We herein describe a 49-year-old woman with a history of severe headache for two weeks and vomitting which in turn was approved to be secondary to unilateral venous caudate nucleus infarction. Conventional and contrast enhanced magnetic resonance imaging (MRI) sequences besides Magnetic Resonance Venography (MRV) confirmed the thrombosis of sinus rectus and superior sagittal sinuses. Diffusion-weighted MRI revealed mild restricted diffusion. To the best of our knowledge it is the first case of unilateral venous infarction of the caudate nucleus described in the literature.

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