Direct carotid-cavernous sinus fistulas that present with signs and sy
mptoms contralateral to the arterial supply of the fistulas are not un
common. We present a thoroughly documented case of a dural-cavernous s
inus fistula with symptoms exclusively contralateral to the arterial s
ource, a rarer entity. The patient presented with a red, proptotic rig
ht eye and a history of transient horizontal diplopia and a ''feeling
of fullness'' in that eye. Magnetic resonance imaging (MRI) of the bra
in and orbits performed at another hospital had shown no abnormalities
. Carotid angiography performed on the right side was normal; carotid
angiography performed on the left side showed a dural-cavernous sinus
fistula, with shunting from branches of the left external carotid arte
ry directly to the right cavernous sinus. Orbital duplex color-flow so
nography showed reverse flow in a dilated right superior ophthalmic ve
in. This unusual manifestation of a dural-cavernous sinus fistula offe
rs insight into the pathophysiology of arteriovenous fistulas involvin
g the cavernous sinus, and is a reminder that bilateral injections are
required when performing carotid angiography to characterize these di
sorders.