epidermoid cysts are benign sequestration cysts in body that may be either congenital or acquired . the congenital variety is related to aberrant embryogenic ectodermal implantation during at the time of closure of neural groove or when epithelial surfaces fuse . acquired cysts are due to traumatic iatrogenic implantation of epidermal fragments during trauma or surgery . epidermoid cysts are seen throughout the body with only 7% cases seen in oro - facial region and 1.6% within oral cavity . we herewith report a case of exceptional existence of intraoral multiple epidermoid cysts in both sides of the buccal mucosa in a 35-year - old male in the buccal cheek region . to the best of our knowledge a 35-year - old male reported to surgical department with the chief complaint of swelling in the both sides of buccal mucosa for 3 months , which was incidentally noticed . intraoral examination revealed a bilateral 1.5 cm  1.5 cm nontender swelling in lateral buccal mucosa with normal covering mucosa . extraorally , the skin overlying the swelling did not show any indurations . there was no associated regional lymphadenopathy . a detailed case history of the patient provided no association of cheek biting with the lesion . excision of the lesion lying immediately underlying the buccal mucosa was done under local anesthesia by giving an elliptical incision in the bilateral buccal mucosa . specimen on gross examination showed gray - white to gray - brown soft - tissue pieces , altogether measuring 0.5 cm  0.5 cm in size . microscopically , swelling from both the sides showed three subepidermal cysts arranged in linear fashion , lined by stratified squamous epithelium and lumen partially filled with keratin [ figure 1a  d ] . surrounding stroma showed fibrocollagenous tissue with mild diffuse chronic inflammatory infiltrate comprising lymphocytes , plasma cells , and macrophages . neither dysplasia nor malignancy was noted . a histopathological diagnosis of bilateral multiple epidermoid cysts was made . ( a and b ) both right and left side linearly arranged multiple linearly arranged epidermoid cysts with overlying stratified squamous epithelial clusters ( h and e , 40 ) ; ( c ) section shows cyst lined by stratified squamous epithelium with granular cell layer . no appendages are noted within the wall of cyst ( h and e , 100 ) ; ( d ) cyst wall lined by orthokeratotic stratified squamous epithelium with keratin flakes in the lumen ( h and e , 400 ) most of the cases are reported in between 15 and 35 years of age group . the incidence of epidermoid cysts in head and neck has been reported from 1.6% to 6.9% . most of the intraoral cases are reported in the midline and floor of the mouth . rare cases are reported involving tongue , lips , uvula , temporomandibular joint , intracranial , maxilla and mandible , and buccal mucosa . intraoral epidermoid cyst often remains asymptomatic for years until they reach a size that interferes with mastication , speech , and very rarely breathing . epidermoid cysts may be classified as congenital or acquired even if there is no difference between the two on presentation or histologically . congenital cysts are dysembryogenetic lesions that arise from ectodermic elements entrapped during the midline fusion of the first and second branchial arches between the 3 and 4 weeks of intrauterine life . alternatively , they may arise from the tuberculum impar of his which , with each mandibular arch , forms the floor of the mouth and the body of the tongue . acquired cysts derive from traumatic or iatrogenic inclusion of epithelial cells or from the occlusion of a sebaceous gland duct . however , for formation of acquired cysts , a proper combination of events that is a history of trauma along with implanted epithelium that is capable of proliferation and coexistent minimal inflammation are needed to occur simultaneously . these events occurring bilaterally along with the linear arrangement of the cyst probably explain the rarity of this case . epidermoid , dermoid , and teratoid cysts are nonodontogenic cystic lesions of the oral cavity . they are rare lesions derived from germinal epithelium . while a dermoid cyst has an epidermal lining with skin adnexa such as hair follicles and sebaceous glands , the epidermoid cyst contains no such adnexa . teratoid cysts in addition to skin appendages have tissue from other embryonal sources such as respiratory , gastrointestinal , and connective tissues such as bundles of striated muscle and distinct areas of fat . however , to the best of our knowledge , till now , no case has been reported as multiple epidermoid cysts in bilateral buccal mucosa . however , numerous epidermoid cysts , especially in head and neck , are associated with gardner 's syndrome . differential diagnosis should include a variety of conditions which can be developmental , neoplastic , and infectious . infectious conditions such as odontogenic infections are mostly symptomatic and can be ruled out in the present case as it was not associated with any clinical symptoms . swelling due to lipid , salivary , and vascular benign tumors should be considered . among developmental category , an oral lymphoepithelial cyst which is seen adjacent to waldeyers 's ring should also be kept in mind . the bilaterality of the lesion and confirmation on histopathology help to rule out the above pathologies . cases which are symptomatic and others that have undergone malignant transformation have been reported in the literature . cortezzi and de albuquerque in 1994 reported an epidermoid cyst of the floor of the mouth that reached a huge proportion and caused a life threatening situation because of secondary infection . basal cell carcinoma and squamous cell carcinoma arising in the wall of an otherwise conventional epidermoid cyst has been seen . however , no carcinomatous transformation was noted in our case . epidermoid cysts are found to exist in rare site which emphasize the need for further research into the etiopathogenesis of the cysts . the present case shows no variation from the normal histopathology , but they prove to be significant because of its variation in their anatomical presentation that is buccal mucosa , multiplicity , and bilaterality . more importantly , if ever malignancy is confirmed in such cases , appropriate evaluation is need to be carried out followed by definitive management .