Intraosseous ‘haemangioma’ of the zygoma: More appropriately termed a venous malformation

doi:10.1016/j.ijom.2009.05.010

International Journal of Oral and Maxillofacial Surgery

Volume 38, Issue 10, October 2009, Pages 1066-1070

https://doi.org/10.1016/j.ijom.2009.05.010 Get rights and content

Abstract

Vascular anomalies often affect the soft tissues and primary intraosseous lesions are uncommon, with only 33 cases reported in the zygoma. Intraosseous vascular anomalies of the zygoma have traditionally been designated ‘haemangiomas’ with little attempt to clarify the specific type of the lesion. Recently, specific immunohistochemical markers such as erythrocyte type glucose transporter protein 1 (GLUT-1) have been described to differentiate haemangiomas and vascular malformations. The authors report a case of intraosseous venous malformation of the zygoma and provide supporting evidence to justify the nomenculature. The literature on zygomatic ‘haemangiomas’ is critically reviewed and the basis for their proper designation as venous malformations highlighted.

Section snippets

Case report

A 66-year-old woman presented with a gradually enlarging asymptomatic swelling of the right cheek prominence of 4 years duration. The patient was unable to recall the presence of the lesion in childhood and gave no history of previous trauma. Clinical examination revealed a 3 × 3 cm well-defined, bony hard mass involving the body of the right zygoma. A CT scan revealed a well-marginated benign appearing bony mass extending anteriorly from the body of the zygoma, demonstrating intact inner and

Histology

Routine haematoxylin and eosin sections demonstrated mature bony trabeculae separated by a network of thin-walled, dilated vascular spaces lined by flattened endothelial cells demonstrating no mitotic activity and lacking a uniform muscular layer (Figure 2). Immunohistochemistry did not reveal any uptake of GLUT-1 (Figure 3) and the smooth muscle actin stain demonstrated smooth muscles in the walls of the abnormal veins lining the vascular spaces (Figure 4). Based on the history, clinical

Discussion

Congenital vascular lesions are divided into haemangioma and vascular malformations, which exhibit unique characteristics and demand appropriately tailored treatment plans7, 12, 18. Haemangiomas often appear after birth (up to 30% are present at birth) and are characterised by a rapid proliferative stage in the very early years followed by a prolonged involutive phase until the teenage years. This involution may not be complete and the patient may be left with a fibrofatty residuum7, 19.

Competing interests

None declared

Funding

None

Ethical approval

Not required

Acknowledgements

We would like to thank Ms Becky Ward, Medical Photography Department, Poole General Hospital, for help with the illustrations.

References (31)

J.B. Boyd et al.
Skeletal changes associated with vascular malformations
Plast Reconstr Surg
(1984)
N.C. Cheng et al.
Intraosseous haemangiomas of the facial bone
Plast Reconstr Surg
(2006)
L. Clauser et al.
Haemangioma of the zygoma
Report of two cases with review of literature. J Craniomaxillofac Surg
(1991)
M. Cuesta Gil et al.
Intraosseous haemangioma of the zygomatic bone
Int J Oral Maxillofac Surg
(1992)
E. Davis et al.
Haemangioma of bone
Arch Otolaryngol
(1974)
O. Enjolras et al.
Non involuting congenital haemangioma: a rare vascular anomaly
Plast Reconstr Surg
(2001)
M. Ethunandan et al.
Haemangioma and vascular malformations of the maxillofacial region-a review
Br J Oral Maxillofac Surg
(2006)
L.R. Fernandez et al.
Radiographic features of osseous haemangioma in the maxillofacial region. Bibliographic review and case report
Med Oral
(2003)
G. Har-El et al.
Hemangioma of the zygoma
Ann Plast Surg
(1987)
T.S. Jeter et al.
Cavernous haemangioma of the zygoma: report of cases
J Oral Maxillofac Surgery
(1990)

A.K. Greene et al.

Intraosseous “Haemangiomas” are malformations and not Tumours (letter)

Plast Reconstructive Surg

(2007)

L.B. Kaban et al.

Vascular anomalies of the maxillofacial region

J Oral Maxillofac Surg

(1986)

R.J. Konior et al.

Intraosseous zygomatic haemangioma

Otolaryngol Head Neck Surg

(1999)

S. Koybasi et al.