Fibrous dysplasia is a benign, developmental bone disorder of unknown aetio
logy. With involvement of the facial bones, indications for surgical interv
ention include cosmetic deformity and/or progressive neurological dysfuncti
on. We present the case of a 36-year-old female with bilateral orbital lesi
ons whose initial presentation was that of progressive proptosis and visual
loss. She underwent bilateral pterional craniotomies with gross total rese
ction of her rumours. Review of the literature reveals several cases of uni
lateral orbital fibrous dysplasia. This appears to be the first reported ca
se of distinct, bilateral orbital fibrous dysplasia.