title: Blindness as a complication of Le Fort I osteotomy for maxillary distraction.
abstract: High Le Fort I osteotomy and maxillary distraction has become an accepted method for the treatment of maxillary retrusion in children and teenagers with cleft lip and palate or craniofacial anomalies. This procedure effectively corrects the dentofacial deformity in these patients. No major surgical morbidity has been reported. During the past 4 years, 94 cleft patients with maxillary hypoplasia received Le Fort I osteotomy and distraction osteogenesis at the authors' center. Two of them developed blindness after this operation. The first case was a girl with bilateral cleft lip and palate with median facial dysplasia. She received high Le Fort I osteotomy at age 12 years 4 months to correct maxillary retrusion. Right eye swelling and ecchymosis was found after surgery. The patient complained of vision loss in that eye 2 days later. Computed tomography showed subarachnoid hemorrhage and skull base hematoma. There were no atypical fractures in the orbit, pterygoid plates, sphenoid bone, and skull base. Angiogram revealed left ophthalmic and basilar artery aneurysm. The second case was a 12-year-old boy with left cleft lip and palate. He received Le Fort I osteotomy to correct maxillary retrusion. During surgery, abnormal pupil dilatation was found after the osteotomy and down-fracture of maxilla. Emergent computed tomography found no hemorrhage or atypical fractures. Examination revealed complete left optic neuropathy and partial right abducens nerve palsy with mydriasis. Magnetic resonance imaging, magnetic resonance angiography, and repeated computed tomography revealed no sign of orbital injury, vascular problem, or abnormal fractures. The cause of blindness was unknown. In both cases, a steroid was used. Maxillary distraction was continued. Recovery of meaningful visual sense did not occur after 3 and 2 years' follow-up, respectively. A review of the literature revealed five other patients who suffered from visual loss after Le Fort I osteotomy. Inadvertent skull base fractures were identified in two cases, but a cause for the blindness was not known in the others. Induced hypotension and indirect trauma may be responsible for the optic nerve injury. In none of the cases was meaningful visual sense recovered, although high-dose steroids were given. In conclusion, a total of seven cases developed blindness after Le Fort I osteotomy. Once blindness develops, the prognosis is poor. High Le Fort I osteotomy should be performed with extreme care, and perhaps the informed consent should include visual loss as a complication of the procedure.
authors.affiliation: Department of Plastic and Reconstructive Surgery, Chang Gung Memorial Hospital, Taipei, Taiwan. lunjoulo@ms1.hinet.net
authors.forename: Lun-Jou
authors.initials: LJ
authors.lastname: Lo
authors.forename: Kai-Fong
authors.initials: KF
authors.lastname: Hung
authors.forename: Yu-Ray
authors.initials: YR
authors.lastname: Chen
mesh_terms: D020434:Abducens Nerve Diseases / Q000209:etiology; D000293:Adolescent; D001766:Blindness / Q000209:etiology*; D002648:Child; D002971:Cleft Lip / Q000150:complications; D002972:Cleft Palate / Q000150:complications; D005260:Female; D006801:Humans; D002532:Intracranial Aneurysm / Q000150:complications / Q000209:etiology; D008297:Male; D008437:Maxilla / Q000002:abnormalities / Q000601:surgery*; D009901:Optic Nerve Diseases / Q000209:etiology; D019857:Osteogenesis, Distraction / Q000009:adverse effects*; D019340:Osteotomy, Le Fort / Q000009:adverse effects*; D013345:Subarachnoid Hemorrhage / Q000209:etiology