This study aims to report a rare complication, anisocoria (mydriasis), in a patient with cleft lip and palate in the immediate postoperative period after orthognathic surgery and discuss the literature surrounding this event. Mydriasis after orthognathic surgery may be related to the separation of the pterygoid plate from the sphenoid bone, which can injure the cranial nerves. Therefore, knowledge of possible anatomical variations of the posterior region of the maxilla becomes extremely important to minimize complications in cleft patients. The reported case highlights the management of anisocoria as a complication as well as the importance of its knowledge.
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