Case Report

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Case Report

Clinical and Immunohistochemical Features of Oral

Angioleiomyoma: A Comprehensive Review of the Literature and Report of a Case in a Young Patient

Amerigo Giudice ,

^1,2

Francesco Bennardo ,

²

Caterina Buffone,

¹

Ylenia Brancaccio,

¹

Francesca Maria Plutino,

³

and Leonzio Fortunato

^1,2

1School of Dentistry, Magna Graecia University of Catanzaro, Italy

2Department of Health Sciences, Magna Graecia University of Catanzaro, Italy

3Istituto Clinico De Blasi, Reggio Calabria, Italy

Correspondence should be addressed to Amerigo Giudice; a.giudice@unicz.it Received 2 November 2018; Accepted 13 February 2019; Published 27 February 2019 Academic Editor: Pablo I. Varela-Centelles

Copyright © 2019 Amerigo Giudice 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 (AL) is an uncommon benign soft tissue neoplasia arising from the tunica media of the smooth muscle cells. AL appears as a solitary and slow-growing mass and seldom is observed in oral tissues. We reported a rare case of AL involving the cheek of a 17-year-old young woman. A review of the English-language literature was performed entering the keywords

“angioleiomyoma” and “oral” in the search fields of PubMed. 70 results were identified. Excluded were cases that were not in the oral cavity or not compatible with the AL diagnosis or report lacking immunohistochemical analysis. According to the exclusion criteria, we selected 30 studies that included 63 cases of AL. The results of the review showed an average age of 42.97 years with a prevalence between the fourth and fifth decade of life with a male-to-female ratio of 1.95 : 1. The most affected sites were palate, buccal mucosa of the cheek, lip, tongue, and gingiva. Surgical excision was the treatment of choice, and diagnosis was possible through histopathological and immunohistochemical analysis. SMA, vimentin, CD34, desmin, and S-100 were the most common markers to guide the histopathological diagnosis of oral AL. In conclusion, oral AL is a rare entity, especially in adolescence as in the reported case of AL of the cheek in a 17-year-old woman. The clinical aspects of AL did not allow clinicians to make a correct presumptive diagnosis. A scrupulous histopathological analysis and immunohistochemical examinations are fundamental to differentiate AL from other lesions.

1. Introduction

Leiomyoma is a circumscribed benign smooth muscle neo- plasia that frequently occurs on the skin, especially on the lower extremities, in the gastrointestinal tract and female genital tract. Given the low presence of smooth muscle cells in the oral cavity, leiomyoma rarely localizes in the mouth:

it develops from smooth muscle cells of tunica media or excretory ducts of salivary glands [1, 2].

In relation to the prevailing histological pattern, it can be classi ﬁed into three types: solid leiomyoma, vascular leio- myoma (angioleiomyoma), and sporadic form of epithelioid leiomyoma (leioblastoma) [3].

Angioleiomyoma (AL) is the most common microscopic pattern a ﬀecting the oral cavity. Nevertheless, oral AL is a rare benign tumor. Typically, it occurs in middle-aged man between the fourth and sixth decades of life [4].

AL was found in the cheek, lips, tongue, mandible, palate, and gingiva. AL is commonly present as well circumscribed and slow-growing asymptomatic lesion [5–7].

The clinical aspects of AL are similar to many other lesions of the oral cavity. Therefore, the diﬀerential diagnosis of AL in the oral cavity includes other benign conditions of the salivary glands as mucocele, pleomorphic adenoma, mes- enchymal tumors, lymphangioma, pyogenic granuloma, and schwannoma [8, 9].

Volume 2019, Article ID 2498353, 9 pages https://doi.org/10.1155/2019/2498353

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The diagnosis is possible after histopathological and immunohistochemical analysis due to its nonspeciﬁc clinical features. Surgical excision is the treatment of choice of AL, and recurrences are very rare [9].

We report a rare case of AL in a 17-year-old woman and an extensive review of the literature on oral AL.

