ObjectiveThis study collected and summarized publications related to craniomaxillofacial distraction osteogenesis(DO) from 2000 to 2021, investigated trends in related research, and compared publications from different countries, institutions and journals. The aim is showcasing hotspots and frontiers in the field and providing a reference for future research.BackgroundCraniomaxillofacial DO serves to treat different types of craniomaxillofacial dysplasia and bone defects and deformities. DO can significantly reduce surgical trauma, complications, and recurrence rate compared to conventional surgery. However, there is a lack of bibliometric analyses regarding Craniomaxillofacial DO.MethodsCiteSpace and VOSviewer were used to analyze and visualize 3,141 articles and reviews searching through the Web of Science Core Collection(WOSCC) to obtain publications on craniomaxillofacial DO from 1 January 2000 to 31 December 2021.ResultsIn the last 21 years, there has been a significant increase in the number of publications. The United States, the People's Republic of China, and Italy produce the vast majority of publications. University of Milan and University of Bologna are the most influential in this field. McCarthy JG is the most influential author. Obstructive sleep apnea, TMJ ankylosis and cleft lip and palate are potential research direction in this field.ConclusionFuture research should focus on the precise indications and optimal timing of craniomaxillofacial DO and the evaluation of the long-term outcomes of various modified procedures. This study provides a relatively objective reference for related researchers, medical practitioners, and global health systems.