Printer Friendly

Leiomyoma of the Cheek.

1. Introduction

Leiomyomas are benign mesenchymal tumors arising from nonepithelial tissues, such as smooth muscle [1, 2]. These tumors are well-circumscribed neoplasms that are encountered frequently in the dermis, the gastrointestinal tract, and the female genital tract, in particular, the uterus [3, 4]. As smooth muscle cells are relatively rare in the oral cavity, in comparison with the gastrointestinal tract, oral leiomyomas are extremely rare, with an incidence of less than 1% of all benign, soft tissue tumors [5]. Leiomyomas are neoplasms which develop due to mutation that results in the loss of the growth regulation mechanisms of the smooth muscle cells [5, 6]. The development of this benign tumor depends on a complex interaction between hormones (estrogen, progesterone), growth factors, and cytokines, as well as genetic predisposition [7, 8]. The risk factors include middle age (fourth to sixth decades of life), dark skin, women with a history of chronic disease, including recurrent gynecological infections, and a high body mass index [9]. Leiomyomas are manifested clinically as slow-growing, asymptomatic lesions that are well-circumscribed and often purplish in color [10, 11]. Histologically, three types of leiomyoma can be distinguished: (a) leiomyoma (solid leiomyoma), (b) angioleiomyoma (vascular leiomyoma), and (c) epithelioid leiomyoma (leioblastoma) [12, 13]. The clinical aspects of leiomyomas are indistinguishable from other tumors located in the tongue or in the cheek, including pleomorphic adenomas, lymphangioma, pyogenic granulomas, and schwannomas [14, 15]. Diagnosis is based on histopathological examination, and surgical excision is the treatment of choice, with recurrence being extremely rare [16, 17]. The present study describes a rare case of a leiomyoma in the cheek of a 43-year-old woman.

2. Case Report

A 43-year-old woman, a nonsmoker, with no medical antecedents was referred to the Departments of Oral and Maxillofacial Surgery of a dental college in northern Brazil for evaluation of a well-circumscribed, solitary, asymptomatic, mobile mass in the right cheek, which was slightly purplish in color, causing facial asymmetry (Figures 1(a) and 1(b)).

The patient reported having had a swelling in the right cheek over the previous two years. She also reported that she was not on any drug therapy and did not consume alcohol regularly. Physical and oral examination revealed a well demarcated, palpable, hard elastic mass measuring approximately 4cmx3 cm, located in the submucosal layer of the right cheek. The overlying mucosa appeared clinically normal and was not ulcerated. A CT scan revealed a homogenous mass with well-defined margins and no evidence of maxillary sinus infiltration or bone resorption (Figures 2(a) and 2(b)).

The patient underwent an excisional biopsy (Figure 3). Microscopic examination revealed uniform spindle-shaped cells with elongated nuclei, eosinophilic cytoplasm; several blood vessels lined with a thin layer of endothelial cells were embedded within the lesion (Figure 4(a)). Immunohistochemical stains with monoclonal antibodies against actins, desmin, vimentin, cytokeratins AE1/AE3, CD34, and the S100 protein (Figure 4(b)) were performed. Stains for vimentin, desmin, muscle-specific actin, and smooth muscle actin were positive while stains for cytokeratins AE1/AE3, EMA, S100, and CD34 were all negative in the tumor cells. The histopathological diagnosis was leiomyoma of the cheek. The follow-up examinations over 2 years after the procedure showed no evidence of recurrence (Figures 5(a), 5(b), and 6).

3. Discussion

Baden et al. described leiomyomas as a benign neoplasm that arises in the smooth muscle [18, 19]. As reported in several previous reports [18-28], leiomyomas can be found in areas with an abundance of smooth muscle, such as the uterine or gastrointestinal tracts and the dermis. Given the lack of smooth muscle in the oral cavity, in particular in the cheek, palate, or tongue, the only available substrates in this area would be blood vessels, circumvallate papillae, and heterotopic smooth muscle [20]. The pathogenesis of leiomyoma in the oral cavity is unclear, although oral trauma, venous stasis, hormonal changes, and genetic alterations can be implicated [20,21].

