In general, the skeletal class III has the characteristics of mandibular overgrowth with a normal maxillary growth or maxillary undergrowth with a normal mandibular growth And clinical and radiographic evaluations of the patient are needed. However, the treatment plan is not dependent on these evaluations alone, because patient's general condition and hope for aesthetics varies. The aim of this report is to consider the treatment of a medically compromised patient with an anterior open bite and skeletal class III, which showed a severe mandibular overgrowth. In 2003, a 17-year-old boy with epilepsy, mental retardation presented at our clinic complaining of concave profile. A clinical examination showed severe mandibular prognathism with an anterior open bite. The radiographic examination revealed a short cranial base, a moderate maxillary overgrowth, severe mandibular overgrowth and skeletal open bite tendency. In 2004, he was verified to have no potential of growth by hand-and-wrist radiographs and an endocrine examination. He completed the preoperative orthodontic treatment and orthognathic surgery (sagittal split ramus osteotomy, genioplasty). He was evaluated on the first visit, the preoperative period and the postoperative period with a clinical and radiographic examination. At the first visit, the patient showed moderate overgrowth of the maxilla, severe overgrowth of the mandible, and a subsequential skeletal open bite. After the preoperative orthodontic treatment (preoperative period), the patient showed the same skeletal problem as before and a decompensated dentition for orthognathic surgery. After orthognathic surgery, his profile had improved, but he had still a skeletal openbite tendency because the maxillary orthognathic surgery was not performed. Severe mandibular prognathism with a maxillary overgrowth and anterior open bite should be treated by bimaxillary orthognathic surgery. However, one-jaw orthognathic surgery on the remaining the skeletal open bite tendency was performed for his medical problem and facial esthetics. This subsequential open bite should be resolved with a postoperative orthodontic treatment.