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Leiomyoma of Oral Cavity: Case Report and Literature Review/Oral Kavitede Leiomyom: Vaka Sunumu ve Literatur Taramasi.

Abstract

Leiomyoma is a benign smooth muscle tumor that can be observed at any region but is mostly observed in the female genital tract. Although leiomyomas are uncommon in the oral cavity, they are usually observed on the lips, tongue, and palate. Here we present a case report of leiomyoma in a female patient along with the literature review. A 35-year-old Turkish female presented with a 1-year history of a well-delineated, asymptomatic, and soft mass on the vestibule alveolar mucosa of the right mandibular molar region. Under local anesthesia, excisional biopsy was performed and the specimen was sent for histopathological examination. The histopathological diagnosis was leiomyoma. Based on clinical appearance, it is very challenging to distinguish a leiomyoma from other mesenchymal-origin tumors. Histological analysis determined the final diagnosis of oral leiomyoma, and surgery was performed because it was the only option for the treatment of these lesions.

Keywords: Leiomyoma, benign tumor, oral cavity

Oz

Leiomyoma en sik kadin genital sisteminde olmak uzere herhangi bir bolgede bulunabilen benign duz kas tumorudur. Leiomyomalar oral kavitede yaygin olmamakla beraber dil, dudak ve palatinal mukozada izlenebilir. Bu olgu sunumunun amaci leiomyoma saptanan bir bayan hastayi sunarak literatur taranmasidir. 35 yasinda bir bayan hastada sag mandibular molar bolgede vestibul alveol mukozasinda yaklasik bir yildir sinirli, asemptomatik yumusak doku kitlesi mevcut oldugu saptanmistir. Lokal anestezi ile eksizyonel biyopsi uygulanmis ve alinan ornek histopatolojik incelemeye gonderilmistir. Histopatolojik tani leiomyoma idi. Leiomyomayi diger mezensimal kaynakli tumorlerden klinik olarak ayirmak oldukca zordur. Kesin tani histolojik muayene ile olmali ve bu lezyonlarin tedavisi cerrahi olarak yapilmalidir.

Anahtar Kelimeler: Leiomyom, benign tumor, agiz kavitesi

INTRODUCTION

Leiomyomas are benign smooth muscle tumors that are frequent in the uterus, skin, and gastrointestinal tract. These can be attributed to only 0.42% of all soft tissue lesions reported in the oral cavity, caused by the lack of smooth muscle in this region. These may be observed in any age group, but the greatest prevalence of head and neck leiomyomas is seen between 40 and 49 years of age. Leiomyomas in the oral cavity appear as asymptomatic, slow-growing, submucosal masses, which are usually located on the hard palate, tongue, lips, or buccal mucosa. They are rarely observed on the floor of the mouth and gingiva (1-4).

Histologically, three types of leiomyomas are present: epithelioid leiomyoma, solid -form leiomyoma, and vascular leiomyoma (angiomyomas or angioleiomyomas) (5, 6). Solid leiomyomas are well-delineated tumors that are are introduced as connected bundles of spindle-shaped smooth muscle cells. The nuclei are elongated, pale staining, and blunt ended, and mitotic figures are rare in lesions. Because of hyperplasia of their smooth muscle coats, well-delineated vascular leiomyomas have multiple blood vessels with thickened walls (7, 8). The most recognized variant followed by solid leiomyomas in the oral cavity is vascular or angiomyoma.

Although it is mainly diagnosed by histological examination of hematoxylin and eosin (H&E)-stained tissue sections, microscopic diagnosis may sometimes be difficult because of the fact that spindle cell proliferation shares many similarities with schwannoma, neurofibroma, fibromatosis, and myofibroma. Therefore, special stains that identify collagen may be helpful in distinguishing these lesions. Immunhistochemical demonstration of actins can confirm the diagnosis (9-12).

Surgical excision of lesions appears to be the best choice of treatment. Moreover, recurrence of the lesion is unexpected (1, 2). Here we present a case report of leiomyoma in a female patient along with the literature review.

CASE PRESENTATION

A 35-year-old Turkish female presented with a 1-year history of a well-delineated asymptomatic mobile mass on the lingual alveolar mucosa of the right mandibular molar region. The medical and family history was unremarkable. Intraoral examination revealed a compressible, soft, mucosal colored mass (Figure 1). Under local anesthetics, an excisional biopsy was performed, and the specimen was sent for histopathological examination at the Department of Oncologic Cytology and Tumor Pathology, Institute of Oncology, Istanbul University (Figure 2a, b). This study was performed according to the guidelines of the Declaration of Helsinki concerning ethical principles for medical research involving human subjects, and written informed consent was obtained from the patient.

