# Clinical, Imaging and Histopathology of Angioleiomyoma of the Buccal Cheek

Authors: Mohammad Jafarian; Fatemeh Mashhadi Abbas; Mitra Ghazizadeh Ahsaie; Homeira Saebnoori
Journal: Case Reports in Dentistry
 (2021)

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## Abstract

Angioleiomyoma is a benign neoplasia originating from vascular smooth muscle and very uncommon in the oral cavity. In this report, we present a rare case of angioleiomyoma in oral cavity in a 46-year-old female buccal cheek and discuss the clinical, histological, and immunohistochemical characteristics. As the treatment of choice is the complete excision, the lesion was excised under local anesthesia with no further complications. In addition, a brief update on other reported cases of angiomyoma in the oral cavity is further discussed.


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## Body

## 1. Introduction

Angiomyoma or angioleiomyoma (also known as vascular leiomyoma) are benign neoplasms originating from vascular smooth muscle [1, 2]. Histologically, there are subgroups in three categories: solid, venous, and cavernous [3]. They commonly occur in the extremities and female genital tract and rarely in the oral cavity [4, 5]. The most common site in the oral cavity is in the lips [1]. Other reported sites were the palate, tongue, cheek, gingiva, retromolar area, and submandibular triangle [2, 6–10]. Although the etiology is still unclear, previous studies report trauma or spontaneous development as the causes of origin in oral cavity [10, 11]. The lesion is most often detected in adults of 30 to 50 years old [3]. Clinically, it usually presents as a slow growing, painless nodule or mass of variable size; however, pain has also been reported [12]. The final treatment and diagnosis is based on surgical excision and hematoxylin and eosin (H&E) staining and immune-histochemical (IHC) assessments [1]. In this report, a rare case of angioleiomyoma in the cheek along with a complete imaging assessment including color Doppler ultrasonography, computed tomography (CT), and magnetic resonance imaging (MRI) with an update on the literature is presented.


## 2. Case Report

A 46-year-old female was referred to a private clinic of an Oral and Maxillofacial Surgeon with complaint of a painless swelling in the right side of her cheek since 6 years ago. The swelling was initially small in size and slowly enlarged, although the patients faced no tenderness in the area. The patient had diabetes mellitus and anemia. In addition, no history of previous trauma to the area was present. Extraoral examination showed no asymmetry or marked expansion on the right cheek. On intraoral examination, no clinically visible lesion was detected. The mucosa was intact with no color change. When the area was palpated, a firm, compressible solid mass was detected with moderate attachment to the adjacent tissues. The estimated size of the lesion was1.5∗1cm, and the approximate diameter was 1.5 cm. No pulsation or bruit was present. The patient had previously visited and had prior imaging including Doppler ultrasonography, multidetector computed tomography (MDCT), and MRI. The results of ultrasonography indicated a well-defined hypoechoic heterogeneous mass lying beneath the skin measuring 13∗18mm (Figure 1). The lesion had moderate vascular flow. MDCT showed a circular well-defined homogenous mass over the buccinator muscle (Figure 2). The MRI T1 sequence showed a well-defined low signal mass between the buccinator muscle and buccal fat (Figure 3(a)). The MRI T2 sequence showed that the mass is homogenous and high signal (Figure 3(b)). A soft tissue mass was suspected, and an intraoral surgical excisional biopsy was planned. Upon obtaining consent, an excisional biopsy was performed under local anesthesia and incision was made on the buccal mucosa just over the palpated mass (Figure 4). The lesion was completely excised (Figure 5). H&E staining histopathological examination demonstrated an encapsulated mass composed of fully developed cavernous angioma in a fibrous stroma and occasionally myxomatous change. The thin-wall blood vessels showed papillary projections to the lumen which some had become irregular, large in size, and even sinusoid in some areas (Figure 6(a)). The stroma contains immature spindle-shaped fibroblasts with a paralleling fascicle pattern next to the blood vessels (Figure 6(b)). The mixed inflammatory infiltration and hyalinized area intermixed with adipose tissue were also found. In the IHC staining analysis, the specimen was positive for both SMA (smooth muscle actin) (Figures 7(a) and 7(b)) and desmin (Figures 7(c) and 7(d)); however, the microscopic features in combination with IHC findings were consistent with angioleiomyoma diagnosis. A postoperative follow-up of the patient was uneventful. The area had healed with no complication, and no signs of recurrence were detected.Figure 1
Ultrasonography shows a well-defined hypoechoic heterogenous mass lying between the skin and buccinator muscle.Figure 2
Coronal MDCT image shows a circular well-defined homogenous mass in the right buccal area.Figure 3
(a) Axial T1 MRI shows a low signal homogenous mass in the right lying over the buccinator muscle and under the skin. (b) Axial T2 MRI shows the lesion as high signal and homogenous.
(a)(b)Figure 4
Intraoral incision on the buccal mucosa revealed a spherical lesion just underneath the buccinator muscle.Figure 5
Image shows gross specimen, a spherical brown-gray firm tissue, measuring 1 cm in diameter.Figure 6
(a) Thin blood vessels with papillary projections in the fibrous stroma (×40). (b) Paralleling pattern of fascicle and glomus cells around the blood vessels (×100).
(a)(b)Figure 7
(a) ×40 and (b) ×100 positive IHC staining for SMA. (c) ×40 and (d) ×100 positive for desmin.
(a)(b)(c)(d)


