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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
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CASE REPORT  
Year : 2023  |  Volume : 27  |  Issue : 5  |  Page : 15-19
 

Glomus tumor in the buccal mucosa: A case report and review of the literature


1 Department of Oral and Maxillofacial Medicine, Shiraz Branch, Islamic Azad University, Shiraz, Iran
2 Department of Oral and Maxillofacial Medicine, School of Dentistry, Shiraz University of Medical Sciences, Shiraz, Iran
3 Department of Oral and Maxillofacial Pathology, School of Dentistry, Shiraz University of Medical Sciences, Shiraz, Iran
4 Department of Oral and Maxillofacial Surgery, School of Dentistry, Birjand University of Medical Sciences, Birjand, Iran

Date of Submission27-May-2022
Date of Decision19-Jun-2022
Date of Acceptance19-Jun-2022
Date of Web Publication04-Feb-2023

Correspondence Address:
Saeid Tavanafar
Department of Oral and Maxillofacial Surgery, School of Dentistry, Birjand University of Medical Sciences, Birjand
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jomfp.jomfp_232_22

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   Abstract 


Glomus tumors are rare benign neoplasms usually occurring in the upper and lower extremities. However, oral cavity involvement is rare, with only a few case reports. We present a 37-year-old male patient with a chief complaint of an exophytic lesion in the left buccal mucosa for one year referred to our center. At the time, he did not have any pain or lymphadenopathy. The patient underwent surgery using a high-power CO2 laser. His histopathological examination revealed a glomus tumor. After 2 years of follow-up, no evidence of recurrence was detected. Glomus tumors should be taken seriously when patients have a painless exophytic bluish lesion in the buccal mucosa.


Keywords: Floor of mouth, glomus tumor, oral surgery


How to cite this article:
Afroozi B, Rezazadeh F, Jaafari-Ashkavandi Z, Tavanafar S. Glomus tumor in the buccal mucosa: A case report and review of the literature. J Oral Maxillofac Pathol 2023;27, Suppl S1:15-9

How to cite this URL:
Afroozi B, Rezazadeh F, Jaafari-Ashkavandi Z, Tavanafar S. Glomus tumor in the buccal mucosa: A case report and review of the literature. J Oral Maxillofac Pathol [serial online] 2023 [cited 2023 Mar 27];27, Suppl S1:15-9. Available from: https://www.jomfp.in/text.asp?2023/27/5/15/369169





   Introduction Top


The glomus apparatus is an arteriovenous anastomosis that acts in thermal regulation, located at the dermis's stratum reticularis, mainly in the subungual region, lateral areas of the digit, and palm of the hands, and on the feet ventral surface.[1],[2] Glomus cells are small, uniform, rounded cells with a central nucleus and pale eosinophilic cytoplasm.[3] The most common areas for this tumor are zones rich in glomus bodies. Generally, it appears as a small blue-red nodule associated with tenderness and pain and sensitivity to cold or tactile provocation. It occurs more commonly amongst young or middle-aged adults,[4],[5] but oral cavity involvement is infrequent. To the best of our knowledge, very few cases have been reported to date. There are only 23 well-documented cases of intraoral glomus tumors reported in the English language literature.[1],[6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24],[25],[26],[27] Here, we present a case of an unusual glomus tumor that originated in the left buccal mucosa.


   Case Report Top


A 37-year-old male patient with a one-year history of an exophytic lesion in the left buccal mucosa of his mouth was referred to the Oral Medicine Department, Shiraz University of Medical Sciences, Iran. The patient has signed a written informed consent. Although he experienced slight bleeding, no history of pain or paralysis was reported. His medical and family history was unremarkable. Intraoral examination showed a well-defined 2 × 2 cm-sized exophytic lesion in the left buccal mucosa. The lesion was covered with light bluish smooth mucus, soft to rubbery in consistency, pedunculated [Figure 1]a. The patient experienced bleeding when pressure was applied to the tumor. No lymphadenopathy in the fascial, submandibular, and submental regions was detected upon palpation. All relevant laboratory test results were typical.
Figure 1: Exophytic lesion in the left buccal mucosa before (a) and after (b) surgery, using a high-power CO2 laser

