BACKGROUND In general, venous aneurysm associated with dural arteriovenous fistula (dAVF) is considered to be developed under long standing venous hypertension and manifested as venous ectasia of draining vein itself. However, discrete saccular shaped venous aneurysm without angiographic evidence of venous hypertension arising from the draining vein, like cerebral arterial aneurysm, is quite rare and its pathomechanism remains unclear in patients with dAVF. CASE SUMMARY In this report, we present two cases of ruptured saccular venous aneurysms associated with dAVF without venous hypertension or venous ectasia. In both cases, significant curve or stenosis is observed in draining vein, which is located in just distal portion of the venous aneurysms. These aneurysms were successfully treated with a transarterial embolization. Underlying mechanism of venous aneurysms in these cases is discussed. CONCLUSION Although there is little doubt that hemodynamic stress has a critical role in the development of venous aneurysms in patients with dAVF, preceding venous hypertension or venous ectasia is not necessary for development and enlargement of venous aneurysms. Considering the significant risk of rupture, a careful review of draining vein features including tortuosity or stenosis is needed, especially in venous aneurysms without evidence of venous hypertension.