Previous studies [22-24] have reported leiomyomas of the oral cavity primarily in the lips, followed by the palate, the buccal space, the mandible, the tongue, and the gingiva, while the cheek is the least frequent site of leiomyomas in the oral cavity [25, 26]. Given this, the case of leiomyoma reported here in the cheek of a 43-year-old female can be considered to be relatively rare. The exceptional nature of the present case is reinforced by the fact that most cases of leiomyoma in the oral cavity are relatively small, that is, typically less than 2 cm in diameter, nodular, and slowdeveloping. The cheek leiomyoma described in the present study was larger than any benign smooth muscle tumor reported previously [27,28]. Clinically, it is difficult to distinguish leiomyomas of the cheek from other lesions or tumors of the oral cavity, such as pleomorphic adenoma, lymphangioma, fibroma, lipoma, or pyogenic granuloma [20-24]. However, angioleiomyomas, unlike leiomyomas, appear reddish blue in color and are soft and compressible on palpation. The benign characteristics of leiomyomas include the absence of mitoses and necrosis, as well as cellular atypia and pleomorphism in histopathological evaluation [25-28]. The diagnosis of leiomyoma is based on positivity for desmin, smooth muscle actin, and muscle-specific actin and negativity for the S100 protein [28].

Leiomyomas recur only very rarely, although prognosis depends on the completeness of the surgical excision of the tumor. In one case, an angioleiomyoma was excised from the region of the hard palate, but 9 weeks after the first procedure, the authors noted the recurrence [27]. In the second case of an angioleiomyoma in the region of the hard palate, recurrence was noted two weeks after the first excision [28].

4. Conclusion

A leiomyoma of the cheek is a rare benign neoplasm. We report a case of a leiomyoma located in the buccal mucosa of the oral cavity. Tumors of this type are asymptomatic and may develop for months or even years. The diagnosis of this lesion requires histopathological evaluation.


Appropriate consent was obtained to include images of the patient in the publication.

Conflicts of Interest

The authors declare having no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.


[1] A. Agaimy, M. Michal, L. D. R. Thompson, and M. Michal, "Angioleiomyoma of the sinonasal tract: analysis of 16 cases and review of the literature," Head and Neck Pathology, vol. 9, no. 4, pp. 463-473, 2015.

[2] S. Y. Rawal and Y. B. Rawal, "Angioleiomyoma (vascular leiomyoma) of the oral cavity," Head and Neck Pathology, vol. 12, no. 1, pp. 123-126, 2018.

[3] S. Ishikawa, S. Fuyama, T. Kobayashi, Y. Taira, A. Sugano, and M. Iino, "Angioleiomyoma of the tongue: a case report and review of the literature," Odontology, vol. 104, no. 1, pp. 119-122, 2016.

[4] H. Osano, Y. Ioka, R. Okamoto et al., "Angioleiomyoma of the cheek: a case report," Journal of Oral Science, vol. 57, no. 1, pp. 63-66, 2015.

[5] T. Tsuji, K. Satoh, H. Nakano, and M. Kogo, "Clinical characteristics of angioleiomyoma of the hard palate: report of a case and an analysis of the reported cases," Journal of Oral and Maxillofacial Surgery, vol. 72, no. 5, pp. 920-926, 2014.

[6] E. E. Wallach and N. F. Vlahos, "Uterine myomas: an overview of development, clinical features, and management," Obstetrics and Gynecology, vol. 104, no. 2, pp. 393-406, 2004.

[7] M. Payson, P. Leppert, and J. Segars, "Epidemiology of myomas," Obstetrics and Gynecology Clinics of North America, vol. 33, no. 1, pp. 1-11, 2006.

[8] E. Faerstein, M. Szklo, and N. Rosenshein, "Risk factors for uterine leiomyoma: a practice-based case-control study. I. African-American heritage, reproductive history, body size, and smoking," American Journal of Epidemiology, vol. 153, no. 1, pp. 1-10, 2001.

