International Journal of Pediatric Otorhinolaryngology Extra
Volume 1, Issue 2 , Pages 110-114, June 2006

A very rare form of leiomyoma: Mandibular angioleiomyoma

  • Hüseyin Koca

      Affiliations

    • Department of Oral and Maxillofacial Surgery, School of Dentistry, Ege University, Bornova 35100, Izmir, Turkey
    • ,
  • Pelin Güneri

      Affiliations

    • Department of Oral Diagnosis and Radiology, School of Dentistry, Ege University, Bornova 35100, Izmir, Turkey
    • Corresponding Author InformationCorresponding author. Tel.: +90 232 3881081; fax: +90 232 3880325.
    • ,
  • Erdoğan Çetingül

      Affiliations

    • Department of Oral and Maxillofacial Surgery, School of Dentistry, Ege University, Bornova 35100, Izmir, Turkey
    • ,
  • Taha Önal

      Affiliations

    • Department of Oral Pathology, School of Dentistry, Ege University, Bornova 35100, Izmir, Turkey

Received 24 October 2005; accepted 30 January 2006.

Article Outline

Summary 

Mandibular angioleiomyomas are extremely exceptional, and they may stem from the smooth muscle of vessel walls, aberrant adnexial smooth muscle, arteriovenous anastomoses, ectopic thyroglossal ducts and hamartomas. In this report, a case of angioleiomyoma in the posterior mandibular area was presented, and aetiological, clinical, radiographical, histological characteristics and treatment modalities of the lesion were discussed.

Keywords: Leiomyoma, Angioleiomyoma, Mandible

 

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1. Introduction 

Leiomyomas are benign, smooth muscle neoplasms that may emerge from aberrant adnexial smooth muscle cells [1], or their precursors in the media of blood vessels, in the muscularis layer of the gut, and in the body of the uterus [1], [2], [3], [4], [5]. According to World Health Organization, leiomyomas are classified in three histological groups [6], [7], [8], [9]: (a) vascular [angioleiomyomas], 74% of the cases; (b) solid, 25% of the cases; (c) epitheloid [leiomyoblastomas], less than 1% of the cases [8].

Leiomyomas of the oral cavity are rare due to the lack of smooth muscles within the oral cavity [4], [7], [10], and those of the mandible are extremely exceptional [1]. Among the origins of oral leiomyomas smooth muscle of vessel walls, the circumvallate papilla, and atypical arrectores pilorum muscles in the cheeks are cited [6].

The number of oral leiomyoma cases vary in the literature between 4 and 125 [1], [2], [4], [6], [10], [11]. Clinically, they are slow-growing, generally asymptomatic submucosal masses [4], [9], [10], [12], [13], [14], which can be seen at any age ranging from infancy to 76 years [9], but mostly in the 40–59 year group [4], [11], and are usually discovered when they are 1–2cm in diameter [5]. Although rarely, difficulty in chewing, swallowing and opening the mouth, a change of voice [2], and occasional periods of numbness [13] are reported. The predilection sites are lip (27.46%), tongue (18.30%), palate, and buccal mucosa (15.49%) [4], [11], [12], [16], [15]; however, every now and then, the first two sites are swapped in some reports [5], [7], [9]. The tumor has also been detected in gingiva [4], [11], [12], [15], [16], mandible [15], [16], floor of the mouth [4], [11], salivary glands [4], [17], uvula [4], zygoma [4], palate [6], [14], nasal cavity [3], [14], and maxillary tooth socket [4].

Clinically, 55.9% of the angioleiomyomas appear as red lesions; the rest are mostly gray, white or the color of normal mucosa [11]. Radiographically, leiomyoma may cause advanced bone loss [6], [12], consequently leading to tooth mobility [6].

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2. Case report 

Twelve-year-old Caucasian male was admitted to the Department of Oral Diagnosis and Radiology, School of Dentistry, Ege University with the complaint of swelling in the left retromolar region. His medical history was non-contributory, and his family had no significant medical problems. Clinical examination revealed a rubbery mass of 2cm in diameter in left mandibular third molar area on the alveolar crest, with a small ulcer on its surface (Fig. 1). The second molar was located in a vestibular position. The regional lymph nodes were normal.

Radiographic examination on the orthopantomographic film disclosed a radiolucent mass in the left retromolar area, affecting the lingual compact bone. Additionally, mandibular third molar's angulation and localization were changed due to the pressure of the mass (Fig. 2). In three dimensional computer tomographic images [3D-CT] and conventional CT, a mass extending down lingually to the mylohyoid line, compressing the lingual plate of the mandibular compact bone was observed (Fig. 3).

On 26 October 1999, an incisional biopsy was performed under local anesthesia, and a biopsy specimen that was a vascular appearing soft tissue, approximately 0.5mm in diameter was retrieved. The biopsy report affirmed that the lesion was an angioleiomyoma, and total excision of the lesion was planned. Meanwhile, after taking the biopsy specimen, the lesion had expanded laterally and lingually on the alveolar crest (Fig. 4).

