Angioleiomyoma (AL) is a benign neoplasia originating from smooth muscle and very uncommon in the oral cavity. The most frequent subtype in the oral cavity is the vascular one. AL usually occurs in the extremities: only around 12% are found in other areas such as head and neck. It presents as an asymptomatic, slow growing nodule lodging in the palate, tongue or lips. The diagnosis is essentially by histological exam and special specific stains are helpful to confirm the origin and to distinguish it from other tumors. We present a case of AL found in unusual site: attached to the submandibular region in a deep-seated space.