Abstract
Oral leiomyomatous hamartoma (OLH) is a rare lesion seen in the oral cavity. It mainly presents on the median maxilla and tongue. In the literature in English, there are only 10 reported cases of OLH of the median maxilla. Most of the cases were found in patients of Japanese and Latin American origin. We report a case of OLH in an 18-month-old boy of Middle Eastern ancestry. The lesion presented as a pedunculated, light pink, soft swelling that was located on the labial gingiva of tooth number 21. Microscopically, it showed proliferative smooth-muscle fascicles dispersed in loose fibrous stroma and multiple small vessels. The lesional cells looked mature and elongated and were deeply eosinophilic spindle cells with basophilic, central cigar-shaped nuclei. The diagnosis of OLH was supported by positive immunohistochemical reactivity of smooth-muscle actin and desmin. To our knowledge, this is the first reported case of OLH in a Middle Eastern patient.