Massive noninfiltrating angiolipoma of the buccal mucosa: Report of an extremely rare case

Abstract

We present an extremely rare case of noninfiltrating angiolipoma (AL) of the buccal mucosa in a 90-year-old patient reaching a size of 14 cm × 10 cm. AL is rare in the soft tissues of oral cavity, and till date, only 22 cases have been reported, including our case. If size be taken into consideration, our case can be considered to be the first in literature to reach this massive size. Surgical excision of the tumor mass was performed. The histopathological findings confirmed the diagnosis of noninfiltrating AL. Follow-up for 1 year revealed no signs of recurrence.

INTRODUCTION

Angiolipoma (AL) is a variant of lipoma with marked degree of vascularization. It was first reported by Bowen in 1912.[1] ALs are differentiated into noninfiltrating (encapsulated) and infiltrating (nonencapsulated) types. The common occurrence of these tumors is prevalent among males in their second or third decades of life with site predilection toward trunk and extremities, especially in the forearm.[2] However, the incidence of AL is extremely rare in the intraoral region, with only 21 reported cases in English literature.[3] This case report presents the 22^nd case of intraoral AL of massive size.

CASE REPORT

A 90-year-old woman with no significant medical history was referred to our department for evaluation of painless mass arising from oral cavity. The tumor was noticed by the patient 20 years ago as a small nodule on the right buccal mucosa and since then it gradually increased in its size. The growth reached a size, which could not accommodate in mouth, and due to constant irritation from the tumor, the patient coughed the growth for 5 years. Since then, there was a rapid growth of the tumor. On extraoral examination, a solitary, smooth, nontender and nonpulsatile pedunculated growth of size 14 cm × 10 cm with variable consistency was evident with intraoral origin [Figure 1].

Figure 1.

Clinical image shows preoperative tumor

On intraoral examination, single, smooth, firm and nontender stalk-like pedicle of diameter 2 cm was seen originating from the right buccal mucosa. Based on the history and clinical examination, we provisionally diagnosed it as a benign growth. Surgical excision of the tumor was done under general anesthesia [Figures 2 and 3].

Figure 2.

Clinical image shows excised tumor mass

Figure 3.

Clinical image shows postoperative frontal view

Histopathology confirmed it as noninfiltrating AL [Figures 4–6]. One-year follow-up of the patient revealed no evidence of recurrence. Ethical approval from the Institutional Review Board was obtained for the publication, and patient release form was signed by the patient.

Figure 4.

Clinical image shows histopathology section (×4 magnifications)

Figure 6.

Clinical image shows histopathology section (×40 magnifications)

Figure 5.

Clinical image shows histopathology section (×10 magnifications)

DISCUSSION

Lipomas are the most frequently found soft-tissue benign tumors, but their occurrence in head and neck region is rare.[4] AL, in the oral cavity, was first documented by Davis et al. as a growth on the hard palate.[5] History of trauma, lipomatous differentiation by hormones during puberty, vascular proliferation of a congenital lipoma and fatty degeneration of a central hemangioma have been implicated as possible etiological factors.[6] Based on the studies by Gonzalez-Crussi et al., these ALs have been classified into two histologic types: Infiltrating and noninfiltrating.[7] The noninfiltrating ALs are encapsulated and common in young patients.[8] The infiltrating types are poorly encapsulated and usually diagnosed in elder patients. The mean diameter at the largest portions of all the 21 documented ALs occurring in oral region was 3 cm, and the mean onset age of the patients was about 29 years old.[4]

Although diagnostic modalities such as magnetic resonance imaging, computed tomography, ultrasonography and aspiration biopsy have been used to differentiate between hemangioma, lipomas and AL, the diagnosis of these tumors is confirmed only by histopathology.[9] The standard treatment of choice is surgical excision for the noninfiltrating ALs and complete surgical excision with a clear surgical margin for poorly encapsulated infiltrating ALs to avoid recurrence. There is no report of malignant transformation or recurrence of noninfiltrating ALs in literature.[10]

The case we have presented showed the typical clinical and histological findings of a noninfiltrating AL. Its most striking and unique features were its occurrence in a 90-year-old female with a 20-year long history and pedunculated mass of 14 cm × 10 cm. With regard to the size and its typical pedunculated appearance, our present case can be considered to be the first of its kind in literature.

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Conflicts of interest

There are no conflicts of interest.