2. Case Report

A 17-year-old woman was referred to our Oral Pathology Unit by her family dentist to evaluate a solitary asymptom- atic, mobile, and well-circumscribed mass in her left cheek.

The patient reported a swelling in the left cheek in the last two months. Her family and medical histories were irrele- vant; she was not following any drug therapy; she did not smoke or usually consume alcohol.

Clinically, we observed a palpable hard-elastic mass, measuring 1 5 × 1 0 cm approximately, in the submucosal layer of the left cheek. The patient did not report pain; the skin and the covering mucosa were normal. An ultrasound scan (US) was prescribed and showed a hypoechoic homog- enous mass with well-deﬁned margins.

The patient underwent an excisional biopsy under local anesthesia (Figure 1) after received an antibiotic prophylaxis therapy with 2 grams of amoxicillin 60 minutes before the surgery. She continued antibiotic therapy with 1 gram of amoxicillin every 12 hours until the 4

^th

postoperative day.

As analgesic therapy, the patient received paracetamol 500 mg immediately after the surgery.

The specimen was stored in a tube containing formalin 10% and sent to a laboratory for histopathological analysis.

The tumor was well circumscribed with a thin ﬁbrous capsule, and a sample obtained was ﬁrm, sharply circum- scribed, yellow-white round to ovoidal nodule (1 × 0 6 cm in dimension). It was composed of uniform spindle smooth muscle cells with pale eosinophilic cytoplasm and blunt-ended or cigar-shaped nuclei, with slightly wavy con- tour, vesicular chromatin, and occasionally small nucleolus;

in cross-sections, nuclei appeared surrounded by a clear halo as contained in boxes. Mitotic figures were very rarely seen (1/20 hpf). The cells were arranged in uniform interlacing bundles, with interposition of the low amount of fibrous con- nective tissue, and distributed around numerous small tortu- ous “slit-like” vessels, with virtual lumen and lined by normal-appearing endothelium but with no elastic lamina present, resembling a solid or capillary subtype appearance (closely compacted smooth muscle bundles), in contrast to venous (vessels have thick muscular walls that merge with smooth muscle bundles) and cavernous (dilated vascular channels with minimal smooth muscle that merges with smooth muscle bundles) subtypes; they have no clinical sig- nificance [1].

Necrosis, atypical mitoses, and pleomorphism were not observed in the histological examinations (Figure 2).

In addition to the histopathological analysis, immunohis- tochemical staining of the sample with

α-smooth muscle

actin (SMA), CD34, desmin, and vimentin was performed:

the proliferating spindle cells were di ﬀusely and strongly immunoreactive for SMA, desmin, and vimentin; the

vascular spaces were consistently CD34-positive staining (Figure 3).

The histopathological and immunohistochemical analy- sis suggested the diagnosis of AL.

Follow-up examinations at 1 week, 4 weeks, and 6 months showed mucosal integrity and no sign of recurrence.

3. Review of the Literature

A review of the English-language literature was performed.

The keywords “angioleiomyoma” and “oral” were entered in the search ﬁelds of PubMed. The research was conducted by considering the articles published until August 2018. 70 results were identi ﬁed. Excluded were cases that were not in the oral cavity or not compatible with the AL diagnosis or report lacking immunohistochemical analysis.

According to the exclusion criteria, we selected 30 studies [3, 4, 8–34] that included 63 cases of AL. We analyzed patient’s age, gender, tumor location, size, and immunohisto- chemical markers. The principal features and data pertaining the selected cases and those of the reported case are compiled in Table 1.

Age data were available in all selected studies, except for 14 cases described by Aitken-Saavedra et al. that reported only the mean age of 45.2 [34].

In our study, the average age was 42.97 years (range 2 months-79 years old) with a prevalence between the fourth and ﬁfth decade of life.

All studies included in our review, except one, reported the gender of the subjects. The analysis of the data collected suggested a male predilection with a male-to-female ratio of 1.95 : 1 (63 cases: 41 M, 21 F, and1 not reported). A graph of the age and gender distribution is reported in Figure 4.

