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JPRAS Open 6 (2015) 5e10 Contents lists available at ScienceDirect JPRAS Open journal homepage: http://www.journals.elsevier.

University Medical Center East, Tokyo, Japan article info abstract Article history: Received 15 July 2015 Accepted 29 July 2015 Available online 13 August 2015 Keywords: Intraosseous hemangiomas

1 JPRAS Open 6 (2015) 5e10 Contents lists available at ScienceDirect JPRAS Open journal homepage: jpras-open Case report Intraosseous hemangioma of the zygomatic bone Junji Hishiyama *, Tsukasa Isago, Hiroshi Ito Department of Plastic and Reconstructive Surgery, Tokyo Women's Medical University Medical Center East, Tokyo, Japan article info abstract Article history: Received 15 July 2015 Accepted 29 July 2015 Available online 13 August 2015 Keywords: Intraosseous hemangiomas Zygomatic bone Cavernous hemangioma Angiography Embolization therapy Intraosseous hemangiomas are rare, and although they have been described in the calvaria and vertebrae, those occurring in the zygomatic bone are extremely rare. We report the case of a 52- year-old man who presented with a painless hard swelling in the left zygomatic process. The computed tomography and magnetic resonance imaging findings showed an intraosseous mass located in the left zygomatic bone just inferolateral to the orbit. A pathologic analysis of the surgical specimen revealed a cavernous hemangioma. The surgical defect was reconstructed with an autogenous rib bone that was fixated with titanium miniplates. The literature is reviewed and computed tomography, magnetic resonance imaging, and angiographic findings are discussed with particular reference to a surgical resection and reconstruction The Authors. Published by Elsevier Ltd on behalf of British Association of Plastic, Reconstructive and Aesthetic Surgeons. This is an open access article under the CC BY license ( creativecommons.org/licenses/by/4.0/). Introduction Intraosseous hemangiomas are usually seen in the calvaria and vertebrae. When they arise within the calvaria, they are normally confined to the frontal or parietal bones. 1 The involvement of the maxillofacial skeleton is infrequent. The mandible, maxilla, and nasal bones are the most frequently affected sites. 2 Zygomatic involvement is extremely rare, and only 23 cases have been reported in the Englishlanguage literature. 1e10 Both types of hemangioma (capillary and cavernous) may be encountered in * Corresponding author. Department of Plastic and Reconstructive Surgery, Nishi-Ogu, Arakawa-ku, Tokyo , Japan. Tel.: þ ; fax: þ address: hishiyama.junji@twmu.ac.jp (J. Hishiyama) / 2015 The Authors. Published by Elsevier Ltd on behalf of British Association of Plastic, Reconstructive and Aesthetic Surgeons. This is an open access article under the CC BY license (

2 6 J. Hishiyama et al. / JPRAS Open 6 (2015) 5e10 the zygomatic bone. Intraosseous hemangiomas are seen 3 times more often in women than in men. Although there is not an age predilection, they tend to occur more frequently in the third and fourth decades. 3e5 We present a case of a cavernous hemangioma arising from the zygomatic bone as documented by computed tomography (CT), magnetic resonance imaging (MRI), and angiographic findings. Case report A 52-year-old man presented with a 6-year history of a painless hard swelling in the left zygomatic process, which had gradually been becoming bigger and harder. There was a vague history of a fall, in which he had banged his face on the floor 7 years previously. On examination there was a 3-cm-diameter dome-shaped, nontender bony hard swelling in the body of the left zygomatic bone, with normal overlying skin. There were no unusual signs in the eye and, in particular, no abnormality in vision, ocular movement, or the position of the globe. In addition, no regional paresthesia was observed. Plain radiographs showed an oval radiopaque mass in the body of the left zygomatic bone. CT showed a 3-cm-diameter circumscribed mass in the zygomatic bone with a bulging area in the inferolateral aspect of the orbit anteriorly. There was no involvement of the soft tissue and no periosteal reaction. MRI of the paranasal sinuses confirmed the CT localization of the lesion (Figure 1). The Figure 1. A CT scan shows the hypointense intraosseous mass in the left zygoma with a honeycomb like appearance (upper). An axial T2-weighted MR image. The encroachment on the inferolateral aspect of the orbit is seen (lower).

3 J. Hishiyama et al. / JPRAS Open 6 (2015) 5e10 7 mass had an overall intermediate T1 signal intensity and a high T2 signal intensity. Within the mass, areas of a signal void that corresponded to the trabeculae were seen in a CT study but no extraosseous soft tissue component was apparent. The preliminary diagnosis, based on the CT and MRI appearance, was intraosseous hemangioma of the zygomatic bone. A carotid arteriogram showed a hypervascular lesion in the left zygomatic bone. The lesion was predominantly supplied by the left external carotid artery via the infraorbital and posterosuperior alveolar arteries, the anterior deep temporal artery, and the left facial artery. Supraselective embolization of these vessels was performed with poly (vinyl alcohol) particles, thus resulting in nearly complete devascularization (Figure 2). With these findings, the patient was scheduled for surgery. On surgical exploration, a slightly irregular, subcortical zygomatic mass with a bluish tinge was identified. The mass was located in the anterior half of the zygomatic bone extending to the orbit. After complete excision of the mass with safety margins, the defect in the zygomatic bone was reconstructed with use of an autogenous rib bone that was fixated with titanium miniplates (Figure 3). The histological findings showed mature, lamellar trabecular bone, in which the intratrabecular space was completely occupied by large, dilated, thin-walled vessels, consistent with a cavernous hemangioma of the bone (Figure 4a). Figure 2. A preoperative carotid arteriogram (upper). Carotid arteriogram after supraselective embolization. A hypovascular lesion in the zygoma is seen (lower).

