Congenital leiomyomatous epulis: a case report with immunohistochemical study

The histologic and immunohistochemical findings of an extremely rare case of congenital soft tissue mass on the alveolar ridge in an infant are reported. The lesion clinically mimicked an ordinary congenital epulis (congenital granular cell epulis, granular cell tumor of the newborn); however, histologically it consisted of a conglomerate of spindle-shaped cells, akin to smooth muscle cells, which formed interlacing and whorled fasciculi. Nerve fibers with myxoid degeneration, capillaries and muscle walled small vessels intermingled with fasciculi of spindle-shaped cells. The border between the conglomerate of spindle-shaped cells and the surrounding connective tissue was not evident. Immunohistochemically, most of the spindle-shaped cells were intensely positive for antibodies to alpha-smooth muscle actin, HHF-35 and desmin. These findings suggest that the lesion was composed of mature smooth muscle cells that were of hamartomatous or choristomatous nature. The term 'congenital leiomyomatous epulis' is proposed.