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nial aneurysms of the internal carotid artery with delayed brain infarction. J Nemo1 232:314, 1985 8. New PFJ, Momose KJ: Traumatic dissection of the internal carotid artery at the atlantoaxial level, secondary to nonpenetrating injury. J Neurol 93:41, 1969 9. Davis JM, Zimmerman RA: Injury of the carotid and vertebral arteries. Neuroradiology 2555, 1983 10. Sullivan HG, Vines FS, Becker DP: Sequelae of indirect internal carotid injury. Radiology 109:91, 1973

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11. Moar JJ: Traumatic rupture of the cervical carotid arteries: An autopsy and histopathological study of 200 cases. Forensic Sci Int 34:227, 1987 12. Reyna TM, Cabellon S, Fallon WF: Delayed recognition of carotid artery injury due to blunt trauma. Military Med 151:450, 1986 13. Krajewski LP, Hertzer NR: Blunt carotid artery trauma. Report of two cases and review of the literature. Ann Surg 191:341, 1980

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1990

Cavernous Hemangioma

of the Zygoma:

Report of Cases THOMAS S. JETER, DDS, MD,* FRED L. HACKNEY, THOMAS B. AUFDEMORTE, DDSS

Benign vascular lesions of the osseous structures of the maxillofacial region, ranging from solitary capillary or cavernous hemangiomas to arteriovenous malformations, are rare occurrences.‘*2 The majority of hemangiomas of the facial bones have been reported in the mandible and maxilla,“3 and rarely in the zygoma. 4s This report presents two cases of zygomatic hemangiomas with varied clinical presentations requiring different treatment approaches. Report of Cases

DDS,t AND

blood. A peripheral blood smear and complete blood count of the aspirate showed no platelet sequestration or any other abnormality. The lesion continued to gradually enlarge during the fist week of life, but then stabilized until the 13th day when it markedly enlarged. Examination at that time revealed a 5 x 7 cm ecchymotic mass, resulting in closure of the left eye (Fig 1). No bruits or thrills were noted. Because of the infant’s size, a percutaneous external carotid angiogram was inadvisable. Following a vascular surgical consultation, the patient was taken to the operating room and the left external carotid was exposed. An angiogram performed with a 20-gauge catheter failed to document major arterial anastomoses to the lesion; however, clamping the external carotid produced marked

Case I A l-day-old neonate was seen on consultation for a right incomplete cleft lip. In addition to the incomplete cleft lip, physical examination revealed a l-cm firm enlargement of the left infraorbital rim. The overlying skin was of normal color and texture. Radiographs disclosed a poorly defined loss of cortex in the zygomatic region on the left side. On the third day of life, the infraorbital mass was slightly enlarged and had a firm rubbery consistency. Aspiration at that time produced 3 mL of bright red * In private practice, San Angelo, TX; Clinical Professor, Department of Oral and Maxillofacial Surgery, University of Texas Health Science Center at San Antonio. t In private practice, Lawrenceburg, TN. $ Associate Professor, Department of Pathology, University of Texas Health Science Center at San Antonio. Address corresnondence and reprint requests to Dr Jeter: Department of Oral and Maxillofaciai Surgery, University of Texas Health Science Center at San Antonio, 7703 Floyd Curl Dr, San Antonio, TX 78284. 0 1990 American geons

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of Oral and Maxillofacial

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FIGURE 1. Thirteen-day-old neonate with left infraorbital ecchymotic mass. The left eye is closed.

JETER, HACKNEY,

AND AUFDEMORTE

blanching of the mass. Aspiration of the lesion after the carotid was clamped produced dark red blood and resulted in a smaller, more supple lesion. The left external carotid was ligated and divided. The infraorbital area was then approached with a Weber-Fergusson incision starting at the superior aspect of the incomplete cleft lip. The lesion was bounded by the maxillary alveolus inferiorly, the inferior orbital rim superiorly, the lateral nasal wall medially, and the lateral wall of the zygoma laterally, and extended posteriorly to an area inferior to the orbital apex. The interior of the lesion consisted of delicate septa of bone separating fresh hematomas. The area was curetted and the walls of the defect were covered with Surgicel (Johnson and Johnson Products, New Brunswick, NJ). The remaining defect was packed with benzoin-coated gauze, and the incision was closed. A histologic diagnosis of intraosseous cavernous hemangioma was made. The postoperative course was uneventful, and at 2 weeks postoperatively the gauze pack was removed. At approximately 4 weeks postoperatively, swelling of the left infraorbital area recurred. Aspiration produced 0.5 mL of purulent material. An oral cephalosporin was given empirically pending culture and determination of antibiotic sensitivity. An enterospecies was subsequently identified and found to be sensitive to the cephalosporin. The infection resolved without further intervention. At 3 months of age, a rotation-advancement repair of the incomplete cleft lip was accomplished without complication.6 Six-year follow-up shows no recurrence of the hemangioma. The left midface and maxillary sinus appear to have developed normally (Fig 2), and extraocular movements and vision are intact. Normal eruption of the dentition has occurred, with normal occlusion.