Histopathological features were as follows: the surface epithelium displayed a normal maturation pattern and a well-delineated tumor constituting of spindle-shaped smooth muscle cells. These cells had elongated, fusiform nuclei (Figure 3, 4). In addition, the cells were aligned in sheets of well-delineated cell outlines. Mitotic figures were absent. Immunohistochemical stains for desmin and smooth muscle actin (SMA) revealed positivity within the lesional cells (Figure 5, 6) and a low Ki-67 percentage in tumor cells (Figure 7). The histopathological diagnosis was leiomyoma. At 5-month follow-up, there was no recurrence.

DISCUSSION

Oral leiomyomas are rare benign tumors of the oral cavity that are hypothesized to originate from excretory ducts of salivary glands and vascular smooth muscles (5). There are histological variations (clear cell, epithelioid, granular cell, and myxoid changes) and secondary degenerative changes (hyaline degeneration, cystic change, myxoid degeneration, infection, necrosis, and calcification) in leiomyoma of the oral cavity and head and neck region. It is thought that these changes result from deficient blood supply, leading to a replacement of muscle fibers by collagen, hyaline material, calcium, mucopolysaccharide, or a combination of these factors (13, 14).

The World Health Organization classifies leiomyoma in soft tissue tumors (WHO 2002) that arise from smooth muscle. Three types of leiomyoma are categorized by WHO. The most frequent form of leiomyoma is angiomyoma (74%), which is followed by solid leiomyomas (25%). Only one case of epitheloid leiomyoma has been reported in the literature (9-11).

Oral leiomyomas represent 0.4% of all soft tissue lesions in the oral cavity. The most frequently affected area is the lips (27.5%), followed in a descending order by the tongue (18.3%), buccal mucosa (15.5%), palate (15.5%), and gingiva (8.5%) (1-3, 7, 15). In this Case, the site was vestibular mucosa of the right mandibular molar region.

Histological examination of the case revealed that the tumor was in a solid form and it was a rare type of tumor for the oral cavity. Compared with the vascular type, solid leiomyomas are smaller. Histologically, solid leiomyoma contains interlaced bundles of spindle-shaped or stellate smooth muscle cells with blunt-ended, elongated, pale-staining nuclei: lesions are rather cellular; yet, the packs of tumor cells are separated by collagenic strands. All the histological characteristics of the lesion were observed in our histological examination (9).

Immunohistochemical demonstration of actins can confirm the diagnosis. Actin is a small cytoplasmic filament that has contractile properties. The six actin isotypes differentiate between striated muscle, smooth muscle, and nonmuscle cells. Antismooth muscle actin generally provides good intensity and sensitivity for the detection of leiomyoma. Staining for desmin is less reliable because it is positive in about two -thirds of cases. In our case, immunohistochemical analysis demonstrated the expression of smooth muscle actin and desmin within the tumor cells.

The absolute treatment for benign smooth muscle tumors is mainly surgical excision. Surgery has been performed as the only treatment option in all leiomyomas reported in the literature (1-3). There are no reports of lesion recurrence after total excision, and this indicates the need for cautious and complete excision of the lesion. Under local anesthesia, total excision of the mass was performed in our case. At 5-months follow-up, the patient did not present with any complications and recurrences.

CONCLUSION

The characteristics of leiomyoma may be similar to those of various lesions in clinical and histological examinations. Therefore, dental practitioners and pathologists should be aware of differential diagnosis.

Informed Consent: Written informed consent was obtained from patient who participated in this study.

Peer-review: Externally peer-reviewed.

Author contributions: Concept - F.N.P.; Design - F.N.P.; Supervision - F.N.P.; Resource - O.O.; Materials - E.I.; Data Collection and/or Processing - F.N.P; Analysis and/or Interpretation - V.O.; Literature Search - O.O., E.I.; Writing - F.N.P; Critical Reviews - V.O.

Conflict of Interest: No conflict of interest was declared by the authors.

Financial Disclosure: The authors declared that this study has received no financial support.

Hasta Onami: Yazili hasta onami bu calismaya katilan hastadan alinmistir.

Hakem Degerlendirmesi: Dis Bagimsiz.

Yazar Katkilari: Fikir - F.N.P.; Tasarim - F.N.P.; Denetleme - F.N.P.; Kaynaklar O.O.; Malzemeler - E.I.; Veri Toplanmasi ve/veya islemesi - F.N.P.; Analiz ve/veya Yorum - V.O.; Literatur taramasi - O.O., E.I.; Yaziyi Yazan - F.N.P.; Elestirel Inceleme - V.O.

Cikar Catismasi: Yazarlar cikar catismasi bildirmemislerdir.