4 8 J. Hishiyama et al. / JPRAS Open 6 (2015) 5e10 Figure 3. Intraoperative finding (upper). The surgical defect was reconstructed with an autogenous rib bone that was fixated with titanium miniplates (lower). One year after the operation, no recurrence of the tumor and no cosmetic deformity have been observed. The CT scan reveals how well the rib block fits (Figure 4b). Discussion Hemangiomas are benign lesions of endothelial origin. Histopathologically, they are classified as cavernous or capillary type according to their vascular network. A cavernous hemangioma is composed of large thin-walled vessels and sinusoids lined with a single layer of endothelium. However, a capillary hemangioma is formed by a small fine vascular network filled with blood. Usually these two components are seen together as a mixed hemangioma. 8 In most cases, they are present at birth, whereas in some cases trauma is thought to be the initiating factor. Intraosseous hemangiomas are rare and account for less than 1% of all bony tumors. 1 The sites most commonly involved are the vertebral column (excluding the vertebral bodies) and the skull. Within the calvarium, the parietal bone is most commonly involved, followed by the frontal bone, but lesions also occur in the base of the skull and in particular the occiput and petrous temporal bone. 1 Within the facial skeleton, intraosseous hemangiomas occur in the mandible, maxilla, and nasal bones, but they are extremely rare in the zygomatic bone. 2 To the best of our knowledge, only 23 cases have been reported in the English-language literature. 1e10

5 J. Hishiyama et al. / JPRAS Open 6 (2015) 5e10 9 Figure 4. A microscopic view of the specimen (hematoxylin & eosin, 150, upper). Postoperative CT findings showed no evidence of tumor recurrence and good fit for the autogenous rib graft (lower). CT is considered the most useful imaging technique because of its excellent characterization of the trabecular and cortical details. The CT appearance is variable, and in the calvaria most commonly shows a characteristic sharply marginated expansile lesion with intact inner and outer tables and a sunburst pattern of radiating trabeculae. Soap bubble and honeycomb configurations may also occur. 10 MRI provides information regarding any associated soft tissue elements. The magnetic resonance signals characteristic of a hemangioma are dependent on the quantity of slow-moving venous blood and on the ratio of red marrow to converted fatty marrow present within the lesion. Low or high signal may be seen on the T1-weighted images, and also high signal may be seen on water-sensitive sequences. 10 The usefulness of preoperative angiography and selective embolization has been debated in the literature. Although many authors recommend either embolization or external carotid ligation, 1,3,4,7 others state that proximal vascular control is unnecessary if an adequate margin of normal bone is resected. 2,9 In our case, we felt that preoperative embolization limited the bleeding and optimized a clean field for resection. In surgical treatment, an immediate reconstruction is important to avoid any postoperative deformity. The zygomatic bone is a keystone in facial aesthetics, and refinements in facial symmetry and contour must be considered in its reconstruction. Various reconstruction methods have been used, including a resection without repair, and repair with a Surgicel pack, a silicone implant, hydroxyapatite, an autogenous rib graft, pedicled infratemporal fossa fat, and autogenous free iliac bone graft. 1,2,5e7 An

6 10 J. Hishiyama et al. / JPRAS Open 6 (2015) 5e10 easily harvested autogenous bone graft is one of the most popular sources. Our case had a 1-year clinical and radiological follow-up. The results have been satisfactory from both a functional and a morphological standpoint. Conflict of interest Not applicable. Funding Not applicable. Ethical approval Not applicable. References 1. Warman S, Myssiorek D. Hemangioma of the zygomatic bone. Ann Otol Rhinol Laryngol. 1989;98:655e Konior R, Kelley T, Hemmer D. Intraosseus zygomatic hemangioma. Otolaryngol Head Neck Surg. 1999;121:122e Clauser L, Menenghini A, Riga M, Rigo L. Hemangioma of the zygoma: report of two cases with a review of the literature. J Craniomaxillofac Surg. 1991;19:353e Cuesta Gil M, Navarro-Vila C. Intraosseous hemangioma of the zygomatic bone. Int J Oral Maxillofac Surg. 1992;21:287e Davis E, Morgan L. Hemangioma of bone. Arch Otolaryngol Head Neck Surg. 1974;99:443e Hirano S, Shoji K, Kojima H, Omori K. Use of hydroxyapatite for reconstruction after surgical removal of intraosseous hemangioma in the zygomatic bone. Plast Reconstr Surg. 1997;100:86e Jeter T, Hackney F, Aufdemorte T. Cavernous hemangioma of the zygomatic bone: report of cases. J Oral Maxilofac Surg. 1990; 48:508e Koybasi S, Saydam L, Kutluay L. Intraosseous hemangioma of the zygoma. Am J Otolaryngol. 2003;24:194e Marshak G. Hemangioma of the zygomatic bone. Arch Otolaryngol. 1980;106:581e Moore S, Chun J, Mitre S, Som P. Intraosseous hemangioma of the zygoma: CT and MR findings. Am J Neuroradiol. 2001;22: 1383e1385.

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