INFECTIOUS NATURE OF ORAL ANGIOLEIOMYOMAS – A SYSTEMATIC REVIEW
Main Article Content
Keywords
Oral angioleiomyoma, Vascular leiomyoma, Smooth muscle tumour, Oral cavity, Lips, Buccal mucosa, Palate, Tongue, Histopathology, Immunohistochemistry
Abstract
Background:
Oral angioleiomyoma is a rare benign neoplasm derived from the smooth muscle of vascular walls, representing only a minute proportion of intraoral soft-tissue tumors. Its clinical appearance is often nonspecific, frequently resembling fibrous, salivary, or vascular lesions, which makes diagnosis challenging. Histopathological confirmation, supported by immunohistochemical profiling, is essential; however, published evidence remains limited and scattered.
Objective:
To systematically review the literature on oral angioleiomyomas, focusing on their demographic and anatomical distribution, clinical presentation, histopathological and immunohistochemical characteristics, treatment approaches, and recurrence rates.
Methods:
A protocol registered with PROSPERO (CRD420251148693) guided this review, which was conducted according to PRISMA 2020 guidelines. Comprehensive searches of PubMed/MEDLINE, Embase, Scopus, Web of Science, Cochrane Library, and Google Scholar were undertaken to identify studies describing histologically confirmed oral angioleiomyomas. Eligible designs included case reports, case series, and observational studies presenting original patient data. Information on demographics, lesion site, clinical features, histological subtype, immunohistochemical profile, treatment, and outcomes was extracted.
Results:
Five studies (three case reports and two case series) encompassing 68 patients with 80 oral angioleiomyomas met the inclusion criteria. Patient ages ranged from 27 to 80 years (mean ≈55), with a slight male predominance. Lesions occurred most often on the lips, followed by the buccal mucosa, palate, and tongue. Clinically, they presented as slow-growing, well-circumscribed submucosal nodules, typically painless and <2 cm, with colour ranging from bluish-purple to normal mucosa. Histopathology revealed interlacing fascicles of spindle-shaped smooth-muscle cells surrounding vascular channels; the venous subtype predominated, while solid, cavernous, and adipocyte-rich variants were less frequent. Immunohistochemistry consistently demonstrated diffuse smooth muscle actin, with frequent desmin and h-caldesmon expression and negative S-100 staining. All studies reported conservative excision as the treatment of choice; only one recurrence occurred after incomplete removal, and no malignant transformation was documented.
Conclusions:
Oral angioleiomyoma is an uncommon but well-characterised benign smooth-muscle tumour of the oral cavity. Although its clinical appearance can mimic a range of other lesions, histopathological evaluation with a focused immunohistochemical panel remains the diagnostic gold standard. Simple local excision provides definitive management, with recurrence exceptional when margins are clear. Larger multicentre studies with standardised reporting and long-term follow-up are warranted to refine epidemiological estimates and confirm the biological behaviour of variant subtypes.
References
2.Veeresh M, Sudhakara M, Girish G, Naik C. Leiomyoma: A rare tumor in the head and neck and oral cavity: Report of 3 cases with review. J Oral Maxillofac Pathol. 2013 May;17(2):281-7. doi: 10.4103/0973-029X.119770. PMID: 24250094; PMCID: PMC3830242.
3.Matiakis A, Karakostas P, Pavlou AM, Anagnostou E, Poulopoulos A. Angioleiomyoma of the oral cavity: a case report and brief review of the literature. J Korean Assoc Oral Maxillofac Surg. 2018 Jun;44(3):136-139. doi: 10.5125/jkaoms.2018.44.3.136. Epub 2018 Jun 26. PMID: 29963495; PMCID: PMC6024062.
4.Jafarian M, Mashhadi Abbas F, Ghazizadeh Ahsaie M, Saebnoori H. Clinical, Imaging and Histopathology of Angioleiomyoma of the Buccal Cheek. Case Rep Dent. 2021 Apr 30;2021:9940304. doi: 10.1155/2021/9940304. PMID: 34012685; PMCID: PMC8105109.
5.Matsuyama A, Hisaoka M, Hashimoto H. Angioleiomyoma: a clinicopathologic and immunohistochemical reappraisal with special reference to the correlation with myopericytoma. Hum Pathol. 2007 Apr;38(4):645-51. doi: 10.1016/j.humpath.2006.10.012. Epub 2007 Jan 31. PMID: 17270242.
6.Giudice A, Bennardo F, Buffone C, Brancaccio Y, Plutino FM, Fortunato L. Clinical and Immunohistochemical Features of Oral Angioleiomyoma: A Comprehensive Review of the Literature and Report of a Case in a Young Patient. Case Rep Dent. 2019 Feb 27;2019:2498353. doi: 10.1155/2019/2498353. PMID: 30937194; PMCID: PMC6415311.
7.Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, Shamseer L, Tetzlaff JM, Akl EA, Brennan SE, Chou R, Glanville J, Grimshaw JM, Hróbjartsson A, Lalu MM, Li T, Loder EW, Mayo-Wilson E, McDonald S, McGuinness LA, Stewart LA, Thomas J, Tricco AC, Welch VA, Whiting P, Moher D. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021 Mar 29;372:n71. doi: 10.1136/bmj.n71. PMID: 33782057; PMCID: PMC8005924.
8.Amir-Behghadami M, Janati A Population, Intervention, Comparison, Outcomes and Study (PICOS) design as a framework to formulate eligibility criteria in systematic reviews Emergency Medicine Journal 2020;37:387.
9.Brooks JK, Nikitakis NG, Goodman NJ, Levy BA. Clinicopathologic characterization of oral angioleiomyomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002 Aug;94(2):221-7. doi: 10.1067/moe.2002.125276. PMID: 12221390.
10.Mahima VG, Patil K, Srikanth HS. Recurrent oral angioleiomyoma. Contemp Clin Dent. 2011 Apr;2(2):102-5. doi: 10.4103/0976-237X.83071. PMID: 21957385; PMCID: PMC3180846.
11.Rawal SY, Rawal YB. Angioleiomyoma (Vascular Leiomyoma) of the Oral Cavity. Head Neck Pathol. 2018 Mar;12(1):123-126. doi: 10.1007/s12105-017-0827-9. Epub 2017 Jun 6. PMID: 28589436; PMCID: PMC5873482.
12.Aitken-Saavedra J, da Silva KD, Gomes AP, Vasconcelos AC, Etges A, Nóbrega TG, Tarquinio SB. Clinicopathologic and immunohistochemical characterization of 14 cases of angioleiomyomas in oral cavity. Med Oral Patol Oral Cir Bucal. 2018 Sep 1;23(5):e564-e568. doi: 10.4317/medoral.22649. PMID: 30148476; PMCID: PMC6167101.
13.Bubola J, Bradley G, Leong I, Chugh D, Magalhaes M. Oral angioleiomyomas: a case series and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol. 2025 Oct;140(4):467-475. doi: 10.1016/j.oooo.2025.04.097. Epub 2025 Apr 28. PMID: 40410117.

