Angiographically documented aneurysms of the intraorbital ophthalmic artery are extremely rare. Two cases are presented of intra-orbital ophthalmic artery aneurysms, both associated with arteriovenous malformation (AVM). There have only been isolated reports documenting this combination of vascular anomalies. In the first case, the aneurysm was treated conservatively and in the second case, after staging angiography and interventional embolization, the aneurysm was removed along with the associated AVM. The clinical presentation, pathogenesis and management of this rare combination of vascular anomalies is briefly discussed.
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