Abstract
A 20-year-old man presented to the oral and maxillofacial surgery department for evaluation and treatment of a dentofacial deformity. On examination, it was noted that he had marked hemifacial hyperpigmented papillomatous lesions covering the upper and middle thirds of the right side of his face extending to the right upper lid. Of note, he also had extensive intraoral involvement. These features were highly suggestive of linear epidermal nevus, a congenital hamartomatous lesion of the ectoderm. Linear epidermal nevus with oral manifestations is extremely rare with very few cases reported in the medical literature.
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