Angioleiomyoma (AL) is a benign tumor derived from smooth muscle. The overwhelming majority of ALs occur in the uterus, gastrointestinal tract, or skin; AL of the oral cavity is infrequent, and AL of the tongue is particularly rare. The present report describes a case of AL of the tongue that resulted in a good outcome. We also review the literature with a special emphasis on the etiology of ALs. It is generally agreed that minor trauma, venous stasis, and hormonal changes are etiological factors for AL. The roles of estrogen and progesterone have been particularly emphasized, and the expression of progesterone receptors (PRs) and estrogen receptors (ERs) has been recently evaluated in some cases of AL. To our knowledge, the presence of PRs and ERs has only been evaluated in seven cases of AL arising in different regions of the body including the oral cavity. This is the first report to demonstrate negative expression of both receptors in an AL of the oral cavity. Further study and additional cases are needed to elucidate the influence of PRs and ERs in AL of the oral cavity.
Keywords: Angioleiomyoma; Estrogen receptor; Etiology; Progesterone receptor; Tongue.