Angioleiomyoma affecting the lips: Report of 3 cases and review of the literature

Abstract

Angioleiomyoma is an uncommon benign soft tissue tumor usually found in the lower extremities and rarely observed in oral tissues. It is microscopically characterized as a proliferation of smooth muscle cells intermingled with abundant vascular channels. Oral angioleiomyomas affect mostly the lips, palate, buccal mucosa and tongue, and appears as a submucosal painless nodule. Upper lip is seldom affected and only few cases have been reported. We report three additional cases of angioleiomyoma affecting the lips of elderly patients. All lesions were asymptomatic and presented as submucosal nodules of approximately 1cm. Microscopic analysis on H&E sections revealed similar pattern in all cases, showing well-circumscribed and encapsulated tumors characterized by proliferation of smooth muscle cells and large amount of wide vascular spaces of varying sizes. Most tumor cells were immunoreactive for α-smooth muscle actin, desmin and HHF-35. CD34 was also positive on the endothelial cells. All patients were surgically treated and no recurrence was observed so far. The oral pathologists and clinicians should consider this entity when assessing nodular lesions on upper lip.

Angioleiomyoma presenting as asymmetric persistent swelling of the lower lip

“…1 The reported cases of labial angioleiomyomas typically presented as a small asymptomatic round or raised sessile submucosal nodule, which therefore appeared quite similar to a mucocele, a hemangioma, a salivary gland tumor, a fibrous hyperplasia, or a pyogenic granuloma. 4,6 However, the lesion in our case looked more like a swelling of the lip rather than a well-defined round nodule, which may be attributed to its large size. The lip swelling may result from numerous conditions, including trauma, angioedema, cheilitis, acromegaly, mucopolysaccharidosis, and bacterial or herpes infection.…”

“…2,3 Furthermore, there has been no previously reported case of labial angioleiomyoma greater than 2 cm in diameter to our knowledge. 4,5 Among various etiological factors proposed, minor trauma may be relevant for angioleiomyoma of the lip, which is subject to repeated minor injuries. 1 The reported cases of labial angioleiomyomas typically presented as a small asymptomatic round or raised sessile submucosal nodule, which therefore appeared quite similar to a mucocele, a hemangioma, a salivary gland tumor, a fibrous hyperplasia, or a pyogenic granuloma.…”

Schwannoma of the upper lip

“…The most common lesions in this site are fibroma, inflammatory fibrous hyperplasia, salivary gland tumors, mucous retention cyst and mucocele 4,8 . However, other rare lesions have also been reported in this site, such as tufted angioma 9 and angioleiomyoma 10 .…”

Clinicopathologic conference: bluish gingival nodule

“…6 They are considered uncommon in the oral cavity and usually affect the lips and palate of middle-aged individuals. 7 The main clinical appearance is of a bluish painless nodule measuring less than 2 cm, being rarely observed in the gingiva, as in this case. 6,7 Kaposi sarcoma (KS) is an angioproliferative disorder caused by human herpesvirus 8, usually related to human immunodeficiency virus infection.…”