Clinical and Immunohistochemical Features of Oral Angioleiomyoma: A Comprehensive Review of the Literature and Report of a Case in a Young Patient

Abstract

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.

Clinical, Imaging and Histopathology of Angioleiomyoma of the Buccal Cheek

“…In conclusion, we have reported a case of angioleiomyoma of the buccal cheek that resulted in a good outcome. Among various studies, the most common sites of oral angiomyomas were the lip [2,4,[12][13][14][15]32], cheek or buccal mucosa [5,8,14,[16][17][18], palate [7,[19][20][21], tongue [6,22,23], and submandibular area [9,11,24]. Other areas such as the gingiva, mandible, retromolar area, and anterior maxillary labial fold were only reported once among studies [2,10,17].…”

“…Other areas such as the gingiva, mandible, retromolar area, and anterior maxillary labial fold were only reported once among studies [ 2 , 10 , 17 ]. Due to the benign nature of this lesion, the treatment is excisional biopsy and there have been no recurrences or complications reported so far [ 32 ].…”

Pediatric leiomyoma of the oral cavity

“…[ 13 23 ] The key markers include SMA, vimentin, CD34, desmin and S-100. [ 24 25 26 27 ] Leiomyomas express immunoreactivity for SMA and negativity for desmin and S-100 protein. [ 23 26 27 ] It is advocated that each marker is representative of a particular tissue and should be correlated with clinical finding and the measurement of other markers.…”

Oral Angioleiomyoma in Early Childhood Patient: A Case Report of an Uncommon Lesion and Literature Review

“…Leiomyoma is a benign neoplasm with vascular smooth muscle (tunica media) origin. 1 , 14 It is observed in sites abundant with smooth muscles such as the myometrium, the gastrointestinal tract, and the skin. 15 Leiomyoma is relatively rare or uncommon in oral cavity due to smooth muscle scarceness in this location.…”

“… 11 The mean age is 45 years, with a slight predilection for males, and it is rare in children. 1 , 4 , 11 , 14 We experienced an AL case in a 1-year-old child's palate and it is presented with a literature review (LR) discussing clinical findings, the modern pathological knowledge, and the management of the lesion.…”