mouth since birth. It measured 4 × 5 cm in size. Surgical excision with primary closure was successfully performed. The histopathological report confirmed the diagnosis of epidermoid cyst. The child recovered to full general health with no signs of recurrence. Conclusion: Oral epidermoid cyst is a rare neoplasm that may mimic dermoid cyst and teratoid tumors clinically. The treatment of choice is surgical excision; however, there is a recurrence rate of 33% and rarely transforms into malignancy.
Keywords: Epidermoid cyst, floor of mouth, oral
How to cite this article:
Abdullahi MA, Zarami AB, Sanda AK, Olasoji H. Oral epidermoid cyst: clinicopathological report of a rare case. J Med Trop 2021;23:92-6 |
| Introduction | |  |
Epidermoid cyst of the oral cavity is a very rare clinical entity.[1],[2],[3] It is a benign developmental malformation, with the incidence of 7% in the head and neck region and 1.6% in the oral cavity.[4],[5] It constitutes less than 0.01% of all cystic lesions of the oral cavity.[4],[5] Most of the intraoral cases are reported in the midline and floor of the mouth.[3] Very rare cases are seen involving the tongue, lips, uvula, temporomandibular joint, maxillary, and buccal mucosa.[6] It is a slow but progressive tumor,[4] which is usually developed congenitally especially in children or acquired.[4] The pathogenesis of midline cyst in the floor of the mouth is not well established; however, three hypotheses have been proposed regarding its origin − dysontogenetic, traumatic, and thyroglossal.[4],[5] The tumor has an indolent clinical cause, but when it grows very large, it can present with difficulty in inbreathing, mastication, speech, or secondary infections.[7]
The rarity of oral epidermoid cyst presenting as a huge mass causing obstruction, inability to latch breast milk, and failure to thrive prompted the need for this case report. [Figure 1],[Figure 2],[Figure 3],[Figure 4] | Figure 1 (A and B): Patient before surgery and 3 months after with mouth open showing the tongue and floor of the mouth
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 | Figure 2 (A): Front view of cyst; (B) side view of cyst; (C) failure to thrive; (D) orally intubated
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 | Figure 3 (A and B): Cyst exposed intraoperatively; (C) excised cyst; (D) immediately postoperarively; (E and) patient 3 months postoperatively
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 | Figure 4: Photomicrograph of epidermoid cyst. K shows inclusion cyst lined by stratified squamous epithelium lacking skin adnexal glands; L shows lamellate keratin within the cyst (H and E, ×100)
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| Case presentation | | |
A 40-day-old male infant was referred to oral and maxillofacial surgery department for evaluation and management of large oral mass noticed at birth, initially bean sized but gradually increased to current size with no associated pain, discharge, or bleeding. Failure to thrive due to poor intake and inability to latch breast milk since birth. Patient has been on nasogastric tube feeding of expressed breast milk. There are no respiratory or cardiovascular symptoms. Parents are from rural part of Borno State. Mother is a 20 years old with two children, all alive. Father is 35 years old.
On examination, we found an ill-looking infant, pale, afebrile, anicteric, acyanosed, no pedal oedema with large solitary, well-circumscribed swelling in the midline floor of the mouth protruding extraorally and maximally opening the mouth. It was soft, fluctuant, nondischarging, and nontender and measured approximately 4 × 5 cm in widest dimension. Overlying mucosa appears intact with minimal dilated capillaries and no secondary changes. Tongue position, submandibular, sublingual orifices could not be assessed because of the size of the swelling. Radiographs revealed no calcifications and no bony changes. Fine needle aspiration yielded mucoid aspirate with scanty cheesy material. Cytology showed few squamous cells with mild hyperchromatic nuclei. Visible in the background were sheets of macrophages, few lymphocytes, and abundant keratin flakes. Ultrasound scan showed a well-defined cystic lesion with dense debris seen within the floor of the mouth measuring 3.36–4.31 cm with no calcifications. The cyst was excised under general anesthesia with an endotracheal tube. Excision of the swelling yielded a reddish white, smooth surfaced oval mass measuring approximately 4 × 5 cm, which was soft in consistency and cystic in nature. Primary closure was effected and hemostasis secured. The postoperative recovery was satisfactory and recovery was uneventful evidenced by good tongue movement, normal suckling, and weight gain. Histology report revealed epidermoid cyst. The patient was discharged a week after and reviewed after 3 months in which floor of the mouth was inspected with no recurrence and patient gained adequate weight. Patient is currently on long-term follow-up till adulthood.