In 63 cases reviewed, the analysis of localization in the oral cavity showed 19 in the lip (30%; 11 in the upper lip, 17.4%; 8 in the lower lip, 12.6%), 16 cases in the buccal mucosa of the cheek (or buccal space; 25.3%), 12 in the palate (19%), 5 in/on the tongue (7.9%), 5 in the gingiva (7.9%), and 3 in the mandible (4.7%). One lesion was observed in the ret- romolar area (1.58%). Other localizations were in the lower left back tooth region (1.58%) and in the lingual mucosa of the mandible (1.58%).

Size data were available in 46 of 63 cases. As reported in the literature, the size of the tumor can be very variable with a range from 0 5 × 0 5 to 3 5 × 3 3 × 2 0 cm.

Figure 1: Surgical excision of the lesion.

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The immunohistochemical analysis was performed for the diﬀerential diagnosis in all studies considered. This inves- tigation revealed that the specimens were reactive to SMA (95.2%), desmin (73%), CD34 (44.4%), vimentin (42.8%), S-100 (7.9%), HHF-35 (7.9%), factor VIII (4.7%), h-caldesmon (1.58%), CD31 (1.58%), and NSE (1.58%).

Radiological investigations were not been prescribed in most cases considered; however, the investigations com- monly described by many authors in the literature were MRI, CT, and US.

4. Discussion

Leiomyoma is described by the World Health Organization as a tumor of the soft tissue that arises from smooth muscle.

It can be found in sites rich in smooth muscles such as the gastrointestinal tract, the myometrium, and the skin. Leio- myoma originating from smooth muscle cells of vessels lying on deep soft tissue is rare. Due to the lack of smooth muscle in the oral cavity, leiomyoma is relatively rare or uncommon.

The possible sources of smooth muscle in the oral cavity include blood vessels, circumvallate papillae, and heterotopic smooth muscle [1, 2, 35].

In oral cavity, its origin is not well elucidated, but minor trauma, venous stasis, hormonal changes, and genetic trans- location have been postulated as possible causes [36].

Leiomyoma is classi fied in three different types/major groups according to the prevailing histological pattern: the most common is the vascular form de fined as AL (75%), followed by the solid form defined as leiomyoma (24%),

and some cases of an epithelioid form deﬁned as leiomyo- blastoma are reported in the literature (<1%) [5, 37].

AL is a benign tumor resulting from the tunica media of smooth muscle cells of arterial and venous walls [37].

In the literature, it has been reported that AL accounts for 5% of all benign soft tumors and represents 3-3.9% of neo- plasms that occur in the oral cavity [29, 36]. The incidence of AL in the oral cavity is rare and has been estimated to be roughly 0.065% [3].

Morimoto proposed a subclassi ﬁcation of AL describing solid, cavernous, and venous types [38]. On the authors’

knowledge, only few authors have followed this subclassiﬁca- tion reporting cases of intraoral AL: Liu et al. in a series of 21 tumors reported 5 solid, 6 venous, 9 cavernous, and 1 venous-cavernous AL [28]; Aitken-Saavedra et al. described 8 solid type, 4 cavernous type, and 2 venous type of AL [34].

Most of the AL are diagnosed between the fourth and sixth decade of life [38, 39], even if several studies have doc- umented the tumor occurrence in subjects from 1 month to 84 years old [9]. There is only one study that reported a con- genital tumor [21]. When AL occurs in this population, it is called leiomyomatous epulis, which clinically mimics a con- genital granular cell tumor [28]. AL was rarely found during infancy and adolescence: only 6 reported cases of AL (5 males, 1 female) were diagnosed in patients younger than 20 years of age (Figure 4).

Conversely to extraoral localization, intraoral AL has a male predilection as reported in the literature by several authors [6, 24, 28, 34]. The results of our review of 63 cases con ﬁrm this with a 1.95 : 1 male-to-female ratio.

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(c) (d)

Figure 2: Histological ﬁndings of the case of angioleiomyoma reported. (a, b, c) The tumor is well circumscribed and shows an admixture of bundles of smooth muscle cells surrounding the blood vessels (4x); (d) AL: high-power view (10x).

