Congenital granular cell epulis is a rare benign soft tissue lesion arising from the alveolar ridge in neonates. A rare case of multiple congenital granular cell epulis is reported, alongside a description of its vascular immunohistochemical profile. A female newborn presented with two exophytic pedunculated red nodules located on the alveolar ridge between the future eruption sites of the incisors and canines of the mandible and maxilla. A conservative surgical excision was performed on the second day of life. Histology revealed proliferation of round granular cells containing an abundant eosinophilic cytoplasm with basophilic nuclei, ranging from round to oval in shape. Numerous blood vessels were also seen. Immunohistochemical analysis of the granular cells revealed positivity for CD68, D2-40, Ki67, VEGF, and FGF and negativity for S100, CD34, and CD105. Immunostaining for CD34, CD105, and D2-40 confirmed the presence of a large number of blood and lymphatic vessels. Although rare, an understanding of this lesion is paramount for correct diagnosis and appropriate treatment. In the present report, the immunohistochemical profile confirmed increased vascularization, proving that these lesions are composed of not only new and preexisting blood vessels, but also lymphatic vessels.

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