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Case Report


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Case Report

Clinical, Imaging and Histopathology of Angioleiomyoma of the Buccal Cheek

Mohammad Jafarian ,


Fatemeh Mashhadi Abbas ,


Mitra Ghazizadeh Ahsaie ,


and Homeira Saebnoori


1Department of Oral and Maxillofacial Surgery, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran

2Department of Oral and Maxillofacial Pathology, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran

3Department of Oral and Maxillofacial Radiology, School of Dentistry, Shahid Beheshti University of Medical Sciences, Tehran, Iran Correspondence should be addressed to Mitra Ghazizadeh Ahsaie; mitraghazizadeh@gmail.com

Received 6 March 2021; Accepted 22 April 2021; Published 30 April 2021 Academic Editor: Giovanni Mergoni

Copyright © 2021 Mohammad Jafarian et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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.

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 com- monly 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 pal- ate, tongue, cheek, gingiva, retromolar area, and submandib- ular 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]. Clin- ically, 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 com- plete imaging assessment including color Doppler ultraso- nography, 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 his- tory 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, compress- ible solid mass was detected with moderate attachment to the adjacent tissues. The estimated size of the lesion was 1:5 ∗ 1 cm, 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, mul- tidetector computed tomography (MDCT), and MRI. The



results of ultrasonography indicated a well-de fined hypoe- choic heterogeneous mass lying beneath the skin measuring 13 ∗ 18 mm (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 histopatho- logical 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 fasci-

cle pattern next to the blood vessels (Figure 6(b)). The mixed in flammatory infiltration and hyalinized area intermixed with adipose tissue were also found. In the IHC staining anal- ysis, the specimen was positive for both SMA (smooth mus- cle 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.

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 com- mon sites of oral angiomyomas were the lip [2, 4, 12–15], cheek or buccal mucosa [5, 8, 14, 16–18], palate [7, 19–21],

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.


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 pain- less swelling. Only three cases reported pain associated with the lesion [7, 21, 24].

Preoperative radiologic assessments varied among the studies, from no radiographic assessments and plain radiog- raphy to ultrasound, MRI, and CT evaluations. To our knowledge, this is the first case report of angioleiomyoma



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.

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.


(a) (b)

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)

(c) (d)

Figure 7: (a) ×40 and (b) ×100 positive IHC staining for SMA. (c) ×40 and (d) ×100 positive for desmin.


Table1:Reviewofvariousstudiesonangioleiomyomaofheadandneck.cm:centimeter;CT:computedtomography;D:day;F:female;H&E:hematoxylinandeosin;IHC: immunohistochemistry;M:male;Mo:month;mm:millimeter;MRI:magneticresonanceimaging;SMA:smoothmuscleactin;Y:year;YO:yearsold. NoAuthor& yearAge&sexLocationSizeSign& symptomsRadiologicassessmentsTreatment planAnesthesiaHistologicassessmentsFollow-up 1Mehta etal.,2020 [15]57YO,MLowerlip1×1cmExophytic nodular swelling—Surgical excisionLocal

H&E: Largevascularchannelsof varyingcaliber,surrounded bythickwallsofirregularly arrangedspindle-shaped cells.Whorledbundlesof musclefiberswereseenfused withthevesselwalls IHC: SMA+

3Mo,no recurrence 2Giudice etal.,2019 [32]17YO,FCheek1:5×1:0cmSwelling—Surgical excisionLocal

H&E: Uniformspindlesmooth musclecellswithpale eosinophiliccytoplasm,low amountoffibrousconnective tissue,anddistributedaround numeroussmalltortuous “slit-like”vessels IHC: SMA+ CD34+ Desmin+ Vimentin+

1week,4 weeks,6 months No recurrence 3Pandya etal.,2019 [23]46YO,FPalate1:1:0:5cmExophytic growth,painOcclusalplainfilmSurgical excisionLocal

H&E: Numerousthick-walled bloodvesselsinthe connectivetissueformedof hyperplasticsmoothmuscle fibers IHC: SMA+

6Mo,no recurrence 4Perićetal., 2019[4]36YO,MLip5×3mmPainless swelling—Surgical excisionLocal

H&E: Numerousthick-walled vessel IHC: CD34+ SMA+

7D, normal healing


Table1:Continued. NoAuthor& yearAge&sexLocationSizeSign& symptomsRadiologicassessmentsTreatment planAnesthesiaHistologicassessmentsFollow-up 5Ribeiro etal.,2019 [18]60YO,MAnterior maxillarylabial fold—Swelling—Surgical excision—