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Many reports describe the lips as the most aﬀected site with a frequency of 49%, followed by the palate, the buccal space, the mandible, the tongue, and the gingiva. Rare cases of intraosseous tumors are reported in the literature [25, 29]. The cheek was not the most frequent site of AL in the oral cavity, and the 16 cases reported in our review were observed in adult patients (age > 21). Therefore, we can con- sider our case rare of AL of the cheek reported in a young 17-year-old female teenager.

At clinical examination, AL appears in the oral cavity as a small, solitary, slowly developing mass [37], most commonly painless, and well localized. It presents as a palpable soft mass or elastic ﬁrm mass beneath the mucosa. The color of the mucous surface can be very variable, from normal to rosy or red [6].

Radiological investigations potentially useful for the diag- nosis of AL are MRI, CT, and US. Yanagi et al., analyzing the usefulness of dynamic contrast-enhanced MRI in the differ- ential diagnosis of AL in the buccal space, observed a very high signal intensity on T2WI and extremely high enhance- ment on Gd-T1WI; the inner aspect was homogeneous on T1WI and T2WI [40]. On CT images, AL appeared as a well-de fined mass, heterogeneously well enhanced after the dye injection [22]. US revealed general hypoechogenicity with well-de fined margins; in power Doppler mode, vascu- larity varies in density from low to high [41].

It is diﬃcult to distinguish AL from the other solid lesions of the oral cavity, such as lymphangioma, hemangioma, ﬁbroma, lipoma, pyogenic granuloma, and some other malignant lesions like angioleiomyosarcoma. AL is a benign

(a) (b)

(c) (d)

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Figure 3: Immunohistochemical findings of the case of angioleiomyoma reported. (a) The tumor cells show strong and diffuse SMA expression (4x); (b) blood vessels endothelium marked with CD34 immunostaining (4x); (c) blood vessels endothelium marked with CD34 immunostaining at high magnification (10x); (d) blood vessels endothelium marked with CD34 immunostaining at high magnification (20x); (e) the tumor cells show strong and diffuse desmin expression (4x); (f) uniformly staining for vimentin (4x).

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Table 1

Study (year of publication) N of cases

Age

(years) Gender Tumor location Size (cm) Immunohistochemical markers

(1) Maeda et al. (1989) [10] 1 37 M Upper lip 0.7 × 0.8 Vimentin+, desmin+,

factor VIII+ S-100^-, NSE+

(2) Maeda and Osaki (1989) [11] 1 49 F Cheek 2.0 × 2.0 Vimentin+, desmin+,

factor VIII+ S-100^- (3) Anastassov and

Damme (1995) [12] 1 51 M Upper lip 1.5 × 1.0 SMA+, factor VIII-

(4) Toida et al. (2000) [13] 1 10 M Lower lip 2.0 × 1.5 SMA+, S-100^-

(5) Marden et al. (2004) [14] 1 25 M Tongue 3.2 × 3.0 × 1.2 SMA+, CD34⁺, CD31⁺

(6) Manor et al. (2007) [15] 1 39 M Buccal mucosa 3.5 × 3.3 × 2.0 SMA+

(7) Scheper et al. (2007) [16] 1 67 M Palate 5.0 × 5.0 SMA+

(8) Suresh et al. (2007) [17] 1 51 F Mandible 1.7 × 1.5 SMA+, vimentin+, CD34^-, S-100^-

(9) Cepeda et al. (2008) [18] 5

39 27 43 36 48

F F F M M

Retromolar area Mandible Lower lip Upper lip Upper lip

0.9 × 0.6 × 1.0 1.4 × 1.3 × 1.0 0.7 × 0.5 × 0.4 1.5 × 1.0 × 1.0 1.7 × 1.0 × 1.0

SMA+, vimentin+, desmin+, CD34^-, S-100^- SMA+, vimentin+, desmin+,

CD34^-, S-100^- SMA+, vimentin+, desmin+,

CD34^-, S-100^-

(10) Keerthi et al. (2009) [19] 1 32 M Cheek 4.5 × 4.0 SMA+

(11) Grossman et al. (2009) [20] 1 35 F Palate 1.0 × 1.0 × 0.5 SMA+, vimentin+, desmin+, S-100^-, HHF-35+, AE1/AE3- (12) Kim et al. (2010) [21] 1 2 months Not