| Discussion | | |
The epidermoid tumor was first described in 1683 by Duverney.[8] The first diagnosed case was reported by a French pathologist Cruveilhier in 1829 and since then the disease was named as Cruveilhier tumor.[9] The tumor grossly appears bright white and for that reason Remak[8] first suggested the epidermal origin of the lesion.[9] The lesion is one of the uncommon slow-growing congenital tumors.[9] It develops from ectodermal remnants in the neural tube when it separates during the third and fifth weeks of gestation of embryonic life.[9] In one series, 71% of epidermoid cysts occurred up to the age of 30 years and 91% up to the age of 45 years.[7] The male to female ratio was 3:2 in most reported cases.[7] Based on anatomical location; cysts have been classified into sublingual cyst; those above mylohyoid, genioglossus muscle in the floor of the mouth as we found in this index case presented and submental cyst; and those between mylohyoid and geniohyoid.[4] The floor of the mouth constitutes 71% of cases; nevertheless, they are rare and one-third of the cases are located in the lateral part, 52% sublingual, 16% submental, and 6% submandibular.[7] The tumors generally are asymptomatic, but when they grow large as in our case, they present very early with difficulty in breathing and swallowing.[4] It sometimes poses diagnostic challenges because of the wide differential diagnosis that can resemble the tumor and these include ranula, blockage of Wharton’s duct, thyroglossal cyst, cystic hygroma, and brachial cleft cyst.[4]
Malignant transformation of an epidermoid cyst is exceptional.[4] However, few cases of carcinomatous changes have been reported in the ovary and intracranial location.[4],[9] Rarely the tumor undergoes malignant transformation due to chronic inflammatory stimulation as a result of repeated cyst rupture.[6]
The epidermoid cyst has similar features with the cutaneous type.[10] The cyst cavity is filled with a soft, waxy, or flaky keratohyalin material that results from the progressive desquamation of the cyst wall.[6] The cyst contents are mucoid constituting cellular debris, keratin, and cholesterol, which can easily be aspirated or scooped out.[6] The cyst wall is lined by stratified squamous epithelium supported by an outer layer of the collagenous stroma.[10] Cases having epithelial atrophy of the cyst lining, acanthosis, hyperplasia, and cones formation have been described.[10] Although histological variation also exists between genders, females showed more hyperplasia of the epithelial lining whereas males showed more of calcification, especially in intracranial epidermoid cyst.[10]
The treatment of choice for epidermoid cyst is surgical.[1],[2],[3],[4],[5],[6],[7] However, radical excision of the cyst wall is difficult because of its firm adherence to neurovascular structures.[1],[2],[3],[4] A recurrence rate of 33% has been reported following partial or incomplete resection.[2],[4],[5] Mostly the tumor recurs within 1 to 2 years postoperatively.[2],[4],[10] The major complication during surgery is due to spillage of the cyst contents onto surrounding tissue.[1] Other complications include adhesion and it rarely transforms to squamous cell carcinoma.[5],[6]
| Conclusion | | |
Oral epidermoid cyst is a rare neoplasm and they may mimic dermoid cyst and teratoid tumors clinically. Surgical excision is the treatment of choice; however, a recurrence rate of 33% with increased complication when it ruptures should be noted. The tumor rarely transforms into malignancy.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Koca H, Seckin T, Sipahi A. Epidermoid cyst in floor of mouth: report of a case. Quintess Int 2007;38:473-7.  |
| 2. | King RC, Smith BR, Burk JL. Dermoid cyst in the floor of mouth, review of literature and case reports. Oral Surg Oral Med Oral Pathol 1994;78:567-76. doi: 10.1016/0030-4220(94)90166-x. PMID: 7838461. |
| 3. | Walstad WR, Soloman JM, Schow SR, Ochs MW. Midline cystic lesions of floor of mouth. J Oral Maxillofac Surg 1998;56:70-4. |
| 4. | Tandon PN, Gupta DS. Epidermoid cyst in the floor of mouth with sub mental component. J Maxillofac Oral Surg 2014;13:59-62. |
| 5. | Uppala D, Majumdar S, Rao K, Reddy S. Epidermoid cyst of the soft palate in an infant. J Oral Maxillofac Pathol 2015;19:409. doi: 10.4103/0973-029X.174685. PMID: 26980982; PMCID: PMC4774307. |
| 6. | Suzuki C, Maeda M, Matsumine A, Matsubara T, Taki W, Maier SE et al. Apparent diffusion coefficient of subcutaneous epidermoid cyst in the head and neck comparison with intracranial epidermoid cyst. Acad Radiol 2007;14:1020-29. |
| 7. | Assaf AT, Heiland M, Blessmann M, Friedrich RE, Zustin J, Al-Adam A. Extensive sublingual epidermoid cyst − diagnosis by immunohistological analysis and proof by podoplanin. In Vivo 2012;26:323-326. |
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| 9. | Teegala R. Pure intrinsic brain stem epidermoid cyst: case report, review of the literature and new classification. Nepal J Neuro Sci 2017;14:33-41. |
| 10. | Moreno-Jimenez S, Tena-Suck ML, Collado-Ortiz MA, Castillejos-Lopez M, Alveolar-Moreno D, Holland MK. Intracranial epidermoid cyst in a single Mexican institution, experience of over 16-years. Patol Rev Latinoam 2012;50:182-9. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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