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.

Large Orbital Pediatric Intraosseous Hemangioma

“…Primary orbital intraosseous hemangiomas in children are extremely rare. A recent case report and literature review identified 49 reported cases of zygomatic intraosseous hemangioma; only five of the patients were under the age of 18 at diagnosis and only seven were noted to have ocular findings [11]. None of these cases documented a lesion of our size (5 cm × 6 cm) in the zygomatic location.…”

“…Biopsy specimens without incorporating the bony component can also be nondiagnostic. Histologic subtypes have been classified as either cavernous, capillary, mixed, or scirrhous on the basis of the size of the vascular spaces and amount of connective tissue within the lesion 2 Case Reports in Ophthalmological Medicine [11,18]. While the diagnosis is often suspected on imaging features and gross examination intraoperatively or of the resected specimen, histopathologic examination is useful in differentiating these tumors from other vascular malformations and immunohistochemical analysis for Factor VIII, vimentin, CD31, and GLUT-1 may be appropriate in select cases [12,19,20].…”

“…Given the age of our patient and both the invasiveness of angiography and the need for general anesthesia for the study, preoperative angiography was not pursued. Significant blood loss in the surgical resection of intraosseous hemangiomas has been well documented [9][10][11][12]. Our small patient's body weight was 19.3 kilograms at the time of his surgery, with an estimated blood volume (EBV) of 1351 milliliters (EBV = body weight ð19:3 kgÞ × average blood volume of child ð70 ml/kgÞ).…”

“…Involvement of the facial bones is less common [8]. It is important to consider the possibility of this rare tumor preoperatively as life-threatening blood loss during surgical excision can be encountered [9][10][11][12].…”

Computer guided resection and reconstruction of intra-osseous zygomatic hemangioma: Case report and systematic review of literature

“…Treatment options included surgical interference in the form of total tumor resection through narrow safety margin with or without reconstruction. Also, non-surgical modality in the form of follow-up after confirming the diagnosis was reported [6]. Reconstruction of the residual zygomatic defects poses a challenge to the surgeons to provide the best esthetic results with a wide variety of treatment options including autogenous bone grafts harvested from iliac crest, rib, and calvaria.…”