Epidermoid cysts of the floor of the mouth represent <0.01% of all oral cysts. Only few cases have been reported so far. We report a case of a 24 years man with a large sublingual swelling for 17 years, unable to take solid meals. The swelling was approximately 13 × 13 cm, non-tender, non-transilluminant, with doughy consistency and with patent visible Wharton’s duct openings on both the sides. Initial decompression of the lesion was done to facilitate nasotracheal intubation followed by partial excision of the cyst with marsupialization. Histopathological examination revealed a thick wall cyst lined with pseudostratified squamous epithelium without any evidence of dermal appendages suggestive of epidermoid cyst. Postoperative care included extraction of the loose infected dentures and physiotherapy of the ankylosed temporomandibular joints. Clinical progress was uneventful and cosmetic appearance was acceptable on 6 month and 2 years follow up with normal mastication function.
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