[9] A. P. da Silva, A. de Mello, E. R. Dos Santos, S. T. Paz, C. L. B. Cavalcanti, and M. R. de Melo-Junior, "Histopathological and digital morphometrical evaluation of uterine leiomyoma in Brazilian women," Obstetrics and Gynecology International, vol. 2016, Article ID 2968410, 5 pages, 2016.

[10] J. K. Brooks, N. G. Nikitakis, N. J. Goodman, and B. A. Levy, "Clinicopathologic characterization of oral angioleiomyomas," Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontics, vol. 94, no. 2, pp. 221-227, 2002.

[11] R. Meher and S. Varshney, "Leiomyoma of the nose," Singapore Medical Journal, vol. 48, no. 10, pp. 275-276, 2007.

[12] Y. Liu, B. Li, L. Li, Y. Liu, C. Wang, and L. Zha, "Angioleiomyomas in the head and neck: a retrospective clinical and immunohistochemical analysis," Oncology Letters, vol. 8, no. 1, pp. 241-247, 2014.

[13] K. C. Dutt, S. Bindra, M. Awana, M. Talwar, and G. Lehl, "Intraosseous leiomyoma of the mandible: a case report of the rare entity and review of literature," Journal of Maxillofacial and Oral Surgery, vol. 16, no. 2, pp. 145-151, 2017.

[14] S. Gianluca, R. Marini, F. Tonoli, and M. P. Cristalli, "Leiomyoma of oral cavity: case report and literature review," Annali di Stomatologia, vol. 2, no. 1-2, pp. 9-12, 2011.

[15] B. W. Neville, D. D. Damm, C. M. Allen, and A. Chi, Oral and Maxillofacial Pathology, Saunders Elsevier, St. Louis, 4th ed. edition, 2015.

[16] A. K. El-Naggar, J. R. Grandis, T. Takata, J. Grandis, and P. Slootweg, WHO Classification of Head and Neck Tumours, IARC, Lyon, 4th ed. edition, 2017.

[17] L. Barnes, "Tumors and tumor like lesions of soft tissue," in Surgical Pathology of Head and Neck, L. Barnes, Ed., Marcel Dekker, New York, 2nd ed. edition, 2001.

[18] E. Baden, J. L. Doyle, and D. A. Lederman, "Leiomyoma of the oral cavity: a light microscopic and immunohistochemical study with review of the literature from 1884 to 1992," European Journal of Cancer Part B: Oral Oncology, vol. 30, no. 1, pp. 1-7, 1994.

[19] M. Veeresh, M. Sudhakara, G. Girish, and C. Naik, "Leiomyoma: a rare tumor in the head and neck and oral cavity: report of 3 cases with review," Journal of Oral and Maxillofacial Pathology, vol. 17, no. 2, pp. 281-287, 2013.

[20] T. Hachisuga, H. Hashimoto, and M. Enjoji, "Angioleiomyoma. A clinicopathologic reappraisal of562 cases," Cancer, vol. 54, no. 1, pp. 126-130, 1984.

[21] T. D. Zijlker and R. Visser, "A vascular leiomyoma of the ethmoid. Report of case," Rhinology, vol. 27, no. 2, pp. 129-135, 1989.

[22] J. P. Harcourt and A. P. Gallimore, "Leiomyoma of the paranasal sinuses," The Journal of Laryngology and Otology, vol. 107, no. 8, pp. 740-741, 1993.

[23] L. Ardekian, N. Samet, Y. P. Talmi, Y. Roth, E. Bendet, and J. Kronenberg, "Vascular leiomyoma of the nasal septum," Otolaryngology and Head and Neck Surgery, vol. 114, no. 6, pp. 798-800, 1996.

[24] Y. Sawada, "Angioleiomyoma of the nasal cavity," Journal of Oral and Maxillofacial Surgery, vol. 48, no. 10, pp. 1100-1101, 1990.

