BackgroundOdontogenic keratocyst is one of the most common benign odontogenic neoplasms with a high recurrence rate. Its resection has the potential to lead to mandibular segmental defects. In this case report, we describe a patient with odontogenic keratocyst who underwent radical resection using a novel distraction osteogenesis (DO) method to reconstruct mandibular segmental defect.Case presentationThis case report describes a 19-year-old woman with odontogenic keratocyst of the mandible that recurred after multiple curettages and eventually necessitated radical resection. Mandibular segmental defect after radical resection was reconstructed using a novel DO method that involved directly contacting the segment ends of the defect without the transport disk. However, the distractor broke during the retention period, and a molding titanium plate was used for fixation. This novel distraction method achieved mandibular reconstruction and restored mandibular function and contour.