Appropriate operations in severe anterior open bite (AOB) cases are extremely complicated to perform because of the multiple surgical procedures involved, the difficulty of predicting posttreatment aesthetics, and the high relapse rate. We herein report a 16-year-old girl with skeletal Class II, severe AOB malocclusion, and crowding with short roots, and aesthetic and functional problems. Four-piece segmental Le Fort I osteotomy with horseshoe osteotomy was performed for maxillary intrusion, and bilateral sagittal split ramus osteotomy (SSRO) and genioplasty were performed for mandibular advancement. The malocclusion and skeletal deformity were significantly improved by the surgical orthodontic treatment. Functional and aesthetic occlusion with an improved facial profile was established, and no further root shortening was observed. Acceptable occlusion and dentition were maintained after a two-year retention period. This strategy of surgical orthodontic treatment with a complicated operative procedure might be effective for correcting certain severe AOB malocclusion cases.