[25] G. Marioni, R. Marchese-Ragona, S. Fernandez, J. Bruzon, F. Marino, and A. Staffieri, "Progesterone receptor expression in angioleiomyoma of the nasal cavity," Acta Oto-Laryngologica, vol. 122, no. 4, pp. 408-412, 2002.

[26] M. H. Khan, A. S. Jones, and M. T. Haqqani, "Angioleiomyoma of the nasal cavity report of a case and review of the literature," The Journal of Laryngology and Otology, vol. 108, no. 3, pp. 244-246, 1994.

[27] J. R. Natiella, M. E. Neiders, and G. W. Greene, "Oral leiomyoma: report of six cases and a review of the literature," Journal of Oral Pathology, vol. 11, no. 5, pp. 353-365, 1982.

[28] T. J. Svane, B. R. Smith, B. J. Cosentino, E. J. Cundiff, and J. J. Ceravolo Jr., "Oral leiomyomas: review of the literature and report of a case of palatal angioleiomyoma," Journal of Periodontology, vol. 57, no. 7, pp. 433-435, 1986.

Ricardo Roberto de Souza Fonseca [ID], (1) Mario Augusto Ramos Junior, (2) Douglas Baruchi, (3) Tabata Resque Beckmann Carvalho, (4) Andresa Borges Soares, (3) Silvio Augusto Fernandes de Menezes [ID], (5) Joao de Jesus Viana Pinheiro [ID], (6) and Jorge Nazareno Ferreira Fadul (2)

(1) Laboratory of Virology, Institute of Biological Sciences, Federal University of Para, Belem, PA, Brazil

(2) Department of Oral Pathology, Barros Barreto Hospital, Belem, PA, Brazil

(3) Department of Oral Pathology, Sao Leopoldo Mandic Institute and Research Center, Campinas, SP, Brazil

(4) Department of Periodontology, University Center of State of Para, Belem, PA, Brazil

(5) Department of Periodontology, Para State University, Belem, PA, Brazil

(6) Department of Oral Pathology, Federal University of Para, Belem, PA, Brazil

Correspondence should be addressed to Joao de Jesus Viana Pinheiro;

Received 17 July 2019; Accepted 3 December 2019; Published 20 February 2020

Academic Editor: Maria M. Picken

Caption: Figure 1: (a, b) Clinical aspect of the lesion, showing the large size of the growth.

Caption: Figure 2: (a) Axial plane and (b) coronal plane, showing the lesion as a dense mass of soft tissue with defined contours and foci of calcification.

Caption: Figure 3: Macroscopic aspect of the leiomyoma.

Caption: Figure 4: (a) Proliferation of spindle cells arranged in interlocking bundles in a monotonous pattern of fascicles (hematoxylin and eosin stain; original magnification 20x). (b) Immunohistochemical stain showing strong positivity for desmin (original magnification 20x).

Caption: Figure 5: (a) Axial plane and (b) coronal plane, showing the postoperative aspects of the affected region.

Caption: Figure 6: Two-year follow-up with no signs of recurrence.
COPYRIGHT 2020 Hindawi Limited
No portion of this article can be reproduced without the express written permission from the copyright holder.
Copyright 2020 Gale, Cengage Learning. All rights reserved.

Article Details
Printer friendly Cite/link Email Feedback
Title Annotation:Case Report
Author:Fonseca, Ricardo Roberto de Souza; Ramos, Mario Augusto, Jr.; Baruchi, Douglas; Carvalho, Tabata Res
Publication:Case Reports in Pathology
Geographic Code:3BRAZ
Date:Mar 31, 2020
Previous Article:Vulval Intestinal/Enteric Heterotopia in a Patient with Crohn's Disease.
Next Article:Metastatic Renal Cell Carcinoma to the Testis: A Clinicopathologic Analysis of Five Cases.

Terms of use | Privacy policy | Copyright © 2022 Farlex, Inc. | Feedback | For webmasters |