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The initial clinical diagnosis was a benign vascular neoplasm, benign neural tumor, or inflammatory hyperplasia. The patient was scheduled for surgery using a high-power CO2 laser. The lesion was removed [Figure 1]b under infiltrating perilesional local anesthesia (2% lidocaine with 1:100000 epinephrine). The power was set to 4W. The teeth and adjacent tissues were protected using a wooden spatula. All safety measures were observed during the surgical procedure. The beam was focused on removing the lesion. At the end of the surgery, the beam was defocused to promote better hemostasis. Neither sutures nor dressings were used following the surgery. The patient received standard orientation for the postoperative period, and Gelofen (400 mg, twice a day) and mouthwashes (0.12% chlorhexidine) were prescribed. The postoperation was uneventful, except for mild discomfort caused by removing the lesion.

The tissue sample was fixed in a 10% formalin and sent for histopathological diagnosis. Microscopically, the mucosal mass was covered by keratinized stratified squamous epithelium. The underlying connective tissue showed lobules of endothelial cell proliferation with capillary formation. Some mitosis, areas of hemosiderin pigmentation, and dense chronic inflammatory cell infiltration were also noticed. A benign mesenchymal tumor consisting of sheets of uniform cells surrounding the branching capillary-sized vessels was diagnosed. The tumor cells were round with an indistinct border and eosinophilic cytoplasm [Figure 2].
Figure 2: Histopathological features of glomus tumor. (a) Homogeneous glomus cells with punch-out nuclei in the perivascular area (H&E staining, 400×). (b) A well-circumscribed mesenchymal tumor with the perivascular distribution of round cells (H&E staining, 100×)

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Immunohistochemistry revealed that the tumor cells yielded positive results for vimentin, alpha-smooth muscle actin, and focally positive for CD34. However, negative results for factor 8, anti-cytokeratin (AE) 1 or AE3, cluster of differentiation (CD) 31 and CD34, and S-100 exhibited a Ki-67 index of less than 5%. These findings were consistent with those of a glomus tumor. The tumor cells were positive for vimentin and SMA, focally positive for CD34, and negative for CD31, factor VIII, S-100, and p63. After surgery, the patient had an uneventful recovery with primary healing and no recurrence evidence after two years of follow-up.


   Discussion Top


Glomus tumors are rare mesenchymal tumors, representing less than 2% of all benign soft tissue tumors. Glomus body hyperplasia or hamartomatous development is the most common cause of this tumor. They originate from modified smooth muscle cells.[28] Most glomus tumors are characterized as solitary, small, and nodular painful lesions, commonly seen in superficial soft tissues of adults' distal extremities during the third to fifth decades of life. However, it may occur at any age and location. A predilection for women can be seen in subungual lesions.[23]

The present case showed similar features, with only the location being different, which was in the buccal mucosa, where it is infrequent. Recently published English language literature regarding glomus tumors of the oral cavity was reviewed. We could identify only 25 cases involving the oral cavity (including our case); most of them were on lips [Table 1]. A long history of pain triggered by temperature or tactile stimulation is the most common feature of glomus tumors.[29] Identification of substance P in nerve fibers of GT and the presence of numerous nerve fibers in the capsule of the lesion and contraction of myofilaments in the glomus cells in response to temperature changes increases the intracapsular pressure. This stimulated unmyelinated nerve fibers can explain pain mechanisms in glomus tumors.[30],[31]
Table 1: Cases of oral glomus tumor reported in the literature