## 3. Discussion

Angioleiomyoma is a rare benign soft tissue neoplasm of oral cavity [7]. Although hormonal changes, trauma, and venous stasis are suggested as possible causes, the etiology is still unknown [5]. On our latest review of literature from year 2000 to 2020 using the PubMed Central search engine, 25 articles (32 cases) were retrieved (Table 1). The most common sites of oral angiomyomas were the lip [2, 4, 12–15], cheek or buccal mucosa [5, 8, 14, 16–18], palate [7, 19–21], tongue [6, 22, 23], and submandibular area [9, 11, 24]. The gingiva [10], mandible [2], retromolar area [2], and anterior maxillary labial fold [17] were each reported once among the studies. 73% of patients were male, and 27% were females, which is consistent with previous studies [25, 26]. The mean age was 43±16.57 years old (range 2 to 79). Only one study reported a congenital angiomyoma in the tongue [3]. In all reviewed studies, the patient faced a painless swelling. Only three cases reported pain associated with the lesion [7, 21, 24].Table 1
Review of various studies on angioleiomyoma of head and neck. cm: centimeter; CT: computed tomography; D: day; F: female; H&E: hematoxylin and eosin; IHC: immunohistochemistry; M: male; Mo: month; mm: millimeter; MRI: magnetic resonance imaging; SMA: smooth muscle actin; Y: year; YO: years old.
NoAuthor & yearAge & sexLocationSizeSign & symptomsRadiologic assessmentsTreatment planAnesthesiaHistologic assessmentsFollow-up1Mehta et al., 2020 [15]57 YO, MLower lip1×1cmExophytic nodular swelling—Surgical excisionLocalH&E:Large vascular channels of varying caliber, surrounded by thick walls of irregularly arranged spindle-shaped cells. Whorled bundles of muscle fibers were seen fused with the vessel wallsIHC:SMA+3 Mo, no recurrence2Giudice et al., 2019 [32]17 YO, FCheek1.5×1.0cmSwelling—Surgical excisionLocalH&E:Uniform spindle smooth muscle cells with pale eosinophilic cytoplasm, low amount of fibrous connective tissue, and distributed around numerous small tortuous “slit-like” vesselsIHC:SMA+CD34+Desmin+Vimentin+1 week, 4 weeks, 6 monthsNo recurrence3Pandya et al., 2019 [23]46 YO, FPalate1.5×1.5×0.5cmExophytic growth, painOcclusal plain filmSurgical excisionLocalH&E:Numerous thick-walled blood vessels in the connective tissue formed of hyperplastic smooth muscle fibersIHC:SMA+6 Mo, no recurrence4Perić et al., 2019 [4]36 YO, MLip5×3mmPainless swelling—Surgical excisionLocalH&E:Numerous thick-walled vesselIHC:CD34+SMA+7 D, normal healing5Ribeiro et al., 2019 [18]60 YO, MAnterior maxillary labial fold—Swelling—Surgical excision—H&E:Multiple vessels, thickened muscular layerIHC:CD34+SMA+2 Y, no recurrence6Ribeiro et al., 2019 [18]33 YO, MBuccal mucosaSubmucosal noduleColor Doppler ultrasonography: well-limited hyperechoic area between cutaneous and muscular layers, no change in blood flowSurgical excisionH&E:Multiple vascular spaces of various sizes and calibers, sometimes congested and interconnectedIHC:CD34-SMA+7S.Y. Rawal and Y.B. Rawal, 2018 [22]70 YO, MHard palate2.0×1.5cmPainless mass—Surgical excision—H&E:Brightly eosinophilic, spindle-shaped cellsIHC:SMA+—8Hassona et al., 2017 [14]52 YO, FUpper lip—Painless mass—Surgical excisionLocalH&E:Vascular channels surrounded by fascicles of concentrically arranged spindle cells with eosinophilic cytoplasmIHC:SMA+12 Mo, no recurrence9Osano et al., 2015 [5]45 YO, MCheek22×15×11mmPainless massMRI:T1+contrast: homogeneously enhanced mass and a vessel leading to the tumorT2:Uniform signal and hyperintense signalSurgical excisionGeneralH&E:Proliferation of vessels with a smooth muscle wall and vessels surrounded by smooth muscle cellsIHC:SMA+Desmin+Vimentin+18 Mo, no recurrence10Ishikawa et al., 2014 [6]51 YO, MTongue11 mmNodular mass—Surgical excision—H&E:Brightly eosinophilic, spindle-shaped cellsIHC:SMA+HHF-35 actin+Vimentin+Desmin+S100-CD34-8 Mo, no recurrence11Tsuji