Mydriasis, either bilateral or unilateral, seldom occurs during reconstruction of periorbital fracture. Anisocoria, a unilateral mydriasis, requires more urgent assessment than bilateral mydriasis does. Pharmacologic agents, local anesthetic infiltration, as well as direct or indirect oculomotor nerve damage are possible causes of unilateral mydriasis. Few cases have been reported about intraoperative temporary ipsilateral mydriasis during correction of blowout fracture. We have experienced an unusual case of anisocoria and report the case with literature reviews.