Finansal Destek: Yazarlar bu calisma icin finansal destek almadiklarini beyan etmislerdir.

REFERENCES

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(2.) Lloria-Benet M, Bagan JV, Lloria de Miguel E, Borja-Morant A, Alonso S. Oral leiomyoma: a case report. Med Oral 2003; 8: 215-9.

(3.) Gianluca S, Marini R, Tonoli F, Cristalli MP. Leiomyoma of oral cavity: Case report and literature review. Ann Stomatol 2011; 2: 9-12.

(4.) Alvarez E, Laberry MP, Ardila CM. Multiple oral leiomyomas in an infant: a rare case. Case Rep Dent 2012; 2012: 804305. [CrossRef]

(5.) Sharma SS, Ramakrishnan K, Vijayalakshmi D, Saravanan C. Leiomyoma of oral cavity in a young child. Austin J Clin Case Rep 2015; 2: 1067.

(6.) Koca H, Guneri P, Cetingul E, Onal T. A very rare form of leiomyoma: Mandibular angioleiomyoma. Int J Pediatric Otorhinolaryngol. 2006; 1: 110-4. [CrossRef]

(7.) Yu CH, Tsai TC, HM, Chiang CP. Oral leiomyoma-case report. Chin Dent 2005; 24114-120.

(8.) Nonaka CF, Pereira KM, Miquel MC. Oral vascular leiomyoma with extensive calcification areas. Braz J Otorhinolaryngol 2010; 76: 539. [CrossRef]

(9.) Guitan Cepeda LA, Quezada Rivera D, Tenorio Rocha F, Leyva Huerta ER, Mendez Sanchez ER. Vascular leiomyoma of the oral cavity. Clinical, histopathological and immunohistochemical characteristics. Presentation of five cases and review of the literature. Med Oral Patol Oral Cir Bucal 2008; 13: E483-488.

(10.) Bruecks AK, Trotter MJ. Expression of desmin and smooth muscle myosin heavy chain in dermatofibromas. Arch Pathol Lab Med 2002; 126: 1179-83.

(11.) Zelger BG, Sidoroff A, Zelger B. Combined dermatofibroma: co-existence of two or more variant patterns in a single lesion. Histopathology 2000; 36: 529-39. [CrossRef]

(12.) Williams HK, Williams DM. Oral granular cell tumours: a histological and immunocytochemical study. J Oral Pathol Med 1997; 26: 164-9. [CrossRef]

(13.) Montague LJ, Fitzpatrick SG, Islam NM, Cohen DM, Bhattacharyya I. Extensively ossifying oral leiomyoma: A rare histologic finding. Head and Neck Pathol 2014; 8: 311-6. [CrossRef]

(14.) Vincenzi A, Rossi G, Monzani D, Longo L, Rivasi F. Atypical (bizarre) leiomyoma of the nasal cavity with prominent myxoid change. J Clin Pathol 2002; 55: 872-5. [CrossRef]

(15.) Darling MR, Wehrli B, Zeligman E, Smillie J, Daley T. Unusual benign smooth muscle lesions of the tongue: review and report of two cases. Head Neck Pathol 2012; 6: 121-4. [CrossRef]

Filiz Namdar Pekiner (1), Ozlem Sen Okumus (1), Elif Izgi (1), Vakur Olgac (2)

(1) Department of Oral Diagnosis and Radiology, Marmara University School of Dentistry, Istanbul, Turkey

(2) Department of Oncologic Cytology and Tumor Pathology, Istanbul University Institute of Oncology, Istanbul, Turkey

This study was presented at the Oral Diagnosis and Maxillofacial Radiology Society VI. National Symposium and I. International Congress, 17-19 April 2015, Izmir, Turkey.

Bu calisma Oral Diagnoz ve Maksillofasiyal Radyoloji Dernegi'nin 6.Ulusal Sempozyumu ve 1. Uluslararasi Kongresinde sunulmustur, 17-19 Nisan 2015, Izmir, Turkiye.

Correspondence Author / Sorumlu Yazar: Filiz Namdar Pekiner E-mail/E-posta: fpekiner@gmail.com

Received/Gelis Tarihi: 22.04.2016 Accepted/Kabul Tarihi: 20.07.2016 Available Online Date/Cevrimici Yayin Tarihi: 24.02.2017 DOI: 10.5152/clinexphealthsci.2017.007
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Title Annotation:Case Report / Olgu Sunumu
Author:Pekiner, Filiz Namdar; Okumus, Ozlem Sen; Izgi, Elif; Olgac, Vakur
Publication:Clinical and Experimental Health Sciences
Article Type:Case study
Geographic Code:7TURK
Date:Jun 1, 2017
Words:1939
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