et al., 2014 [7]79 YO, MHard palate1.5×1.5cmPainless swellingPanoramic:No source of odontogenic infectionCT:Well-circumscribed mass, no evidence of surrounding bone resorptionSurgical removal and curettageLocalH&E:Vascular spaces and redundant smooth muscleIHC:Desmin+SMA+6 Mo, no recurrence12Eley et al., 2012 [21]39 YO, MHard palate2 cmPainless swellingMRI:T1:Slightly higher than the surrounding soft tissueT2:Marked hyper intensitySurgical excisionGeneralH&E:Vascular channels surrounded by fascicles of spindle cellsIHC:Desmin+SMA+3 Mo, no recurrence13Minni et al., 2012 [11]54 YO, FSubmandibular space3×3cmPainless swellingMRI:Displacement of submandibular gland, tongue, and oropharynx airwayCompressing the right tonsilUltrasound-guided FNA:Blood-stained aspirateComplete surgical removalGeneralH&E:Smooth muscle tissue punctuated with thick-walled vessels, capillary, and venous-type vesselsIHC:SMA+6 Mo, no recurrence14Menditti et al., 2012 [10]14 YO, MGingiva1-2 cmPainless swelling—Radical excision with 2 mm free marginsLocalH&E:Vascular spaces and redundant smooth muscleIHC:SMA+1 Y, no recurrence15Gueiros et al., 2011 [16]-54 YO, M-66 YO, M-53 YO, MLower lipUpper lipUpper lip−1×1cm−0.8×0.5×0.5cmPainless nodule—Surgical excisionLocalH&E:Spindle-shaped cells and blood vesselsIHC:MSA+-2 Y-1 Y-6 Mo, no recurrence16Vidaković et al., 2011 [8]58 YO, MCheek2×1.5cmPainless swelling—Surgical excisionLocalH&E:Thick-walled vessels with partially patent and smooth muscle fibres7 D, normal healing of wound17Kim et al., 2010 [3]2 YO, MTongue2.5×2cmSoft tissue massMRI:Elevated mass on the base of the tongueT1:Isointense signal to muscleT2:Slightly hyperintense signal with hyperintense rimSurgical removalGeneralH&E:Prominent and thickened vessel walls consisting of benign and mature smooth muscle cellsIHC:Desmin+SMA+26 Mo, no recurrence18Nonaka et al., 2010 [25]38 YO, MTongue2 cmPainless exophytic mass—Surgical excision—H&E:Proliferation of spindle-shaped nucleus cells, numerous blood vesselsIHC:SMA+1 Y, no recurrence19Keerthi et al., 2009 [17]32 YO, MCheek4.5×4cmPainless swellingCT:Large nonhomogenously enhancing mixed density lesion in the infratemporal fossa and the buccal space, moderately enhancing soft tissue component superolaterally, with no calcification or necrosisSurgical removalGeneralH&E:Multiple blood vessels with proliferation of smooth muscle6 Y, no recurrence20Keerthi et al., 2009 [17]32 YO, MCheek3×3cmPainless swellingPlain film radiograph:No bony changesUltrasound:Hypoechoic lesion more in favor of soft tissue swellingSurgical removalGeneralH&E:Spindle-shaped fascicles and many blood vesselsIHC:Masson trichrome stain (+)1 Y, no occurrence21McParland et al., 2009 [19]42 YO, MBuccal mucosal3×3cmPainless swelling—Surgical excisionLocalH&E:Smooth muscle bands are surrounded by slit-like vascular spaces6 Mo, no recurrence22Cepeda et al., 2008 [2]39 YO, F27 YO, F43 YO, F36 YO, M48 YO, MRetromolar areaMandibular regionLower lipUpper lipUpper lip0.9×0.6×1cm1.4×1.3×1cm0.7×0.5×0.4cm1.5×1.0×1.0cm1.7×1.0×1.0cmPainless massRoutine radiographic inspection:A unilocular radiolucency located in a mandibular region (in case 2)Surgical excision—H&E:Several blood vessels lined by a thin layer of endothelial cells were observed intercalated in the fasciculesIHC:SMA+Vimentin+Desmin+CD34-23Manor et al., 2007 [20]39 YO, MBuccal vestibule8Asymptomatic massCT:Hypervascular soft tissue mass, with no invasion of the periosteum or boneSurgical excision—H&E:Bands of smooth muscle cells surrounding multiple vascular spaces of varying sizeIHC:SMA+12 Mo, no recurrence24Ide et al., 2003 [26]40 YO, FSubmandibular gland2.0×1.8×1.5cmPainful swelling—Surgical excisionH&E:Tortuous thick-walled vessels in varying sizes, proliferation of smooth muscleIHC:SMA+CD31–CD34–S-100-25Toida et al., 2000 [12]10 YO, MLower lip2×1.5cmPainful mass—Surgical excisionLocalH&E:Spindle cells