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<p><strong>Background</strong>:&nbsp;Ipsilateral hemiballismus refers to the rare occurrence of hemiballism developing on the same side of a brain lesion.</p><p><strong>Case report</strong>:&nbsp;We describe a rare case of postoperative ipsilateral hemiballism in a patient who underwent pituitary adenoma resection and experienced a right internal cerebral artery territory infarct. We review the literature on hemichorea hemiballismus (HCHB) and explore various mechanisms for its occurrence.</p><p><strong>Discussion</strong>:&nbsp;Only three cases of ipsilateral hemiballism have been described, and the exact pathophysiology remains unknown. A dominant left hemisphere with corpus callosal connections to the right basal ganglia is the most probable explanation for this unusual event.</p><p>&nbsp;</p>