A patient with a ruptured left sylvian fissure arteriovenous malformation (AVM) treated with primary transvenous Onyx embolization, and angiographically cured, is reported. The patient presented with a large intraparenchymal subopercular hematoma and there was angiographic demonstration of a pseudoaneurysm and single draining vein. Trans-arterial approaches were unsuccessful because of the en-passage nature of feeding arteries and hence a transvenous approach was used to embolize the pseudoaneurysm with Onyx. Follow-up angiography at 1 month revealed durable angiographic cure of the AVM. Our case illustrates that in patients with ruptured AVM secondary to pseudoaneurysm and single draining vein, transvenous treatment can be utilized to achieve occlusion of the pseudoaneurysm resulting in AVM cure. To our knowledge, this is the first description of a transvenous approach for endovascular therapy of AVM.