surrounded by numerous slit-like vesselsIHC:SMA+S-100-3 Y, no recurrence26Simon et al., 2000 [9]59 YO, FSubmandibular triangle15×13mmNontender massColor Doppler ultrasound:Well-circumscribed, homogeneous solid mass attached to the posterior surface of the submandibular gland, vascularity within the massSurgical excisionGeneralH&E:Smooth muscle punctuated with thick-walled venous vascular channels—Preoperative radiologic assessments varied among the studies, from no radiographic assessments and plain radiography to ultrasound, MRI, and CT evaluations. To our knowledge, this is the first case report of angioleiomyoma with a thorough imaging assessment including color Doppler sonography, MDCT, and MRI. The lesion was slightly heterogeneous hypoechoic mass with slight vascular flow in the color Doppler sonography; in addition, the T1-weighted and T2-weighted sequences were low and high, respectively. The high T2-weighted sequence may be seen in cysts, benign or low-grade minor salivary gland tumors, and rare hemangiomatous lesions [27]. In this case, differential diagnosis such as benign lesions of salivary gland origin was suggested in lower possibility because the lesion was located between the buccinator muscle and skin. Cystic lesions were also excluded as the sonography of the cyst is homogenous and unechoic [28]. Differential diagnosis may suggest other benign mesenchymal tumors such as fibroma, lipoma, and neurofibroma and vascular lesions including arteriovenous malformation, lymphangioma, and hemangioma. However, these lesions may have different radiographic and imaging characteristics. According to previous studies, the intraosseous angioleiomyomas are radiographically unilocular or multilocular radiolucent lesions. They can have either an ill-defined or a well-defined sclerotic border [2, 11].In this case, H&E histologic examination showed a tumor consisting of thin- and thick-walled blood vessels in a background of smooth muscle proliferation. Having numerous blood vessels may pretend other benign vascular tumors such as hemangioma, hemangiopericytoma, hemangioendothelioma, vascular malformation, and other neurovascular hamartomas [29], but a definite examination could rule out these lesions because of the smooth muscle background. Immunohistochemically markers like SMA and MSA (muscle-specific actin) can be useful in identification of smooth muscle nature of the cellular stroma [15, 30, 31]. In the present study, IHC staining was positive for both desmin and SMA. Various IHC stainings were carried out among studies (Table 1) although SMA and desmin were the most frequent. Depending on the apparent features of the blood vessels, IHC staining of endothelial cell markers such as CD34 and CD31 was not recommended. Other differential diagnosis histopathologically is leiomyosarcoma if there were more cellular pleomorphism and mitosis figures. In addition to well circumscribing of this tumor, lacking of anaplasia and bizarre cells, fewer than 5 mitoses per 20 high-power fields could rule out sarcoma.In all evaluated cases, the lesion was resected with an excisional biopsy under local or general anesthesia. There are no recurrences after resection.


## 4. Conclusion

In conclusion, we have reported a case of angioleiomyoma of the buccal cheek that resulted in a good outcome. Among various studies, the most common sites of oral angiomyomas were the lip [2, 4, 12–15, 32], cheek or buccal mucosa [5, 8, 14, 16–18], palate [7, 19–21], tongue [6, 22, 23], and submandibular area [9, 11, 24]. Other areas such as the gingiva, mandible, retromolar area, and anterior maxillary labial fold were only reported once among studies [2, 10, 17]. Due to the benign nature of this lesion, the treatment is excisional biopsy and there have been no recurrences or complications reported so far [32].



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