Maxillary duplication is a kind of proliferative neurocristopathy and considered to arise from bifurcation of neural crest elements soon after migration into mandibular arch. Sometimes this malformation is accompanied with oblique facial cleft. Usually this type of maxillofacial malformation requires multiple surgical intervention and the results are far from ideal. It became more troublesome if it had not been properly corrected on time, because secondary deformities could be developed from growth and development of abnormal tissues. This is a case of a 25-year-old-female patient who showed severe facial asymmetry thought to secondary deformity of maxillary duplication and masticatory disturbance due to multiple supernumerary teeth on posterior part right maxilla. We successully treated these deformities through four times of surgery of bone resection, orthodontic treatment, zygomatic and orbital reconstruction, orthognathic surgery and scar revision... ect. So we reported this rare case with review of literatures.