H&E: Multiplevessels,thickened muscularlayer IHC: CD34+ SMA+

2Y,no recurrence 6Ribeiro etal.,2019 [18]33YO,MBuccalmucosaSubmucosal nodule

ColorDoppler ultrasonography:well-limited hyperechoicareabetween cutaneousandmuscular layers,nochangeinblood flow Surgical excision

H&E: Multiplevascularspacesof varioussizesandcalibers, sometimescongestedand interconnected IHC: CD34- SMA+ 7

S.Y.Rawal andY.B. Rawal, 2018[22]

70YO,MHardpalate2:0×1:5cmPainlessmass—Surgical excision—

H&E: Brightlyeosinophilic, spindle-shapedcells IHC: SMA+

— 8Hassona etal.,2017 [14]52YO,FUpperlip—Painlessmass—Surgical excisionLocal

H&E: Vascularchannels surroundedbyfasciclesof concentricallyarranged spindlecellswitheosinophilic cytoplasm IHC: SMA+

12Mo,no recurrence 9Osano etal.,2015 [5]45YO,MCheek22×15×11mmPainlessmass

MRI: T1+contrast:homogeneously enhancedmassandavessel leadingtothetumor T2: Uniformsignaland hyperintensesignal Surgical excisionGeneral

H&E: Proliferationofvesselswitha smoothmusclewalland vesselssurroundedby smoothmusclecells IHC: SMA+ Desmin+ Vimentin+

18Mo,no recurrence


Table1:Continued. NoAuthor& yearAge&sexLocationSizeSign& symptomsRadiologicassessmentsTreatment planAnesthesiaHistologicassessmentsFollow-up 10Ishikawa etal.,2014 [6]51YO,MTongue11mmNodularmass—Surgical excision—

H&E: Brightlyeosinophilic, spindle-shapedcells IHC: SMA+ HHF-35actin+ Vimentin+ Desmin+ S100- CD34-

8Mo,no recurrence 11Tsujietal., 2014[7]79YO,MHardpalate1:5×1:5cmPainless swelling

Panoramic: Nosourceofodontogenic infection CT: Well-circumscribedmass,no evidenceofsurroundingbone resorption

Surgical removal and curettage


H&E: Vascularspacesand redundantsmoothmuscle IHC: Desmin+ SMA+

6Mo,no recurrence 12Eleyetal., 2012[21]39YO,MHardpalate2cmPainless swelling

MRI: T1: Slightlyhigherthanthe surroundingsofttissue T2: Markedhyperintensity Surgical excisionGeneral H&E: Vascularchannels surroundedbyfasciclesof spindlecells IHC: Desmin+ SMA+

3Mo,no recurrence 13Minni etal.,2012 [11]54YO,FSubmandibular space3×3cmPainless swelling

MRI: Displacementof submandibulargland,tongue, andoropharynxairway Compressingtherighttonsil Ultrasound-guidedFNA: Blood-stainedaspirate Complete surgical removalGeneral H&E: Smoothmuscletissue punctuatedwiththick-walled vessels,capillary,andvenous- typevessels IHC: SMA+

6Mo,no recurrence 14Menditti etal.,2012 [10]14YO,MGingiva1-2cmPainless swelling—

Radical excision with2mm free margins


H&E: Vascularspacesand redundantsmoothmuscle IHC: SMA+

1Y,no recurrence 15Gueiros etal.,2011 [16]

-54YO,M -66YO,M -53YO,M Lowerlip Upperlip Upperlip−1×1cm −0:8×0:5×0:5cmPainlessnodule—Surgical excisionLocal H&E: Spindle-shapedcellsand bloodvessels IHC: MSA+

-2Y -1Y -6Mo,no recurrence


Table1:Continued. NoAuthor& yearAge&sexLocationSizeSign& symptomsRadiologicassessmentsTreatment planAnesthesiaHistologicassessmentsFollow-up 16Vidaković etal.,2011 [8]58YO,MCheek2×1:5cmPainless swelling—Surgical excisionLocal H&E: Thick-walledvesselswith partiallypatentandsmooth musclefibres

7D, normal healingof wound 17Kimetal., 2010[3]2YO,MTongue2:5×2cmSofttissue mass

MRI: Elevatedmassonthebaseof thetongue T1: Isointensesignaltomuscle T2: Slightlyhyperintensesignal withhyperintenserim Surgical removalGeneral H&E: Prominentandthickened vesselwallsconsistingof benignandmaturesmooth musclecells IHC: Desmin+ SMA+

