Angioleiomyoma (AL) is an uncommon benign soft tissue neoplasia arising from the tunica media of the smooth muscle cells. AL appears as a solitary and slow-growing mass and seldom is observed in oral tissues. We reported a rare case of AL involving the cheek of a 17-year-old young woman. A review of the English-language literature was performed entering the keywords "angioleiomyoma" and "oral" in the search fields of PubMed. 70 results were identified. Excluded were cases that were not in the oral cavity or not compatible with the AL diagnosis or report lacking immunohistochemical analysis. According to the exclusion criteria, we selected 30 studies that included 63 cases of AL. The results of the review showed an average age of 42.97 years with a prevalence between the fourth and fifth decade of life with a male-to-female ratio of 1.95 : 1. The most affected sites were palate, buccal mucosa of the cheek, lip, tongue, and gingiva. Surgical excision was the treatment of choice, and diagnosis was possible through histopathological and immunohistochemical analysis. SMA, vimentin, CD34, desmin, and S-100 were the most common markers to guide the histopathological diagnosis of oral AL. In conclusion, oral AL is a rare entity, especially in adolescence as in the reported case of AL of the cheek in a 17-year-old woman. The clinical aspects of AL did not allow clinicians to make a correct presumptive diagnosis. A scrupulous histopathological analysis and immunohistochemical examinations are fundamental to differentiate AL from other lesions.