Zygomatic Intraosseous Hemangioma: Case Report and Literature Review

Abstract

Intraosseous hemangiomas are uncommon intrabony lesions, representing approximately 0.5 to 1% of all intraosseous tumors. Their description varies from "benign vasoformative neoplasms" to true hamartomatous proliferations of endothelial cells forming a vascular network with intermixed fibrous connective tissue stroma. These commonly present as a firm, painless swelling. Intraosseous hemangiomas present more commonly in females than in males and most likely occur in the fourth decade of life. The most common etiology of intraosseous hemangioma is believed to be prior trauma to the area. They have a tendency to bleed briskly upon removal or biopsy, making preoperative detection of the vascular nature of the lesion of significant importance. There are four variants: (1) capillary type, (2) cavernous type, (3) mixed variant, and (4) scirrhous type. Generally most common in the vertebral skeleton, they can also present in the calvarium and facial bones. In the head, the most common site is the parietal bone, followed by the mandible, and then malar and zygomatic regions. Intraosseous hemangiomas of the zygoma are rare entities with the first case reported in 1950 by Schoenfield. In this article, we review 49 case reports of intraosseous hemangioma of the zygoma, and also present a new case treated with excision followed by polyether-ether ketone implant placement for primary reconstruction.

Keywords: PEEK implant; intraosseous hemangioma; zygoma.

Fig. 1

(a) Initial presentation of patient. (b) Lateral view of right zygoma showing expansion/prominence. (c) Inferior view of right zygoma displaying obvious expansion.

Fig. 2

(a) Initial presentation: computed tomographic (CT) coronal view indicating right zygoma expansion. (b) Initial presentation: CT axial view. (c) Initial presentation: 3D reconstruction indicating site of apparent intraosseous hemangioma.

Fig. 3

(a) Low-power histopathologic view displaying lesion with spindled cells and abundant pale cytoplasm. (b) Medium-power histopathologic view with better representation of spindled cells, abundant pale cytoplasm, and numerous erythrocytes. (c) High-power histopathologic view outlining vascular channels with generally smooth endothelial lining, scattered erythrocytes, and spindled cells.

Fig. 4

(a) Intraoperative view of intraosseous hemangioma. (b) Postresection of lesion. (c) Preoperative stereolithographic model indicating planned borders of resection. (d) Template for custom PEEK implant. (e) Custom PEEK implant mirrored from contralateral side reproducing the bony contours of the zygoma.

Fig. 5

(a) 18-month postoperative view of the patient. (b) 18-month postoperative close-up of the bilateral orbitozygomatic region. (c) 18-month postoperative inferior view indicating acceptable uniformity of zygoma projection. (d) 18-month postoperative computed tomographic (CT) scan with 3D reconstruction. (e) 18-month postoperative CT axial scan indicating no evidence of recurrence of lesion.