26Mo,no recurrence 18Nonaka etal.,2010 [25]38YO,MTongue2cmPainless exophyticmass—Surgical excision—

H&E: Proliferationofspindle- shapednucleuscells, numerousbloodvessels IHC: SMA+

1Y,no recurrence 19Keerthi etal.,2009 [17]32YO,MCheek4:5×4cmPainless swelling

CT: Largenonhomogenously enhancingmixeddensity lesionintheinfratemporal fossaandthebuccalspace, moderatelyenhancingsoft tissuecomponent superolaterally,withno calcificationornecrosis Surgical removalGeneral

H&E: Multiplebloodvesselswith proliferationofsmooth muscle

6Y,no recurrence 20Keerthi etal.,2009 [17]32YO,MCheek3×3cmPainless swelling

Plainfilmradiograph: Nobonychanges Ultrasound: Hypoechoiclesionmorein favorofsofttissueswelling Surgical removalGeneral H&E: Spindle-shapedfasciclesand manybloodvessels IHC: Massontrichromestain(+)

1Y,no occurrence 21McParland etal.,2009 [19]42YO,MBuccalmucosal3×3cmPainless swelling—Surgical excisionLocal

H&E: Smoothmusclebandsare surroundedbyslit-like vascularspaces 6Mo,no recurrence


Table1:Continued. NoAuthor& yearAge&sexLocationSizeSign& symptomsRadiologicassessmentsTreatment planAnesthesiaHistologicassessmentsFollow-up 22Cepeda etal.,2008 [2]

39YO,F 27YO,F 43YO,F 36YO,M 48YO,M

Retromolar area Mandibular region Lowerlip Upperlip Upperlip

0:9×0:6×1cm 1:4×1:3×1cm 0:0:0:4cm 1:1:1:0cm 1:1:1:0cm


Routineradiographic inspection: Aunilocularradiolucency locatedinamandibular region(incase2) Surgical excision—

H&E: Severalbloodvesselslinedby athinlayerofendothelial cellswereobserved intercalatedinthefascicules IHC: SMA+ Vimentin+ Desmin+ CD34- 23Manor etal.,2007 [20]39YO,MBuccalvestibule8Asymptomatic mass

CT: Hypervascularsofttissue mass,withnoinvasionofthe periosteumorbone Surgical excision—

H&E: Bandsofsmoothmusclecells surroundingmultiple vascularspacesofvarying size IHC: SMA+

12Mo,no recurrence 24Ideetal., 2003[26]40YO,FSubmandibular gland2:1:1:5cmPainful swelling—Surgical excision

H&E: Tortuousthick-walledvessels invaryingsizes,proliferation ofsmoothmuscle IHC: SMA+ CD31– CD34– S-100- 25Toidaetal., 2000[12]10YO,MLowerlip2×1:5cmPainfulmass—Surgical excisionLocal H&E: Spindlecellssurroundedby numerousslit-likevessels IHC: SMA+ S-100-

3Y,no recurrence 26Simon etal.,2000 [9]59YO,FSubmandibular triangle15×13mmNontender mass

ColorDopplerultrasound: Well-circumscribed, homogeneoussolidmass attachedtotheposterior surfaceofthesubmandibular gland,vascularitywithinthe mass Surgical excisionGeneral H&E: Smoothmusclepunctuated withthick-walledvenous vascularchannels—


with a thorough imaging assessment including color Doppler sonography, MDCT, and MRI. The lesion was slightly het- erogeneous 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 heman- giomatous lesions [27]. In this case, di fferential 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 neuro fibroma and vascular lesions including arteriovenous malformation, lymphangioma, and hemangioma. However, these lesions may have di fferent radiographic and imaging characteristics. According to previous studies, the intraoss- eous 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 back- ground of smooth muscle proliferation. Having numerous blood vessels may pretend other benign vascular tumors such as hemangioma, hemangiopericytoma, hemangioendothe- lioma, vascular malformation, and other neurovascular hamartomas [29], but a de finite examination could rule out these lesions because of the smooth muscle background.

Immunohistochemically markers like SMA and MSA (mus- cle-specific actin) can be useful in identification of smooth muscle nature of the cellular stroma [15, 30, 31]. In the pres- ent 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 di fferential diagnosis histo- pathologically is leiomyosarcoma if there were more cellular pleomorphism and mitosis figures. In addition to well cir- cumscribing 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 subman- dibular area [9, 11, 24]. Other areas such as the gingiva, man- dible, 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].

Data Availability

The data used to support the findings of this study are avail- able from the corresponding author upon request.

Conflicts of Interest

The authors declare no conflict of interest.


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