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Case 2 A 50-year-old white woman was referred for evaluation of a mass of the left cheek. The patient had noted the gradually enlarging lesion 1 year previously (Fig 3). Physical examination revealed a 1.5-cm oval, firm lesion of the left infraorbital rim, and radiographs showed an oval radiolucent lesion of the left zygomaticomaxillary complex. Before trephine biopsy of the lesion, aspiration produced 2 mL of dark red blood. A histologic diagnosis of intraosseous cavernous hemangioma was made. preoperative bilateral external and internal carotid angiograms were performed, which revealed no vascular malformations of the left midface. However, a vascular malformation was noted in the left submandibular area that was asymptomatic and not clinically evident. This incidental finding was not treated, but the infraorbital intraosseous lesion was addressed via a subciliary incision. The lesion was found to include portions of the left infraorbital rim, orbital floor, lateral orbital wall, and the body of the zygoma (Fig 4), and was resected in total (Fig 5). Immediate reconstruction with an autogenous iliac bone graft was performed (Fig 6). The postoperative course was uncomplicated. Follow-up at 4 years postoperatively showed no evidence of recurrence of the hemangioma, and the left zygoma has retained proper contour (Fig 7). PATHOLOGY Histologic examination of both lesions revealed cavernous vascular spaces lined with a single layer of uniform endothelial cells containing numerous erythrocytes. The cavernous spaces were bordered by osseous trabeculae (Fig 8). No muscular coated vessels were noted. The findings were consistent with a diagnosis of intraosseous cavernous hemangioma.

Discussion Benign intraosseous vascular lesions range from capillary or cavernous hemangiomas to vascular malformations. The vascular malformation represents an abnormality of blood vessel morphogenesis, resulting in dilation of the vessels.’ In contrast, a hemangioma is a proliferation of endothelial-lined capillaries or cavernous sinuses.2 The intraosseous hemangioma of the facial bones is a rare lesion as compared with soft tissue hemangiomas.’ Two thirds of intraosseous hemangiomas have been re-

FIGURE 2. Six-year follow-up view showing normal development of the midface.

ported in the mandible,iv3 but rarely have they been described in the zygoma.435 This report presents two cases of zygomatic hemangiomas. Intraosseous hemangiomas have a wide range of clinical presentation. Hemangiomas most commonly occur in the second decade of life, with one half of the cases occurring in patients less than 20 years of age.’ The lesions are more common in females, with a 2:l predominance.‘** The cases presented here represent a wide age range, with one

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FIGURE 3. Fifty-year-old woman with a firm mass of the left infraorbital rim.

occurring in a male neonate and one in a 50-year-old woman. Intraosseous hemangiomas of the facial bones often present as an asymptomatic, gradually enlarging swelling. The patient may have been aware of the lesion for several months or years.’ This is consistent with the second case in which the patient had been aware of the gradually enlarging lesion for 1 year, and in sharp contrast to the rapidly progressing hemangioma in the neonate. Some authors have reported that diagnosis based on the radiographic presentation is possible.’ In FIGURE 4. Surgical exposure of the intraosseous hemangioma through a subciliary incision.

FIGURE 5. Osseous defect of the orbit and zygoma following resection of the hemangioma. The maxillary sinus can be visualized through the defect.

FIGURE 6. Bony reconstruction of the surgical defect. Thin strips of cancellous bone were also onlayed to contour small irregularities.

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FIGURE 7. Four-year follow-up view showing proper contour of the left zygoma.

others have reported the radiographic presentation of intraosseous hemangiomas to be a nonspecific radiolucent lesion, which may be solitary, multilocular, or honeycombed.‘.” A radiographic sunburst appearance has also been reported with this lesion.” The radiograph of the neonate showed only poorly defined loss of cortex. If the lesion had not required early treatment, it may have developed a more definite radiolucent appearance with time. The adult presented with a solitary radiolucent lesion consistent with previous reports.’ Intraosseous hemangiomas are benign lesions, and radical therapy is contraindicated unless complications force such treatment. One such complication is the possibility of uncontrollable hemcontrast,

FIGURE 8. Photomicrograph of cavernous spaces lined with a single layer of endothelial cells. Erythrocytes and osseous trabeculae can also be identified. (Hematoxylin-eosin stain. Original magnification, X40.)

orrhage.‘* Additionally, congestive heart failure has been reported in neonates with large hemangiomas.13 The possibility of fatal hemorrhage of the rapidly progressing hemangioma in the neonate prompted treatment. A neonate’s small intravascular volume and subsequent inability to tolerate hemorrhage were important considerations, and were managed by intraoperative angiography and external carotid ligation. Since the angiography failed to demonstrate major vascular anastomoses, embolization of the lesion was not feasible. Other treatment considerations in an infant relate to depletion of platelets (Kasabach-Merritt. syndrome) and clotting factors during excessive bleeding14*15; however, the initial aspirate from the neonate showed no sequestration of platelets, and preoperative coagulation studies were normal. Even with these aggressive procedures, the blood loss in the neonate was 80 mL, over 25% of his estimated blood volume. The SO-year-old woman represented a different therapeutic problem. The biopsy and the angiogram had established that the lesion did not have a direct vascular connection that could result in lifethreatening hemorrhage. Intralesional steroids have been used for superficial hemangiomas; however, complications have been reported with periorbital lesions treated in this manner.‘6V’7 Some authors have suggested a policy of noninterference if the lesions are asymptomatic and static.” However, this lesion was slowly enlarging and represented a cosmetic defect for the patient. The goals of treatment in this case were resection and osseous reconstruction to restore normal bony contours.

