Congenital melanotic macules and Sebaceous Choristoma arising on the tongue of a newborn: epidermal choristoma?

Abstract

Oral hyperpigmentation is a common event in older individuals, however, is exceptional in neonates (congenital melanotic macules). Conversely, 70-80% of people have sebaceous glands in the oral mucosa, with the tongue representing an ectopic location and termed sebaceous choristoma by some authors. We report a case that fulfills both conditions in a tongue lesion. A 1-month-old boy presented with a pigmented macula on his tongue noted at birth. An excisional biopsy was performed showing a lesion lined by an epidermal-like epithelium with basal pigmentation, under which, sebaceous glands, abortive hair follicles and ductal structures mimicking apocrine glands were found. Seven cases of congenital melanotic macules of the tongue have been reported, however, none of them showed sebaceous glands under the lesion. Furthermore, there has not been a reported case of sebaceous choristoma of the tongue present at birth. We present a case that shares clinical and histological features of both conditions and propose the name 'epidermal choristoma'.

Clinicopathologic conference: bluish gingival nodule

“…18 There are few reports of oral sebaceous choristoma in the English language literature, and most of the lesions were located in the posterior midline region of the tongue of adult patients. 19,20 To the best of our knowledge, this is the first report of a gingival sebaceous choristoma with the clinical appearance of a bluish nodule.…”

Congenital Melanotic Macules of the Tongue. A Case Report and Brief Review of the Literature

“…Congenital melanotic macules of the tongue are a rare and probably underestimated benign entity with only few cases reported in the literature . Table summarizes the clinical and histopathologic findings of those cases.…”

Epidermal choristoma arising on the midline gingiva as a congenital epulis: A case report

“…Epidermal choristoma was first reported by Azorin et al (2005). These investigators presented the case of a 1-month-old boy with three pigmented macula on the tongue.…”