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Parry-Romberg syndrome (PRS) refers to a relatively rare dysfunction disease that is characterized by chronic progressive maxillofacial atrophy, especially one side of facial skin, subcutaneous tissue, muscle, and bone. According to the atrophy degree of skin, subcutaneous tissue, and skeleton in the area innervated by the trigeminal nerve, PRS can be classified into mild, moderate, and severe. In general, cases with different severity have specific treatment regimens. For mild and moderate cases, soft tissue augmentation techniques are the optimal strategy for aesthetic reconstruction. In this study, the authors report a 19-year-old female with severe PRS. Considering the severity of the case, a combined surgical and orthodontic treatment was performed, which was involved in alveolar bone augmentation, preoperative and postoperative orthodontic treatment in combination with orthognathic surgery, medpor filling of zygomatic and maxillary complex, free fat grafting, as well as angulus oris and lip trimming. Comprehensive treatment is recommended for severe cases with extensive atrophy of soft tissue and craniofacial bone, obvious deviation of the chin and occlusal plane.