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It is interesting to note that each patient presented with a second congenital malformation. The neonate had an incomplete cleft lip and the adult had a submandibular vascular malformation. A review of the literature revealed little documentation of the incidence of concomitant congenital defects and intraosseous hemangiomas of the facial bones. Summary Osseous hemangiomas of the facial bones are rare lesions, with most cases occurring in the maxilla and mandible. Reported cases in the zygoma are sparse. Two cases of zygomatic hemangiomas with widely differing presentations are reported. One case in a neonate required surgery to prevent fatal hemorrhage, and the other in an adult required correction of a cosmetic deformity. Each patient presented with a second congenital anomaly. Therapeutic problems associated with these lesions and their management are discussed. References 1. Batsakis JG: Tumors of the Head and Neck, Clinical and Pathological Considerations (ed 2). Baltimore, MD, Williams & Wilkins, 1979, pp 390-398 2. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 3). Philadelphia, PA, Saunders, 1974, p 104

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3. Pierce11 MP, Waite DE, Nelson RL: Central hemangioma of the mandible: Intraoral resection and reconstruction. J Oral Surg 33:225, 1975 4. Walker EA, McHenry LC: Primary hemangioma of the zygoma. Arch Otolaryngol 81:199, 1965 5. Most DS: Hemangioma of the maxillary sinus. Oral Surg Oral Med Oral Path01 60:485, 1985 6. Millard DR: Retinements in rotation-advancement cleft lip technique. Plast Reconstr Surg 33:26, 1964 7. Kaban LB, Mulliken JB: Vascular anomalies of the maxillofacial region. J Oral Maxillofac Surg 44:910, 1986 8. Dahlin DC: Bone Tumors, General Aspects and Data on 6221 Cases (ed 3). Springfield, IL, Thomas, 1978 9. Bucy PC, Capp CS: Primary hemangioma of bone with special reference to roentgeonlogic diagnosis. Am J Roentgenol Radium Ther Nucl Med 23: 1, 1930 10. Selfe RW, Sherman M, Miller TF: Arteriovenous malformation of the mandible. ORL J Otorhinolaryngol Relat Spec 86:659, 1978 11. Loring MF: Hemangioma of the mandible, diagnosis and therapy. Arch Otolaryngol 85648, 1967 12. Sklar G, Meyer I: Vascular tumors of the mouth and jaws. Oral Surg 19:335, 1965 13. Vaksmann G, Rey C, Marache P, et al: Severe congestive heart failure in newborns due to giant cutaneous hemangiomas. Am J Cardiol60:392, 1987 14. Way LW: Current Surgical Diagnosis and Treatment (ed 7). Los Altos, CA, Lange, 1985, p 1092 15. Mulliken JB: Cutaneous vascular lesions of children, in Se&in D, Georgiade NG (eds): Pediatric Plastic Surgery. St Louis, MO, Mosby, 1984, pp 137-154 16. Sutula FC, Glover AT: Eyelid necrosis following intralesional corticosteroid injection for capillary hemangioma. Ophthalmic Surg 18: 103, 1987 17. Droste PJ, Ellis FD, Sondhi N, et al: Linear subcutaneous fat atrophy after corticosteroid injection of periocular hemangiomas. Am J Ophthalmol 103:65, 1987

Surg

1990

Angiolipoma

of the Cheek:

Report of a Case GREGORIO

SANCHEZ ANICETO, MD,* RAFAEL SALVAN SAEZ, MD,t AND APOLINAR GARCiA PEfiIN, MDt

Lipoma is a very common benign tumor composed of mature fat cells, usually with a slow

Received from the Oral and Maxillofacial Surgery Unit, Hospital 12 de Octubre, Madrid, Spain. * Senior resident. t Staff surgeon. Address correspondence and reprint requests to Dr Sanchez Aniceto: Paseo Santa Maria de la Cabeza N. 68-B, 4-C Madrid, 28045 Spain. 0 1990 American geons

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growth pattern. Angiolipoma is a variant of lipoma, with a prominent vascular component, commonly appearing on the trunk and extremities of young adults. In a series of 268 cases of angiolipoma, Howard and Helwig’ found that all of them occurred in patients older than 16 years, and all were located on the trunk and extremities. It is very uncommon for this tumor to occur in the maxillofacial region, with only a few cases reported in the literature: one case of angiolipoma of the cheek in a 4-year-old boy;2 two cases of intraosseous angiolipoma of the mandibles of a 39-year-old man3 and a 56-year-old woman4 and a case of an angiolipoma