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We report a case of holoprosencephaly with cleft lip and palate. The patient was a 26-year-old woman who presented with malocclusion and masticatory dysfunction. She had undergone cleft surgery, including lip and palatoplasty, and alveolar bone grafting until 18 years of age, but her occlusion was not effectively managed.Facial anomalies included slight orbital hypotelorism and trigonocephaly, severe mid-facial delayedgrowth with a flat nose and a scar from bilateral chiloplasty. Intraoral examination showed malocclusion, cross bite, a severely contracted dental arch, and multiple tooth defects of the maxilla.We successfully treated her malocclusion by the following procedures:(1) alveolar bone graft for implants, (2) orthodontic treatment of the mandible and dental treatment of the upper and lower teeth, (3) sagital splitting ramus osteotomy (SSRO), (4) application of dental implants and vestibular extension with a skin graft, and prosthetic treatment of the maxilla.

A case of adult holoprosencephaly with cleft lip and palate treated by occlusal reconstruction