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A 37-year-old woman presented with a rare cavernous malformation of the ventral midbrain with brainstem hemorrhage manifesting as sudden onset of headache and vomiting. The lesion was removed successfully through a transsylvian approach and a medial peduncular route. Postoperatively, her oculomotor nerve paresis worsened temporarily, but diplopia disappeared 2 months after surgery. We recommend the transsylvian-transpeduncular approach if the lesion is located in the ventral midbrain and facesdoi:10.2176/nmc.48.569 pmid:19106496 fatcat:njkp3pbtinay3l3h4nx6iyzapq