Angiolipoma presenting as a nasal mass: how rare a presentation?

Abstract

Angiolipomas are benign adipose tumors. There are two types: infiltrating and noninfiltrating. An angiolipoma presenting as a nasal mass is rare. To our knowledge, this is only the second documented case of angiolipoma presenting in the nasal dorsum. Total surgical excision by an external rhinoplasty technique with close follow-up is advised. Recurrence is not common.

Introduction

Angiolipomas are tumors consisting of adipose tissue marked by vascular proliferation. They present most commonly in young adults as subcutaneous tender or painful nodules, and they represent 6 to 17% of all lipomas. (1,2) The forearm is the most commonlyreported site, representing two-thirds of cases, with the trunk and the upper arm representing most of the remaining one-third of cases. (3) Multiple angiolipomas are more common than solitary nodules, and 5% of these cases are familial. (1)

Recurrence after excision is exceedingly rare, and there has been no cytologic evidence of malignant transformation. An extensive review by Alvi et al revealed only 17 adequately documented cases of head and neck angiolipomas in the English literature. (4) The sites of these tumors included the cheek, palate, neck, jaw, nose, eyelid, parotid, tongue, and mandible.

In 1998, a group from Japan reported the first case of an angiolipoma in the internal auditory canal. (5) The first case of nasal angiolipoma was reported by Shohet et al, also in 1998. (6) The present case is the second report of a nasal angiolipoma, located on the nasal dorsum.

Case report

A 52-year-old man presented with a 2-year history of a gradually enlarging painless mass over his nasal dorsum that had recently increased in size. He had occasional symptoms of right-sided nasal obstruction and frontal facial pain. Physical examination revealed a 3 x 2-cm soft, mobile mass over the dorsum. The mass was located in the subcutaneous tissue, slightly off the midline toward the right side, and the overlying skin was slightly darker than the surrounding skin. There was no evidence of an intranasal mass.

Computed tomography (CT) was performed to evaluate the possibility of a central nervous system connection. It revealed a mass with low attenuation within the subcutaneous region, without intracranial extension. Fine-needle aspiration of the mass revealed blood and scanty stromal cells and hence was inconclusive for a diagnosis.

An excision biopsy was performed through an external rhinoplasty approach for exposure. The mass was easily separated from the overlying skin and from the cartilaginous and bony nasal structure. The lesion was vascular, and copious bleeding was encountered during its removal; complete removal was accomplished. A cartilage graft was harvested from the septum and was used to fill the defect after the excision of the lesion.

Histologic examination of the mass showed nodules of mature fat cells separated by a network of vessels (figure, A). A few small vessels showed fibrin thrombi (figure, B). These features are consistent with a diagnosis of noninfiltrating angiolipoma.

The patient was asymptomatic at a 1-year follow-up examination.

Discussion

Angiolipomas are benign adipose tumors that differ from typical lipomas in that they usually arise around the time of puberty. On physical examination, the typical angiolipoma is less than 4 cm in diameter and generally tents the skin. It is rarely associated with overlying skin discoloration. Most patients present with mild, constant, dull pain that may be exacerbated by pressure. The pain is thought to be related to intravascular thrombi or to engorgement with associated edema and compression of the adjacent tissue, including nerves.

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The diagnosis can be further supported by CT. (7) Lipomas are typically homogeneous masses with low attenuation. Although it does not appear to be enclosed in a capsule, the mass is usually easily discerned from surrounding tissue. On a contrast-enhanced study, an angiolipoma shows marked enhancement as a result of its intense vascularity. (8) Fine-needle aspiration is rarely helpful. The definitive diagnosis of angiolipoma can only be made after histologic examination of the resected specimen.

Angiolipomas can be divided into noninfiltrating and infiltrating types. Noninfiltrating lesions are much more frequent and are so named because of their fibrous tissue encapsulation. Infiltrating angiolipomas arise in the deep soft tissue, including muscle, and may infiltrate adjacent structures. They are characteristically unencapsulated and have a clinical behavior similar to that of hemangiomas. (9)

Treatment of angiolipomas consists of total surgical excision. Simple excision of noninfiltrating angiolipomas is curative, with no tendency for recurrence. Successful treatment of infiltrating angiolipomas depends on the extent of their infiltration into surrounding tissues and poor encapsulation. The infiltrating type can present difficulty in excision, and their recurrence rate is high (approximately 50%). (10) In cases of inadequate resection, radiation therapy may be necessary. (2) The natural history of an angiolipoma, unlike that of a hemangioma, appears to include a tendency for increased growth; no cases of spontaneous regression have been reported. Koopman has suggested that waiting for spontaneous regression in angiolipoma will allow the lesions to become more destructive and infiltrative, leading to further cosmetic problems. (10)

References

(1.) Howard WR, Helwig EB. Angiolipoma. Arch Dermatol 1960;82: 924-31.

(2.) Lin JJ, Lin F. Two entities in angiolipoma. A study of 459 cases of lipoma with review of literature on infiltrating angiolipoma. Cancer 1974;34(3):720-7.

(3.) Pfannenstiel TJ, Boseley M, Roach L.A case of paranasal angiolipoma. Laryngoscope 2003;113(6):1080-1.

(4.) Alvi A, Garner C, Thomas W. Angiolipoma of the head and neck. J Otolarngol 1998;27(2):100-3.

(5.) Murakami S, Yanagihara N, Takahasi H, et al. Angiolipoma of internal auditory canal presenting repeated sudden hearing loss. Otolaryngol Head Neck Surg 1997;117(6):S580-S584.

(6.) Shohet JA, Simpson B, Coleman JR, Geiger XJ. Angiolipoma presenting as a nasal mass. Otolaryngol Head Neck Surg 1998; 118(6):848-9.

(7.) Som PM, Scherl MP, Rao VM, Biller HF. Rare presentatations of ordinary lipomas of the head and neck: A review. AJNR Am J Neuroradiol 1986;7(4):657-64.

(8.) Reilly JS, Kelly DR, Royal SA. Angolipoma of the parotid: Case report and review. Laryngoscope 1988;98(8 Pt 1):818-21.

(9.) Pribyl C, Burke SW, Roberts JM, et al. Infiltrating angiolipoma or intramuscular hemangioma? A report of five cases. J Pediatr Orthop 1986;6(2):172-6.

(10.) Koopman CF Jr. The "Pinocchio" nasaldeformity--hemangioma vs. angiolipoma: Esthetic correction and etiology. J Otolaryngol 1988; 17(4):169-72.

Sridhara Suryanarayan Rao, MS; Naresh K. Panda, MS, DNB, FRCSEd; Uma Nahar Saikia, MD; Karuppiah Saravanan, MS, DNB

From the Department of Otolaryngology (Dr. Rao, Dr. Panda, and Dr. Saravanan) and the Department of Histopathology (Dr. Saikia), Post Graduate Institute of Medical Education and Research, Chandigarh, India.

Corresponding author: Dr. Naresh K. Panda, Department of Otolaryngology, Post Graduate Institute of Medical Education and Research, Chandigarh. India, Pin 160012. Phone: [phone]; e-mail: [e-mail]