Witzelsucht is a tendency to tell inappropriate and poor jokes. It usually occurs after a focal lesion involving orbitofrontal cortical or paramedian thalamic regions, especially on the right side. Here we report a 56-year-old man developing witzelsucht and hypersexuality after a right putaminal hemorrhage. The hematoma extended to the sublenticular part of posterior internal capsule and mesencephalon. The hemorrhage might have disconnected the fibers in the ascending reticular systems, and the fibers between paramedian thalamus and orbitofrontal cortex, and thus could be responsible for the patient's rare clinical manifestations.