reported Tongue 2.5 × 2.0 SMA+, vimentin-, desmin+, S-100^-

(13) Kim et al. (2010) [22] 1 51 M Buccal space 3.0 × 3.0 SMA+, CD34^-, S-100^-

(14) Nonaka et al. (2011) [23] 1 39 M Tongue 2.0∅ SMA+

(15) Gueiros et al. (2011) [24] 3

54 66 54

M M M

Lower lip Upper lip Upper lip

1.0 × 1.0 Not reported 0.8 × 0.5 × 0.5

SMA+, desmin+, CD34⁺, S-100⁺, HHF-35+, D2-40- SMA+, desmin+, CD34⁺, S-100^-,

HHF-35+, D2-40- SMA+, desmin+, CD34⁺, S-100⁺,

HHF-35+, D2-40- (16) Patil et al. (2011) [25] 1 57 M Lower left back

tooth region 3.0 × 1.5 × 1.0 SMA+, vimentin+, desmin+, S-100^- (17) Menditti et al. (2012) [26] 1 14 M Lingual mucosa of

mandible 1.0/2.0∅ SMA+

(18) Eley et al. (2012) [27] 1 39 M Palate 2.0∅ Desmin+, actin+

(19) Liu et al. (2014) [28] 14

62 49 36 51 49 10 30 20

F M F F F F F M

Buccal mucosa Buccal mucosa Buccal mucosa Buccal mucosa

Palate Mandible Buccal mucosa

Gingiva

1.5∅ 2.0∅ 2.0∅ 1.0∅ 1.0∅ 3.5∅ 3.0∅ 2.5∅

SMA+, vimentin+, desmin+, CD34⁺in all cases S-100⁺only in 3 cases

(not speciﬁed)

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tumor with a low rate of malignant transformation, and deﬁnitive diagnosis needs histopathological analysis. The presence of cellular atypia, pleomorphism, and necrosis at histological analysis is common in both AL and angioleio- myosarcoma, while the number of mitosis is the main crite- rion to establish the malignancy. Tumors that have 4-10 mitosis for 10 high-power ﬁelds (HPF) should be considered as potentially malignant, while those with at least 5 mitosis for 10 HPF as malignant [6, 19, 42].

The prognosis of AL also depends on surgical treatment, in fact the complete surgical excision represents the best strategy of treatment of AL [25].

In such cases, recurrence of AL occurs mainly due to incomplete excision of the lesion: in literature, recurrence have been reported in a few cases [19]. It is important to per- form a complete tumor resection and a long-term follow-up observation [3].

Immunohistochemical analysis represents an essential tool in the diagnosis of AL. The most common markers assessed to conﬁrm the diagnosis of AL are SMA, vimentin, CD34, desmin, and S-100 [28].

The results of this review reported a di ﬀuse positivity to SMA in almost all cases. The markers vimentin, CD34, and desmin were reactive in about half of the cases. Only two

Table 1: Continued.

Study (year of publication) N of cases

Age

(years) Gender Tumor location Size (cm) Immunohistochemical markers 60

34 58 18 19 47

M M M M M M

Palate Palate Lip Palate Tongue Buccal mucosa

1.5∅ 1.0∅ 6.0∅ 3.5∅ 3.5∅ 2.0∅

(20) Tsuji et al. (2014) [3] 1 79 M Palate 1.5 × 1.5 SMA+, desmin+

(21) Ishikawa et al. (2014) [29] 1 51 M Tongue 1.1∅ SMA+, vimentin+, desmin+,

CD34^-, S-100^-, HHF-35+

(22) Ranjan and Singh (2014)

