Summary: Le Fort I osteotomy is a frequent surgical procedure used in orthognathic surgeries to treat severe malocclusions and is associated with relatively rare surgical complications. Here, the authors report a case of thrombotic ischemic stroke as a result of this procedure, a complication still not described in the literature. A 19-year-old man with class II malocclusion and retrognathia underwent orthognathic surgery for aesthetic purposes. The surgery included a Le Fort I maxillary osteotomy with vertical impaction, bilateral sagittal split ramus osteotomy for mandibular advancement, and genioplasty. Postoperatively, the patient developed left eye blindness, headache, somnolence, aphasia, and right hemiplegia. Medical imaging showed the Le Fort I line of fracture extending from the maxillary osteotomy to the left optic canal and to the left carotid canal, with osseous fragments impinging the petrous segment of the internal carotid artery, left carotid artery occlusion and associated to an ischemic stroke at the left middle cerebral artery territory. Treatment required decompressive craniectomy and later focused on clinical stabilization, infection management, orthognathic care, neurorehabilitation, and cranioplasty. The hemiplegia and aphasia partially recovered during 12 months, and final dental occlusion was appropriate. Our report demonstrates that an unfavorable Le Fort I fracture trajectory can lead to ischemic stroke and severe neurological deficits.