Angiolipoma presenting as a nasal mass☆☆☆★★★

Angiolipoma presenting as a nasal mass JACK A. SHOHET, MD, BLAKE SIMPSON, MD, JOHN R. COLEMAN, MD, and XOCHI J. GEIGER, MD, Nashville, Tennessee

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48-year-old woman presented with a 2-year history of a mass over her nasal dorsum that had recently begun to enlarge. She had symptoms of right-sided nasal obstruction, nasal congestion, and frontal facial pain. A physical examination revealed a 3 ´ 2 cm soft, mobile mass over the nasal dorsum (Fig. 1). The mass was located in the subcutaneous tissue slightly off the midline to the left. The overlying skin was slightly darker than the surrounding skin. An intranasal examination revealed a deviated septum and a hypertrophic left inferior turbinate. There was no evidence of the mass intranasally. A computed tomography (CT) scan was performed to evaluate for the possibility of a central nervous system connection. This study revealed a low attenuation mass within the subcutaneous region without intracranial extension. At this point, fine-needle aspiration of the mass was performed, which revealed blood and scant stromal cells. An excisional biopsy was undertaken through an external rhinoplasty approach for exposure. The mass was easily separated from the overlying skin and from the underlying cartilaginous and bony nasal structures. The lesion was vascular and copious bleeding was encountered during removal of the mass. The mass was fibrofatty in character and was able to be removed en bloc. A cartilage graft was harvested from the septum and was used to fill the defect left from the excision. The overlying skin was then redraped and a nasal splint placed over the nasal dorsum. A follow-up examination showed that the nasal obstructive symptoms of the patient had resolved. She maintains a good nasal airway with markedly improved cosmesis of her nose (Fig. 2). A final histopathologic analysis revealed a diagnosis of angiolipoma (Figs. 3 and 4). DISCUSSION Angiolipomas are benign adipose tumors that represent 5% to 17% of all lipomas. These tumors are very rare in the head and neck.1 Only nine head and neck cases have been reported in the literature, and the current case represents the first presentation on the nasal dorsum. Previous cases have been reported to involve the parotid gland,2 intraosseous mandible,3-5 cheek,6,7 palate,8 and cervical area.9,10 Angiolipomas differ from typical lipomas in that they usually arise around the time of puberty.

From the Departments of Otolaryngology (Drs. Shohet, Simpson, and Coleman), and Pathology (Dr. Geiger), Vanderbilt University. Reprint requests: Jack A. Shohet, MD, Department of OtolaryngologyÐHead and Neck Surgery, UCI, 101 City Dr. South, Bldg. 25, Orange, CA 92868. Otolaryngol Head Neck Surg 1998;118:848-9. Copyright © 1998 by the American Academy of OtolaryngologyÐ Head and Neck Surgery Foundation, Inc. 0194-5998/98/$5.00 + 0 23/4/78270 848

Eighty percent of patients will have multiple lesions and they may have a familial component.1 On physical examination, the typical angiolipoma is less than 4 cm in diameter1 and generally tents the skin but is rarely associated with overlying skin discoloration. The mass is smooth, with mobile overlying skin. Most patients present with mild, constant, dull pain, which may be exacerbated by pressure. The pain is thought to be related to intravascular thrombi or to vascular engorgement with associated edema and compression of the adjacent neural tissue. A diagnosis can be further aided by CT scan findings.11 Lipomas are typically homogeneous masses with low attenuation values between Ð65 and Ð125 Hounsfield units. Although a capsule does not appear to surround the mass, it usually is easily discerned from the surrounding tissue. On a contrastenhanced study, an angiolipoma shows marked enhancement as a result of its intense vascularity.2 The noncontrasted study of an angiolipoma shows the homogenous low attenuation of a typical lipoma. Liposarcoma must be excluded when evidence of soft tissue or bony invasion on CT is found. Two different histopathologic forms of angiolipomas exist: circumscribed and infiltrating. Circumscribed lesions are much more frequent and are named as such because of their fibrous tissue encapsulation. They are, with few exceptions, limited to the subcutaneous tissue and do not exceed 4 cm in diameter. Microscopically, angiolipomas usually are sharply circumscribed and consist of mature fat cells separated by a branching network of uniformly small, thick-walled vessels. The proportion of fatty tissue and vascular channels varies and frequently the vascular component is particularly prominent at the periphery of the tumor. Fibrin microthrombi are common in angiolipomas, a feature that is absent in ordinary lipomas. Hypercellular angiolipomas in which the vascular component predominates may at first glance be confused with Kaposi's sarcoma or angiosarcoma. The encapsulation of the lesion and the characteristic presence of microthrombi, however, point to angiolipoma, a benign neoplasm. 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.12 Although the vast majority occur in the lower extremities or paraspinal region, two cases have been reported in the head and neck.9,10 Both of these cases were in the cervical region and represent the only types of angiolipomas found in the cervical region. Treatment of angiolipomas consists of total surgical excision for both the circumscribed and infiltrating type. Circumscribed lesions almost never recur, whereas infiltrating lesions may recur in 35% to 50% of cases.10,13 REFERENCES 1. Howard WR, Helwig EB. Angiolipoma. Dermatology 1960;82:924-31.

OtolaryngologyHead and Neck Surgery Volume 118 Number 6

Fig. 1. Preoperative photograph.

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Fig. 3. Intermediate magnification photograph of angiolipoma showing a mixture of uniform fat cells and numerous small vessels. (Hematoxylin and eosin stain.)

Fig. 4. At higher magnification, mature fat cells interspersed among branching, thick-walled vessels are demonstrated. (Hematoxylin and eosin stain.)

Fig. 2. Postoperative photograph.

2. Reilly JS, Kelly DR, Royal SA. Angiolipoma of the parotid: case report and review. Laryngoscope 1988;98:818-21. 3. Polte HW, Kolodny SC, Hooker SP. Intraosseous angiolipoma of the mandible. Oral Surg 1976;41:637-42. 4. Lewis DM, Brannon RB, Isaksson B, Larsson A. Intraosseous angiolipoma of the mandible. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1980;50:156-9.

5. Manganaro AM, Hammond HL, Williams TP. Intraosseous angiolipoma of the mandible: a case report and review of the literature. J Oral Maxillofac Surg 1994;52:767-9. 6. Campos GM, Grandi SA, Lopes RA. Angiolipoma of the cheek. Int J Oral Maxillofac Surg 1980;9:486-90. 7. Weitzner S, Moyniham BC. Angiolipoma of the cheek in a child. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1978;45:95-7. 8. Flaggert JJ, Heldt LD, Keaton WM. Angiolipoma of the palate. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1986;61:333-6. 9. Stimpson N. Infiltrating angiolipomata of skeletal muscle. Br J Surg 1971;58:464-6. 10. Lin JJ, Lin F. Two entities in angiolipoma. Cancer 1974;34:720-7. 11. Som PM, Scherl MP, Rao VM, Biller HF. Rare presentations of ordinary lipomas of the head and neck: a review. AJNR Am J Neuroradiol 1986;7:657-64. 12. Pribyl C, Burke SW, Roberts JM, MacKenzie F, Johnston CE. Infiltrating angiolipoma or intramuscular hemangioma? A report of five cases. J Pediatr Orthop 1986;6:172-6. 13. Dionne GP, Seemayer TA. Infiltrating lipomas and angiolipomas revisited. Cancer 1974;33:732-8.