[30] 1 45 F Gingiva 3.0 × 3.0 SMA+

(23) Inaba et al. (2015) [31] 1 45 F Cheek Not reported SMA+, factor VIII+

(24) Osano et al. (2015) [9] 1 45 M Cheek 2.0∅ SMA+, vimentin+, desmin+,

CD34^-, S-100^- (25) Arpağ et al. (2016) [32] 2 25

55

M F

Gingiva Gingiva

0.5 × 0.5 1.5 × 2.0

SMA+

(26) Bajpai et al. (2016) [33] 1 39 M Gingiva 3.0 × 3.0 SMA+, vimentin+, desmin+

(27) Hassona et al. (2017) [4] 1 52 F Upper lip Not reported SMA+

(28) Rawal and Rawal (2017) [8] 1 70 M Palate 2.0 × 1.5 SMA+

(29) Matiakis et al. (2018) [7] 1 51 M Labial mucosa of

the upper lip 0.8∅ SMA+, h-caldesmon+

(30) Aitken-Saavedra et al.

(2018) [34] 14

Total mean 45.2

Male = 8 Female = 6

Lower lip Lower lip Lower lip Lower lip Upper lip Upper lip Upper lip Buccal mucosa Buccal mucosa Buccal mucosa Buccal mucosa Soft palate Soft palate Hard palate

Not reported Not reported Not reported Not reported Not reported Not reported Not reported Not reported Not reported Not reported Not reported Not reported Not reported Not reported

SMA+, AE1/AE3-, CD68^-, desmin+, S-100^-in all cases

CD34⁺only in 10 cases (not speciﬁed)

Case report 1 17 F Cheek 1.5 × 1.0 SMA+, vimentin+, desmin+,

CD34⁺

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study showed S-100 positivity in small nerve fibers in five cases of AL [24, 28]. Matiakis et al. found the positivity of h-caldesmon in one case: they identi fied this marker as more speci fic for smooth muscle fibers than SMA and desmin, also to differentiate AL from myopericytoma [7].

Maeda et al. showed that vascular walls, hardly identiﬁed by hematoxylin eosin stain, became visible through factor VIII immunohistochemical staining [10, 11]. The factor VIII is synthesized by endothelial cells, and its expression has been reported in numerous vascular neoplasms [43].

Endothelial cells also express CD31; therefore, antibodies to CD31 have been used as a tool to identify the vascular ori- gin of neoplasms [44].

Kim et al., Gueiros et al., and Ishikawa et al. described a positive staining for HHF-35 that, in addition to reactivity for SMA, vimentin, desmin, and S-100, can assist in the diag- nosis as an adjunct to H&E staining [22, 24, 29].

Cepeda et al. emphasized the importance of immunohis- tochemical analysis in order to diﬀerentiate AL from other types of spindle cell tumor, including leiomyoma (CD34

^-

and S-100

^-

), myopericytoma (desmin

^-

, CD34

^-

, and S-100

^-

), and myo ﬁbroma (desmin

^-

, CD34

^-

, and S-100

^-/+

) [18].

Kim et al. and Aitken-Saavedra et al. reported a negativity of the sample for AE1/AE3 and CD68 antibodies and showed that only SMA can be elected as a good marker for AL and be of help in the diagnosis of this lesion [22, 34].

Gueiros et al. immunohistochemical analysis showed a negativity of the sample for D2-40 and a positive staining for SMA, desmin, CD34, HHF-35, and S-100 in 2 cases [24].

Each marker is a characteristic of a speciﬁc tissue, but its detection is useful only in combination with clinical judge- ment and the measurement of other markers.

In conclusion, oral AL is a rare entity, especially in ado- lescence. We have reported a new rare case of AL of the cheek in a young woman. The clinical aspects of AL did not allow us

to make a correct presumptive diagnosis. A scrupulous histopathological analysis made it possible to identify the pathological entity of the lesion. Immunohistochemical examinations are fundamental to di ﬀerentiate AL from other lesions. SMA, vimentin, CD34, desmin, and S-100 are the most commonly investigated markers to guide histopatho- logical diagnosis of oral AL.

Conflicts of Interest

The authors declare that they have no con ﬂicts of interest.

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