Ⅰ. INTRODUCTION

Glomangiomas, or glomuvenous malformations, are rare cutaneous venous malformations that show glomus cells, along with the venous system in histopathological examination.¹⁾ Most commonly, they present as solitary lesions in the extremities, especially the subungual region, but they may present elsewhere. However, their occurrence in the oral and maxillofacial region has rarely been reported.²⁾ Glomangiomas are histologically characterized by malformed venous channels that are lined by several layers of glomus cells. These cells are defined as α-actin-positive cells arranged in solid strains or perivascular rows. Therefore, glomangiomas have features of both venous malformation and proliferation of glomus cells.³⁾ Glomangiomas arising in the oral and maxillofacial region have the possibility of being diagnosed late or misdiagnosed as a venous malformation or hemangioma owing to their rarity and propensity for specific anatomic sites. Herein, we report a rare case of solitary glomangioma on the chin that was clinically diagnosed as hemangioma but was ultimately diagnosed as a solitary glomangioma by histopathological examination.

Ⅱ. CASE REPORT

A 63-year-old man was referred to the Department of Oral and Maxillofacial Surgery, Dankook University Dental Hospital, in June 2020 for a mass in the chin area. The patient had no history of trauma or systemic diseases. No pain was present spontaneously and upon palpation, and the mass had grown gradually over the past 2 years. Clinical examination showed a 1.2 cm bluish, spherical nodule with slight mobility, and no pus discharge or paresthesia was observed (Figure. 1A, 1B). The lesion was clinically diagnosed as a hemangioma. Since the probability of bleeding was minimal considering the location and size of the lesion, magnetic resonance imaging, computed tomography, and angiography were not performed. Excisional biopsy was planned under local anesthesia. Aspiration performed after local anesthesia showed no specific bleeding, and the amount of aspirate was low. Complete excision was performed with secure surgical margins, and hemorrhagic findings were not observed.

The specimen was a well-encapsulated nodule 1.2 × 1.0 × 0.4 cm in size. Histopathological examination showed dilated blood vessels surrounded by glomus cells (Fig. 2) that had well-defined cell margins and consisted of round to ovoid cells (Fig. 3).

Ultimately, the diagnosis of glomangioma was established. The patient’s recovery was uneventful postoperatively, and no sign of recurrence was observed at the 1-year follow-up.

Ⅲ. DISCUSSION

Glomus tumors are rare benign tumors that consist of 1– 2% of all soft tissue tumors.⁴⁾ Glomus tumors often clinically manifest as a subcutaneous nodule that is often purple, red, or blue in color during the third to fifth decades of life.⁵⁾ The characteristic symptoms of glomus tumors include the classic triad of severe pain, localized tenderness, and cold hypersensitivity, although these symptoms cannot be shown.⁶⁾

Various proportions of glomus cells, smooth muscle tissue, and vascular structures constitute typical glomus tumors. Based on their predominant components, glomus tumors can be classified into the following three types: solid glomus tumors, glomangiomas, and glomangiomyomas. Among these, glomangiomas account for 20% of all glomus tumors and have a high proportion of blood vessels.⁷⁾ While most glomus tumors clinically present as a solitary form, glomangiomas usually occur as multiple form at birth or during childhood and do not involve the subungual region. ^8,9) However, in contrast to their multiple form, solitary glomangiomas most frequently arise from the subungual regions of digits. The clinical manifestation of solitary glomangiomas in regions outside this area is rare.⁷⁾ Compared with solitary tumors, glomangiomas are large and less well circumscribed and have a male predilection. Clinically, they present less often with the classic triad signs, making their differentiation from other cutaneous vascular malformations difficult.¹⁰⁾ Similar to other solitary tumors, glomangiomas are treated by complete excision, including a surgical margin of sound tissue, as incomplete excision can lead to recurrence. In particular, symptoms can recur within 2–3 years after surgery in multiple forms, which warrants proper follow-up.¹¹⁾ This case showed no evidence of recurrence at the 1-year follow-up. This case was misdiagnosed as hemangioma because of the absence of the classic triad signs and a solitary and well-circumscribed manifestation of the lesion.

In addition, glomangioma has not been reported in the chin region as a solitary lesion in the oral and maxillofacial surgery literature, which justifies the report of this case.