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The neoplasms developed in 13 women and 12 men in the age range of 10–85 years (with a mean age of 48.5 years). There was no gender predilection similar to extraoral glomus tumors, except for subungual lesions commonly seen in females. Some lesions present pain or tenderness, and most involve the lips (54.2%), followed by the hard palate. Only a few cases were described in the gingiva, tongue, and buccal mucosa. In most cases, the tumor was situated on the lip (n = 12, lower 3, upper 9), followed by the hard palate (n = 4), buccal mucosa (n = 3), tongue (n = 3), gingiva (n = 1) and multiple locations (n = 1), with an upper to lower lip ratio of 2.3:1 (37.5% and 16.6%, respectively). Depending on the specific localization (skin or mucosa), the tumor cells can be either dermal or submucosal origin. The possibility of glomus tumors' presence in the head and neck is proportional to the distribution of glomus bodies in the face.[14] Hence, the distribution of the glomus body in the oral mucosa must be determined.

Unfortunately, many cases had an unknown clinical presentation or medical history. Of the cases with available information, including the present case, a mean size of 12 mm was revealed. Intraoral glomus tumors are slightly larger than extraoral ones. Glomus tumors in the dermis or subcutaneous tissues of the hands and feet are usually 10 mm or smaller in size.[3] Due to the low occurrence rate of glomus tumors in the head and neck area, accurate information on the peak incidence and gender ratio is still unclear.

Cases of malignancy have been documented in extra-oral glomus tumors, but it is rare in the head and neck area.[32] The accurate diagnosis of malignant transformation of this tumor is via histopathologic examination. Tumors more than 2 cm in diameter with atypical mitotic figures, moderate-to-high nuclear grade, and more than five mitotic figures per 50 high-power fields are more susceptible to malignant transformation.[33] Due to low incidence and lack of specific clinical and radiographic features, accurate diagnosis of this tumor is very difficult. A salivary tumor, sebaceous cyst, neurofibromatosis, dermoid cyst, teratoid tumor, vascular malformation, or another type of mesenchymal neoplasm can be mistakenly diagnosed as a glomus tumor.[34] Color duplex ultrasonography can rule out vascular malformations and cystic soft tissue lesions. Since there is no specific diagnostic guideline, histological examination and immunohistochemical analysis are the gold standards.

Three histological patterns were described, depending on the proportion of glomus cells, blood vessels, and smooth muscle. These different patterns can occur in the same case: solid (75%), glomangioma with a vascular predominance (20%), and glomangiomyoma with smooth muscle cell predominance (5%).[2],[3] The solid pattern is the most common in oral glomus tumors, similar to skin tumors. Glomus tumor is a smooth muscle-like phenotype, with constant positivity for specific-muscle actin (HHF-35), α-SMA, and h-Caldesmon. Pericytic/smooth muscle differentiation was observed by transmission electron microscopy.[5] These features indicate that glomus tumors can arise from modified smooth muscle cells of the glomus body. Moreover, PAS highlighted strong pericellular positivity for type IV collagen in a “chicken-wire” pattern in glomus tumors, confirming a basement membrane's presence around the glomus cells. Other immunomarkers such as desmin, cytokeratin, and S-100 are usually negative.[2],[29]

Positivity for desmin, CD34, and BRAF mutations was identified in some cases.[35] First, glomus tumors should be distinguished from epithelial tumors. Epithelial markers were negative in this case; hence, the tumor was not a neoplasm of epithelial original.[36] Second, hemangioendothelioma, epithelioid hemangioma, Kaposi-form angiodermatitis, reactive angioendotheliomatosis, and angiosarcoma should be included in the differential diagnosis of glomus tumor.[22] Histomorphologic features and the expression of endothelial cell markers can exclude these tumors.[37] Most glomus tumors are benign; therefore, surgical excision can be an effective treatment. Recurrence transpires due to incomplete resection, and local recurrence is infrequent. Malignant glomus tumors are sporadic and require multimodal integrated treatment.[38]

We reported a rare glomus tumor in the left buccal mucosa with no specific symptoms, which complicated its early diagnosis. Glomus tumors should be included in the initial differential diagnosis in patients presenting painless exophytic bluish color lesions in the buccal mucosa. Surgical excision using a high-power CO2 laser is an effective modality, and patients should receive long-term follow-up due to the risk of recurrence.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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