The surgical intervention was performed under local anesthesia on 9 November 1999. After administration of 3% citanest-octapressin, an incision with approximately 3mm security demarcation lines was made on the vestibule of the retromolar area passing through the normal gingiva. An additional incision distal to the first molar was created, and a mucoperiosteal flap was elevated. A silk suture was used to hold and raise the mass from the alveolar crest, and its association with mandible was eradicated by curetting the mass with a raspatorium. Thus, the mass easily dissected from its bed and was removed from the bone in one single piece. A radical approach was preferred to prevent the recurrence of the lesion, and the second molar and impacted third molar teeth were also removed. The wound was closed primarily. One week after the operation, the patient was reexamined and no complications were observed.

Macroscopically, the specimen consisted of two pieces: one was a molar tooth with uncompleted roots, and a piece of tooth germ. The other was a roughly spherical soft tissue mass, 3mm×2mm×1.5mm in size, appearing encapsulated (Fig. 5). In histological examination, the specimen was thinly pseudocapsulated, composed of proliferating masses of fibrocellular tissue with many evenly spaced patent capillaries, which were scattered throughout the stroma (Fig. 6). Proliferating round cells with ovoid blunt ends in some areas, and spindle-shaped cells with elongated nuclei in others, which were characteristic of smooth muscle tumor cells were also noted (Fig. 6).

No complications were observed during the consecutive post-operative controls in 5 years; a perfect bone healing without any recurring pathoses and a flawless mucosal healing area were observed on his last ortopantomograph taken on 8 September 2005 (Fig. 7, Fig. 8).

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3. Discussion 

Among all oral leiomyomas reported, 74% are angioleiomyomas [10]. They are rare, slow-growing, dermal or subdermal tumors [8], [11], and are mostly observed at the lower extremities of middle-aged women [8]. They may stem from the smooth muscle of vessel walls, aberrant adnexial smooth muscle, arteriovenous anastomoses, ectopic thyroglossal ducts, and hamartomas [8], [10], [12]. However, the interconnection between the smooth-muscle cells, and with those of the nearby vessels indicate that this benign tumor is related to the smooth muscle of the vascular wall [8], [11]. It has been suggested that vascular leiomyomas are a stage in a continuous process of smooth muscle proliferation, and the sequence of progression is presented as hemangioma, angioma, vascular leiomyoma, leiomyoma, and solid leiomyoma [2], [6]. Microscopic diagnosis may be difficult since the spindle cell proliferation has many similarities with mucocele, lipoma, neurofibroma, schwannoma, fibroma, fibrosarcoma, myofibroblastoma, other myofibroblastic proliferations, and hemangiopericytoma [1], [4], [5], [7], [10], [12]. In previous reports, a tendency for rapid growth of oral leiomyomas which may indicate a malignant predisposition was noticed [1], [12], [18]; and doubling time, maximum tumor diameter, mitotic count, Ki-67 labelling index [representing growth fraction], p53 reactivity [indicating malignancy] were utilized to establish the exact nature of the lesion [1], [4], [10]. For oral leiomyomas, immunohistochemical staining with vimentin, desmin, α-smooth muscle actin were positive, whereas glial fibrillary acidic protein, cytokeratin, endothelin, S100, neuron-specific-enolase, myoglobin CD56 were not present [3], [7], [10], [19].

As also observed in our case, vascular leiomyomas are made of blood vessels of varying size, whose lumens also differ in patency. Throughout the tumor, two cell types are prominent: spindle shaped smooth muscle cells surrounding vascular spaces [12], [13], and polygonal endothelial cells [12]. Moreover, electronmicroscopic evaluation has showed the presence of myogenic characteristics such as subplasmalemmal dense patches, myofilaments, dense body and basal lamina [1], [12]. Both spindle-shaped and polygonal cells were demonstrated; the former having myofilaments, and the latter having endothelial nature [12].

In a previous report of mandibular myofibromatous lesion, computed tomographic scan has revealed a well-defined, highly vascularized, enhanced soft tissue mass, causing compression of the buccal plate [19]. The same appearance was observed in the radiographic examination of the lesion in the present case. It was also shown that impacted teeth may be displaced due to the pressure of the mass, as well.

Leiomyomas are surgically excised, and recurrence is unexpected [3], [14]. However, in rare instances, vascular type leiomyomas [angioleiomyomas] may reappear [2], [10]. It is reported that 20% of all smooth muscle tumors in oral cavity were malignant; therefore, signs of malignancy shall be thoroughly investigated [10]. It is reported that leiomyosarcomas have relatively bland histology, which mimic benign lesions [12], [18], and they should be considered in the differential diagnosis of any well circumscribed, hypervascular soft tissue tumor in the mouth [20]. For all cases wide surgical excision shall be performed, and careful post-operative observation shall be continued for a period of at least 5 years [10]. In this case of angioleiomyoma, no post-operative complications and no recurrence were observed in 5 years period.

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PII: S1871-4048(06)00031-1

doi:10.1016/j.pedex.2006.01.007

International Journal of Pediatric Otorhinolaryngology Extra
Volume 1, Issue 2 , Pages 110-114, June 2006

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