A young girl presented with an insidious onset, gradually increasing midline submental swelling of 1-year duration and pain on swallowing for 6 months. Ultrasonography of the neck suggested a hypoechoic cystic swelling of the submental region between the muscles of the floor of the mouth, with no increased vascularity. An extraoral surgical enucleation was done and a postoperative biopsy suggested an epidermoid cyst. Epidermoid cysts of the submental region are extremely rare and any midline head and neck lesion in children requires critical examination and evaluation to avoid complications. Here, we present a rare case of a paediatric submental epidermoid cyst and its clinical features and management.
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